Respir Case Rep 2020;9(2): 38-42 DOI: 10.5505/respircase.2020.40316

OLGU SUNUMU CASE REPORT

38

Sol Jin1, Jin-Young Lee1, Jehun Kim

2

Since mid-December 2019, a novel coronavirus

(COVID-19) has spread to many countries around

the world, and the number of critically ill patients with

COVID-19 is increasing as the number infections

increase. The optimum treatment and prognosis of

the disease is still unknown. Here, we present the

clinical course and serial computed tomography of a

critically ill Korean patient with COVID-19. The pro-

gression of COVID-19 infection is fast and aggressive,

and no treatment protocol has yet been established.

Additional clinical data are required to determine

whether or not corticosteroid use is clinically benefi-

cial.

Key words: Coronavirus, viral load, corticosteroids,

pneumonia.

Yeni korona virüs (Covid-19) 2019 Aralık ortaların-

dan beri dünyada pek çok ülkede yayılmakta ve

COVID-19'lu ağır hasta sayısı, enfeksiyon sayısı art-

tıkça artmaktadır. Hastalığın optimum tedavisi ve

prognozu halen bilinmemektedir. Burada, COVİD-

19’lu Kore’li bir ağır hastanın klinik seyri ve seri bilgi-

sayarlı tomografi bulgularını sunduk. COVİD-19

enfeksiyonunun progresyonu hızlı ve agresif olup

henüz tedavi protokolü oluşturulmamıştır. Kortikoste-

roid kullanımının klinik yararı olup olmadığını belir-

lemek için ilave klinik verilere ihtiyaç vardır.

Anahtar Sözcükler: Korona virüs, virüs yükü, kortikos-

teroid, pnömoni.

1Department of Infectious Disease, Kosin University Gospel Hospi-

tal, Busan, South Korea

2Department of Pulmonology, Kosin University Gospel Hospital,

Busan, South Korea

1Kosin Üniversitesi Gospel Hastanesi, Enfeksiyon Hastalıkları

Servisi, Busan, Güney Kore

2Kosin Üniversitesi Gospel Hastanesi, Göğüs Hastalıkları

Servisi, Busan, Güney Kore

Submitted (Başvuru tarihi): 17.04.2020 Accepted (Kabul tarihi): 25.04.2020

Correspondence (İletişim): Jin-Young Lee, Department of Infectious Disease, Kosin University Gospel Hospital, Busan, South Korea

e-mail: rejim@hanmail.net

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Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 39

Since mid-December 2019, a novel coronavirus (COVID-

19) has spread to many countries around the world. On

March 11, 2020, the World Health Organization de-

clared a pandemic, identifying COVID-19 as a public

health emergency of international concern (1).

In the second week of March 2020, the number of con-

firmed cases in Korea passed the 8,000 mark, although

accurate counting was a difficult task being experienced

worldwide. The numbers of patients who are being cured

and discharged is increasing over time, although the

number of patients classified as critically ill with COVID-

19 that require ventilator or extra-corporeal membrane

oxygenator support is also gradually increasing. While

data on the epidemiology and clinical manifestations of

the disease is accumulating, clinical data on critically ill

patients is lacking. The clinical course changes in com-

puted tomography (CT) findings and cycle threshold (Ct)

values, which means that the cycle number at which the

fluorescent signal of the reaction crosses the threshold

can help to confirm the characteristics of the disease and

support the creation of a treatment plan for the patients.

To offer an overview of the clinical features of COVID-19

infection, we present a case of a patient in Korea.

CASE

A 78-year-old man with no remarkable past or family

medical history presented with chills, myalgia, cough and

sputum on February 28, 2020. The symptoms and clini-

cal course of the patient are presented in Figure 1.

Upper respiratory tract (URT) and lower respiratory tract

(LRT) specimens were collected from the patient. Naso-

pharyngeal and oropharyngeal swabs were collected for

URT, and sputum was used for the LRT specimen (2).

Quantitative real-time polymerase chain reaction amplifi-

cation was carried out using the AllplexTM 2019-nCoV

assay (Seegen, Seoul, Korea) (3). Ct values were checked

for the RNA-dependent RNA polymerase gene (R gene)

and E gene.

The patient was found to be COVID-19 positive (Ct value:

upper R gene, 19.86; upper E gene, 17.1; lower R gene,

21.92; lower E gene, 18.59) upon examination by the

public health center, and the patient was hospitalized in a

community hospital in Busan, Korea. On day 2, a chest

radiography revealed mild haziness in the left lower lobe,

and a chest CT revealed ground-glass opacities in the left

lower lobe (Figure 2). Shows the serial changes in chest

CT and radiography. The patient was started on lop-

inavir/ritonavir (Kaletra, AbbVie); 2 tablets (lopinavir 200

mg/ritonavir 50 mg) were given orally bid. On day 4,

fever and sputum persisted and loose stool started. Chest

radiography findings worsened, and the patient had a

fever of 39.0°. Accordingly, the ceftriaxone antibiotic was

started on day 5, and piperacillin/tazobactam and

levofloxacin were started on day 6. The patient showed

no improvement.

Figure 1: Clinical course of the patient. Ct values: cycle threshold value; URT: upper respiratory tract; LRT: lower respiratory tract; cefa: cephalosporin;

pip/taz: piperacillin/tazobactam; FiO2: fraction of inspired oxygen

The 2019 Novel Coronavirus Disease (COVID-19) causing Severe ARDS: Serial Computed Tomography Findingse | Lee et al.

40 www.respircase.com

On day 6, the patient was transferred to the hospital in

Busan, Korea. Upon presentation, he had no dyspnea,

but required oxygen supplementation via a nasal cannula

(2L/min). Vital signs: blood pressure, 148/88 mmHg;

pulse rate, 85 beats/min; respiratory rate, 13 breaths/min;

and body temperature, 38.3°C. Laboratory tests revealed

a white blood cell count (WBC) of 5,290/μL, lactate de-

hydrogenase (LDH) of 570 U/dL, and high sensitivity C-

reactive protein (Hs-CRP) of 14.8 mg/dL. Table 1 shows

the detailed blood test results. A follow-up chest CT

showed an increase in the extent of the multifocal

peribronchial ground grass opacity in bilateral lungs and

mild pleural effusion. A sustained dose of lop-

inavir/ritonavir was given and hydroxychloroquine and

antibiotics (meropenem 1g tid, vancomycin 1g bid,

levofloxacin 750 mg qd) were administered. Despite the

use of acetaminophen and non-steroidal anti-

inflammatory drugs (NSAIDs), the high fever persisted.

Although the test for influenza was negative, peramivir

was injected clinically. The patient had no underlying

disease, although a chest CT showed underlying pulmo-

nary fibrosis. Methylprednisolone (0.5 mg/kg) was admin-

istered from day 7 to day 9.

Subsequently, the patient’s dyspnea worsened and an

intubation was performed on day 9. A follow-up COVID-

19 test was positive on day 12 and Ct values were: upper

E gene, 25.43; R gene, 7.45; lower E gene, 18.96; R

gene, 20.69.

Serial laboratory tests and a chest radiography were per-

formed. The chest radiography revealed diffuse consoli-

dation in bilateral lungs. On day 15, a follow-up chest

CT showed diffuse ground-grass opacity, consolidation in

the bilateral lungs and increased interstitial thickening.

The COVID-19 test was still positive (Ct values: upper R

gene, 27.07; upper E gene, 25.95; lower R gene, 21.83;

lower E gene, 20.36).

A tracheostomy was performed and injections of

methylprednisolone (1 mg/kg) were started on day 16.

After starting methylprednisolone, the patient’s oxygen

demand decreased and his chest radiography findings

improved. (Figure 3) The clinical situation was improved

through the use of a higher dose of corticosteroid; how-

ever, on day 21 the methylprednisolone was stopped due

to gastrointestinal bleeding. Close monitoring and opti-

mum supportive care were continued, with measurements

of the Ct value.

Figure 2: Radiologic findings of the patient. Chest radiography on day 2

(A), chest radiography on day 6 (B), Chest radiography on day 15 (C),

chest computed tomography on day 2 (D and G), computed tomogra-

phy on day 6 (E and H), chest computed tomography on 15 (F and I)

Figure 3: Chest radiography after methylprednisolone administration.

Chest radiography on day 16 (A), chest radiography on day 18 (B),

chest radiography on 20 (C)

DISCUSSION

As the number of COVID-19 infections increase world-

wide, the number of critically ill patients with COVID-19

is also increasing. COVID-19 infection is particularly risky

for older patients and those with underlying diseases (4).

The patient in the present study was otherwise healthy,

with no specific past history, aside from the 78 years of

age. At the time of the first diagnosis, CT showed mild

pneumonia, while the peribronchial pneumonic consoli-

dation gradually increased on follow-up CT. Pneumonia

progressed rapidly in a short period.

The progression of pneumonia was apparent on a serial

chest radiography, although it was difficult to determine

the exact degree, and so a follow-up chest CT was per-

formed, showing far more severe lesions than the chest

radiography.

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 41

Table 1: Laboratory test results of the patient

Variables Day 2 Day 6 Day 8 Day 10 Day 12 Day 15 Day 17 Day 19

WBC, /μL 5400 5290 4560 11860 7870 8790 5520 5500

Segment neutrophil, % 66.0 84.9 91.1 96.5 80.0 78.0 74.0 81.0

Lymphocyte, % 2.4 7.5 6.1 1.8 4.0 4.0 7.0 5.0

Eosinophil, % 0.1 0.0 0.0 0.0 0.0 1.0 0.0 0.0

Hemoglobin, g/dL 15.0 13.3 14.3 12.6 14.0 12.3 9.8 10.5

Platelets, ×103/μL 147 104 100 125 79 96 96 117

BUN, mg/dL 16.5 20.8 19.8 29.2 38.6 39.1 62.3 73.9

Creatinine, mg/dL 0.99 0.89 0.78 0.92 0.77 0.68 1.51 1.1

Total bilirubin, mg/dL 0.67 0.77 1.14 1.79 2.79 2.81 2.19 6.00

AST, U/L 19 34 49 34 33 52 32 56

ALT, U/L 20 20 29 27 26 28 25 46

LDH, U/dL 207 570 865 852 885 441 535

Sodium, mEq/L 135.0 125.1 134.2 132.3 136.0 138.0 139.5 142.1

Potassium, mEq/L 4.50 4.17 4.48 4.78 4.27 4.30 4.01 3.96

Chloride, mEq/L 101.0 98.9 104.0 104.5 102.0 102.6 102.6 104.7

Total protein, g/dL 6.8 6.2 5.9 5.4 5.1 5.2 5.4 5.9

Albumin, g/dL 4.1 3.3 3.5 2.8 2.7 2.2 2.2 2.9

Hs-CRP, mg/dl 7.43 14.80 18.30 21.54 20.69 23.76 21.60 4.48

Pro-calcitonin, ng/mL 0.392 0.378

PT, sec 11.4 13.8 17.5 12.8 13.5 14.0 14.1 14.1

PT INR 1.04 1.04 1.41 0.94 1.01 1.06 1.07 1.07

Troponin i 14 370 151 107 72

WBC: white blood cell; BUN: blood urea nitrogen; AST: aspartate aminotransferase; ALT: alanine aminotransferase; LDH: lactate dehydrogenase;

Hs-CRP: high sensitivity C-reactive protein; PT: prothrombin time; INR: international normalized ratio

Another unusual finding was that the patient’s oxygen

demand was not as large as would be expected based on

the chest CT images. On day 6, upon his presentation to

our hospital, the patient had no complaints of dyspnea,

and oxygen saturation was 92~98% with nasal oxygena-

tion of 2L. On day 15, chest CT showed damage to the

entire lung because of the pneumonia, but the fraction of

inspired oxygen on ventilator was 0.5~0.6.

In reports concerning the laboratory tests in early stages

of the disease, lymphocytopenia appears to be a negative

prognostic factor (4). Furthermore, highly elevated LDH

and CRP levels are associated with disease severity (5),

and the patient in the present study showed similar char-

acteristics (Table 1).

Similar to the influenza virus, the amount of COVID-19

output was large in the early phase, and it was confirmed

that the virus output of symptomatic and asymptomatic

people was similar (6,7). In this case, comparing the Ct

values at the time of the first CT (day 2) and at the time of

the last CT (day 15) revealed pneumonia to be more

severe in the CT performed later, although the viral load

decreased. It was thus considered that the viral load was

not related to the patient's lung condition or the severity

of the infection.

In Korea currently, lopinavir/ritonavir and hydroxychloro-

quine are being administered for the treatment of

COVID-19 (6), with antibiotics administered together with

both drugs, considering the possibility of bacterial pneu-

monia. However, it is questionable whether lop-

inavir/ritonavir and hydroxychloroquine are helpful. While

they may help lower the concentrations of the virus, they

have not prevented the rapid clinical progression. These

results are in part consistent with the randomized con-

trolled trials in China comparing the lopinavir/ritonavir

group with a standard care group (8). Despite the medi-

cation, the patient’s fever persisted and the pneumonia

The 2019 Novel Coronavirus Disease (COVID-19) causing Severe ARDS: Serial Computed Tomography Findingse | Lee et al.

42 www.respircase.com

progressed. After peramivir was administered for approx-

imately 6 days, the fever improved.

Although, intravenous glucocorticosteroids were com-

monly used in patients with severe Middle East respiratory

syndrome or severe acute respiratory syndrome, their

effects remain controversial, and their efficacy for the

treatment of COVID-19 is as yet undetermined (9). Inject-

ing methylprednisolone (0.5 mg/kg) on days 7–9 resulted

in no significant changes, while clinical improvement was

noted after injecting methylprednisolone (1 mg/kg) on

day 15. It is not known exactly what it was that affected

the clinical course, but these results may derive from the

dose of methylprednisolone or the timing of the disease

progression. More data on methylprednisolone will be

needed in the future.

CONCLUSION

Severe COVID-19 infection proceeds rapidly, according

to the clinical finding and chest CT findings, although no

effective drug has yet been identified. In such situations,

the use of glucocorticosteroids may be clinically useful.

The number of patients continues to increase worldwide,

while data on the treatment and prognosis of the disease

are still insufficient. Further research is warranted in the

future.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - S.J., J.Y.L., J.K.; Planning and Design - S.J.,

J.Y.L., J.K.; Supervision - S.J., J.Y.L.1, J.K.; Funding -;

Materials -; Data Collection and/or Processing - S.J.;

Analysis and/or Interpretation - S.J., J.K.; Literature Re-

view - J.K.; Writing - S.J.; Critical Review - J.K., J.Y.L

YAZAR KATKILARI

Fikir - S.J., J.Y.L., J.K.; Tasarım ve Dizayn - S.J., J.Y.L.,

J.K.; Denetleme - S.J., J.Y.L., J.K.; Kaynaklar -; Malzeme-

ler -; Veri Toplama ve/veya İşleme - S.J.; Analiz ve/veya

Yorum - S.J., J.K.; Literatür Taraması - J.K.; Yazıyı Yazan -

S.J.; Eleştirel İnceleme - J.K., J.Y.L

REFERENCES

1. Gorbalenya AE, Baker SC, Baric RS, de Groot RJ, Dros-

ten C, Gulyaeva AA, et al. The Species Severe Acute

Respiratory Syndrome-Related Coronavirus: Classifying

2019-nCoV and Naming it SARS-CoV-2. Nat Microbiol

2020; 5;536–44. [CrossRef]

2. World Health Organization. Laboratory Testing for 2019

Novel Coronavirus (2019-nCoV) in Suspected Human

Cases: access date: 19 March 2020. Place of access:

https://www.who.int.

3. Guidlines for the Laboratory Diagnosis of 2019 Novel

Coronavirus(2019-nCoV) in Korea. 1 ed: Central for

Disease Control; 2020. [CrossRef]

4. Yang X, Yu Y, Xu J, Shu H, Xia J, Liu H, et al. Clinical

course and outcomes of critically ill patients with SARS-

CoV-2 pneumonia in Wuhan, China: a single-centered,

retrospective, observational study. Lancet Respir Med

2020; pii: S2213-2600(20)30079-5. [CrossRef]

5. Singhal T. A Review of Coronavirus Disease-2019

(COVID-19). Indian J Pediatr 2020; 87:281-6. [CrossRef]

6. Kim JY, Choe PG, Oh Y, Oh KJ, Kim J, Park SJ, et al.

The First Case of 2019 Novel Coronavirus Pneumonia

Imported into Korea from Wuhan, China: Implication for

Infection Prevention and Control Measures. J Korean

Med Sci 2020; 35:e61. [CrossRef]

7. Zou L, Ruan F, Huang M, Liang L, Huang H, Hong Z, et

al. SARS-CoV-2 viral load in upper respiratory specimens

of infected patients. N Engl J Med 2020; 382:1177–9.

[CrossRef]

8. Cao B, Wang Y, Wen D, Liu W, Wang J, Fan G, et al. A

trial of Lopinavir-Ritonavir in adults hospitalized with se-

vere Covid-19. N Engl J Med 2020; 382:1787-99.

[CrossRef]

9. Russell CD, Millar JE, Baillie JK. Clinical evidence does

not support corticosteroid treatment for 2019-nCoV lung

injury. Lancet 2020; 395:473-5. [CrossRef]

Respir Case Rep 2020;9(2): 43-46 DOI: 10.5505/respircase.2020.98360

OLGU SUNUMU CASE REPORT

43

Tayfun Kermenli1, Cebrail Azar

2

One of the potential complications of acupuncture is

pneumothorax, caused by the entry of the needle to

the visceral pleura and damaging the lung paren-

chyma. Our patient suffered a bilateral pneumotho-

rax and sudden respiratory failure. Our intention in

this study is to offer a reminder of this rare and life

threatening complication with this case report. A 51-

year-old male patient presented to the emergency

department with dyspnea after acupuncture treatment

at a hotel spa center. A physical examination re-

vealed no respiratory sound in either lung. A chest X-

ray revealed a bilateral pneumothorax. The patient

was treated with a bilateral tube thoracostomy.

Key words: Iatrogenic, pneumothorax, acupuncture,

tube thoracostomy, chest tube.

Akupunkturun komplikasyonlarından biri, iğnenin

viseral plevraya ulaşması ve akciğer parankimine

zarar vermesi sonucu oluşan pnömotorakstır. Acil

servise başvuran hastamızda bilateral pnömotoraks

ve ani solunum yetmezliği vardı. Bu olgu sunumu ile

nadir görülen ve hayatı tehdit eden komplikasyonu

hatırlatmayı amaçladık. Elli bir yaşında erkek hasta,

bir otel spa merkezinde akupunktur tedavisi sonrası

acil servise nefes darlığı şikayeti ile başvurdu. Fizik

muayenesinde her iki akciğerde de solunum sesi

alınamadı. Çekilen akciğer grafisinde bilateral pnö-

motoraks saptandı ve hasta bilateral tüp torakostomi

ile tedavi edildi.

Anahtar Sözcükler: İyatrojenik, pnömotoraks, aku-

punktur, tüp torakostomi, göğüs tüpü.

1Department of Thoracic Surgery Clinic, Medicalpark Elaziğ Hos-

pital, Elazığ, Turkey

2Department of Chest Diseases Clinics, Medicalpark Elaziğ Hospi-

tal, Elazığ, Turkey

1Medicalpark Elazığ Hastanesi, Göğüs Cerrahisi Kliniği,

Elazığ

2Medicalpark Elazığ Hastanesi, Göğüs Hastalıkları Kliniği,

Elazığ

Submitted (Başvuru tarihi): 04.11.2019 Accepted (Kabul tarihi): 14.02.2020

Correspondence (İletişim): Tayfun Kermenli, Department of Thoracic Surgery Clinic, Medicalpark Elaziğ Hospital, Elazığ, Turkey

e-mail: tayfunkermenli@gmail.com

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Pneumothorax should be considered first in patients pre-

senting to the emergency department with respiratory

distress. It can be classified as spontaneous or traumatic

pneumothorax, depending on its etiology (1). Iatrogenic

pneumothorax is evaluated in the traumatic pneumotho-

rax group, and its incidence is higher than spontaneous

pneumothorax (2). It is most commonly seen after a trans-

thoracic needle biopsy (24%), subclavian vein catheteri-

zation (22%), thoracentesis (20%), a transbronchial lung

biopsy (10%), pleural biopsy (8%) and positive pressure

ventilation (7%) (3). The incidence of iatrogenic pneumo-

thorax following acupuncture is reported to be 0.87 in

every 1,000,000 acupuncture treatments, and 1.75 in

anatomical regions close to the thoracic cavity (4). We

present the case of a patient who was diagnosed with

bilateral pneumothorax due to respiratory distress in our

emergency department.

CASE

A 51-year-old male patient presented to our emergency

department with dyspnea. A physical examination re-

vealed no respiratory sound in either lung and the patient

was cyanosed. Ecchymotic areas were noted on the skin

in the lateral region of the vertebral column and in the

medial region of the scapula resulting from cup therapy

and acupuncture needles (Figure 1a). The pulse count

was 103/min and the respiratory rate was 17. Oxygen

saturation in the room air was 86% and blood pressure

was 95/45 mmHg. An anamnesis taken from the relatives

of the patient revealed that the patient had undergone

cup therapy and acupuncture in a hotel spa 1 hour pre-

viously for the treatment of back pain. Blood samples

were obtained from the patient for cardiac tests and the

EKG was observed. The pH of the patient's blood gas was

7.23, PCO2: 58 mmHg, PO2: 67 mmHg. Budesonide-

fluticasone nebulization was initiated in the emergency

room due to the patient's history of asthma. An urgent

chest X-ray was performed with a portable bedside device,

revealing bilateral pneumothorax (Figure 1b).

The green colored angiocate (18G) was entered into the

thoracic cavity to decompress the air, and 80 mg predni-

solone was given intravenously to prevent re-expansion

edema in the lung. Bilateral chest tubes were inserted by

a thoracic surgeon, and the patient was subsequently

taken to the intensive care unit. There was minimal air

leak from the left thorax tube. No air leakage was noted

from the thorax drain in the 2 days following the insertion

of the thorax tube, and both lungs were expanded (Figure

2). The right thorax drain was removed on the third day

and the left thoracic drain was removed on the fourth day,

and the patient was discharged as cured. A chest x-ray

was found to be normal in an outpatient clinic control

one month later.

DISCUSSION

Acupuncture and cup therapy are popular alternative

therapies around the world for the treatment of various

ailments, although it has no scientific basis. In many case

reports published in literature, side effects such as pain,

fatigue, bleeding, vasovagal syncope and numbness

resulting from acupuncture have been described (5,6).

More serious complications that have been reported in-

clude pneumothorax, central nervous system injury, infec-

tion, epidural hematoma, subarachnoid hemorrhage,

cardiac tamponade, gallbladder perforation, hepatitis

and death. The most common of these serious complica-

tions is iatrogenic pneumothorax (7,8), as with our case,

and sometimes these complications can have fatal con-

sequences. Re-expansion edema following the insertion

of a thorax tube as a pneumothorax treatment is a signifi-

cant problem. The main reason for decompression in our

patient was to prevent any re-expansion edema that may

occur in the lung. In the present case, steroid treatment

for re-expansion edema prophylaxis was also given, alt-

hough there are limited studies in literature on this topic

(9).

Merchart et al. (10) carried out a study of 97,733 pa-

tients, in which serious side-effects were seen in six pa-

tients. Suicidal ideation in a 36-year-old man diagnosed

with chronic depression, hypertensive crisis in a 66-year-

old female patient with ischemic stroke history, vasovagal

syncope in a 51-year-old man, acute asthma attack in a

62-year-old woman with an asthma history, and two

women, aged 43 and 73, reported that pneumothorax

developed during needle processing.

Figure 1: Patient photo shows acupuncture needle entries (black arrow)

and ecchymotic areas resulting from cup therapy (a), AP Chest X-ray

showing the bilateral pneumothorax (yellow arrowhead) (b)

A Life-threatening Complication of Acupuncture Theraphy: Bilateral Pneumothorax | Kermenli et al.

45 www.respircase.com

Figure 2: Chest X-ray showing thorax tubes in the right (black arrow)

and left hemithorax (arrowhead)

A case report published by Hampton et al. (11) detailed a

43-year-old female patient with chronic neck pain for

which a chest tube insertion was not necessary as a result

of high volume oxygen therapy. The authors also stated

that the area between the posterior edge of the scapula

and the vertebra was a risky site for pneumothorax. Jian

et al. (12) reported a case diagnosed with postmortem

pneumothorax after acupuncture application. As stated in

literature, acupuncture can be sufficiently dangerous to

be potentially lethal. This suggests that acupuncture may

have serious complications and should be performed only

by specialist health professionals.

CONCLUSION

Iatrogenic pneumothorax should be kept in mind in pa-

tients presenting to the emergency department with dysp-

nea. As the first diagnostic method, anamnesis and physi-

cal examination followed by chest radiography are usual-

ly sufficient. Treatment usually requires chest tube inser-

tion, but in the presence of tension pneumothorax, de-

compression with green or gray angiocet (16-18 G) may

be used to relieve the patient's breathing until the chest

tube is inserted. Close follow-up and high volume oxygen

supplementation may considered as a treatment option in

patients with a small pneumothorax.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - T.K., C.A.; Planning and Design - T.K., C.A..;

Supervision - T.K., C.A.; Funding - T.K.; Materials - T.K.,

Y.P.G., N.K.; Data Collection and/or Processing - T.K.,

C.A.; Analysis and/or Interpretation - C.A.; Literature

Review - T.K., C.A.; Writing - T.K.; Critical Review - T.K.,

C.A.

YAZAR KATKILARI

Fikir - T.K., C.A.; Tasarım ve Dizayn - T.K., C.A.; Denet-

leme - T.K., C.A.; Kaynaklar - T.K.; Malzemeler - T.K.;

Veri Toplama ve/veya İşleme - T.K., C.A.; Analiz ve/veya

Yorum - C.A.; Literatür Taraması - T.K., C.A.; Yazıyı Ya-

zan - T.K.; Eleştirel İnceleme - T.K., C.A.

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puncture treatments in Taiwan. Acupunct Med 2019;

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5. Öncel M, Tezcan B, Sunam GS. Iatrogenic bilateral

pneumothorax after acupuncture. Respir Case Rep 2013;

2:158-9. [CrossRef]

6. Özgen E, Bozkaya Yücel B, Yücel M, Güzel M, Gürgün

KE, Yürümez Y. A rare complication after acupuncture:

pneumothorax. Geleneksel ve Tamamlayıcı Anadolu

Tıbbı Dergisi 2019; 1:1-4.

7. Tucciarone M, Taliente S, Gómez-Blasi Camacho R,

Souviron Encabo R, González-Orús Álvarez-Morujo R.

Extensive pyomyositis of prevertebral muscles after acu-

puncture: Case report. Turk J Emerg Med 2019;

19:113-4. [CrossRef]

8. Huisma F, Konrad G, Thomas S. Pneumothorax after ac-

upuncture. Can Fam Physician 2015; 61:1071-3.

9. Kepka S, Lemaitre L, Marx T, Bilbault P, Desmettre T. A

common gesture with a rare but potentially severe com-

plication: Re-expansion pulmonary edema following

chest tube drainage. Respir Med Case Rep 2019;

27:100838. [CrossRef]

10. Melchart D, Weidenhammer W, Streng A, Reitmayr S,

Hoppe A, Ernst E, et al. Prospective investigation of ad-

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Cilt - Vol. 9 Sayı - No. 2 46

verse effects of acupuncture in 97 733 patients. Arch In-

tern Med 2004; 164:104-5. [CrossRef]

11. Hampton DA, Kaneko RT, Simeon E, Moren A, Rowell S,

Watters JM. Acupuncture-Related Pneumothorax. Med

Acupunct 2014; 26:241-5. [CrossRef]

12. Jian J, Shao Y, Wan L, Zhang M, Liu N, Zhang J, et.al.

Autopsy diagnosis of acupuncture-induced bilateral ten-

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puted tomography: A case report. Medicine (Baltimore)

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Respir Case Rep 2020;9(2): 47-51 DOI: 10.5505/respircase.2020.86570

OLGU SUNUMU CASE REPORT

47

Dilek Erdem1, İrem Karaman

2, Adem Dirican

3, Şevket Özkaya

4

A phantom tumor of the lung is a localized collection

of transudative interlobar pleural fluid in the settings

of decompensated congestive heart failure that re-

sembles a lung neoplasia on chest X-ray, but that

vanishes after the appropriate diuretic therapy. Here,

we report on a phantom tumor with a real lung neo-

plasia in the same location, both of which were con-

tributing to the complaints and the clinical picture of

the patient. Although phantom tumors are rare and

should not be misdiagnosed as masses, it is worth

making a further diagnosis and being cautious for

possible neoplasia in the same setting. Since the co-

existence of heart failure and lung cancer significantly

changes the management and follow-up procedure

of the patient, a multidisciplinary approach and

elaborative work by the pulmonologist, oncologist

and cardiologist is required on a case-by-case basis.

In such patients, cardiotoxic chemotherapy agents

should be avoided as a treatment to prevent the

progression of heart failure. The present case con-

firms the rare possibility of the co-occurrence of lung

neoplasia and phantom tumor, and recommends the

delicate management of such patients.

Key words: Phantom tumor, lung cancer, congestive

heart failure.

Akciğerde fantom tümörü, dekompanse konjestif kalp

yetmezliği durumunda lokalize transüdatif interlobar

plevral sıvı toplanması sonucu görülür ve akciğer

grafisinde akciğer neoplazisine benzer kitle görünü-

müne sebep olur, ancak uygun diüretik tedavisinden

sonra kaybolur. Burada, aynı lokasyonda gerçek bir

akciğer neoplazisi ve fantom tümörünün görüldüğü

ve bu iki durumun hastanın şikayetlerine ve klinik

tablosuna neden olduğu bir olgu bildirdik. Fantom

tümörleri nadir olarak görülür ve akciğerdeki kitle

görünümüyle karıştırılmamalıdır, ancak olası durum-

larda akciğer kanseri için temkinli olmak ve ileri test-

leri yapmak gerekir. Kalp yetmezliği ve akciğer kanse-

ri birlikteliği hastanın yönetim ve takip prosedürünü

önemli ölçüde değiştirdiğinden, münferit olgu bazın-

da pulmonolog, onkolog ve kardiyologtan oluşan

multidisipliner bir yaklaşım ve özenli bir çalışma ge-

reklidir. Bu hastalarda kalp yetmezliğinin ilerlemesini

önlemek için tedavi sırasında kardiyotoksik kemote-

rapi ajanlarından kaçınılmalıdır. Bu olgu, akciğer

neoplazisinin fantom tümörü ile nadir olarak ortaya

çıkma olasılığını doğrulamaktadır ve bu tür hastaların

hassas yönetimine dikkat çekmektedir.

Anahtar Sözcükler: Fantom tümörü, akciğer kanseri,

konjestif kalp yetmezliği.

1Department of Internal Medicine, Bahçeşehir University Faculty of

Medicine, Division of Medical Oncology; Istanbul, Turkey; De-

partment of Medical Oncology, VM Medical Park Samsun Hospi-

tal, Samsun, Turkey

2Bahcesehir University, School of Medicine, Istanbul, Turkey

3Department of Pulmonary Medicine, Samsun Medical Park Hos-

pital, Samsun, Turkey

4Department of Pulmonary Medicine, Faculty of Medicine,

Bahçeşehir University, Istanbul, Turkey

1Bahçeşehir Üniversitesi Tıp Fakültesi, Tıbbi Biyoloji Anabilim

Dalı, İstanbul

2Bahçeşehir Üniversitesi Tıp Fakültesi, İstanbul

3Samsun Medical Park Hastanesi Göğüs Hastalıkları

Bölümü, Samsun

4Bahçeşehir Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları

Anabilim Dalı

Submitted (Başvuru tarihi): 29.02.2020 Accepted (Kabul tarihi): 16.04.2020

Correspondence (İletişim): Dilek Erdem, Department of Internal Medicine, Bahçeşehir University Faculty of Medicine, Division of

Medical Oncology; Istanbul, Turkey; Department of Medical Oncology, VM Medical Park Samsun Hospital, Samsun, Turkey

e-mail: dilekgurgenyatagi@yahoo.com

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A phantom tumor of the lung is a localized collection of

transudative interlobar pleural fluid in the settings of de-

compensated congestive heart failure that resembles a

lung neoplasia on chest X-ray, but which vanishes after

the appropriate diuretic therapy (1). Since it is a rare

condition, awareness of this form of pleural effusion is

crucial in the differential diagnosis of pulmonary masses

(2). Here, we reported on a case with a phantom tumor

and with a real lung tumor beneath it, in which both

masses contributed to the appearance of a pulmonary

mass in a chest X-ray.

CASE

A 78-year-old male patient with no history of hyperten-

sion or diabetes mellitus presented to the clinic with com-

plaints of exertional dyspnea, dry cough, paroxysmal

nocturnal dyspnea and angina. The patient had a history

of three coronary angioplasty operations, and he was

using clopidogrel, acetyl salicylic acid, atorvastatin,

metoprolol, spironolactone and escitalopram at the time

of admission.

In his first examination, his vitals were within the normal

limits, with heart rate of 64 and blood pressure of

120/80 mmHg. After an echocardiography examination,

he was diagnosed with grade III mitral insufficiency, ac-

companying grade III tricuspid insufficiency and pulmo-

nary artery hypertension (pulmonary artery pressure=65

mmHg). A chest examination revealed diminished pulmo-

nary sounds and extensive rales in the base of both lungs.

A pulmonary function test revealed a restrictive pattern. In

posteroanterior chest X-ray, an enlarged cardiac silhou-

ette with a bulky opacity in the right lung that was sharply

marginated was found, consistent with a phantom tumor.

(Figure 1) In a thorax HRCT, a phantom tumor in the

interlobar fissure was seen, as a large mass. In addition,

pericardial effusion with bilateral pleural fluids in the

oblique fissure and in the pleural cavity was also reported

(Figure 2). Laboratory tests showed no abnormal findings,

and the sample from pleural fluid was consistent with

transude. The diagnostic hypothesis was decompensated

congestive heart failure in the presence of a phantom

tumor of the lung.

Following hospitalization, no improvement was seen in

the symptoms with IV diuretic treatment. The chest X-ray

was repeated after the first 24 hours, but the phantom

tumor was still present. Consequently, a thorax CT with

PET scan was performed due to a suspicion of lung neo-

plasia. In the PET scan, a 45x39 mm hypermetabolic

mass was observed in the middle lobe of the right lung

under the phantom tumor (Figure 3A). The phantom

tumor (Figure 3B) did not indicate any FDG-uptake, while

its right side demonstrated an increased FDG-uptake.

Based on the findings, a tru-cut biopsy procedure was

planned. The patient was discharged following the biopsy,

but returned to the emergency room one week later with

decompensated atrial fibrillation and ST elevation. The

patient was admitted to the cardiovascular ICU and an

amiodarone infusion was started. After one week of

treatment with anti-arrhythmics and diuretics, the symp-

toms improved, and the patient was discharged until the

results of the biopsy came through.

Figure 1: In the posteroanterior chest X-ray, enlarged cardiac silhouette

with a bulky opacity in right lung which was sharply marginated was

found, which was consistent with phantom tumor

Figure 2: In thorax CT, a phantom tumor in the interlobar fissure in the

appearance of a big mass has reported. A pericardial effusion with

bilateral pleural fluids in oblique fissure and in pleural cavity has also

reported

Phantom Tumor with Real Tumor of The Lung: An Unreported Case | Erdem et al.

49 www.respircase.com

Figure 3: In PET scan, 45x39 mm hypermetabolic mass observed in

middle lobe of the right lung under hypometabolic phantom tumor with

bilateral pleural fluid (A), The phantom tumor did not indicate any FDG-

uptake, while its right side demonstrated an increased FDG-uptake

which belongs to the tumor tissue (B), Hypermetabolic metastatic lesions

were seen in bilateral adrenal glands (C)

Figure 4: In chest x-ray after four months of immunotherapy and cardiac

monitoring revealed the disappearance of phantom tumor with the real

tumor under it

Figure 5: In PET-CT scan results after four months of treatment. Phan-

tom tumor totally disappeared, and the PET scan revealed diminished

pleural effusion with decreased hypermetabolic focuses

The biopsy results reported grade II adenocarcinoma of

the lung with metastasis to the bilateral adrenal glands

(Figure 3C) and increased PD-L 1 levels. At the same time,

a left ventricular thrombus was identified upon admission

to emergency room with night sweats, hemoptysis, angina

and fever. After a final evaluation, a real tumor of the

lung beneath the phantom tumor was diagnosed. The

patient was prescribed with low molecular weight heparin

for the left ventricular thrombus. The patient was hemo-

dynamically stabilized, after which immunotherapy with

nivolumab was started. After the two months of treatment,

a chest X-ray and PET scan revealed a diminished pleural

effusion with decreased hypermetabolic focus (Figure 4

and 5). In the fifth month, the patient’s general status and

symptoms have improved, and the exertional dyspnea

and cough have resolved.

DISCUSSION

Literature contains few reports of phantom tumors of the

lung, all of which resolved after the appropriate treatment

(1-6). The reported cases occurred predominantly in men,

and commonly involved a transverse fissure of right lung

(2). When the transudative fluid in the extravascular space

is unable to pass into the pleural lymphatics properly due

to pleuritis in decompensated heart failure, phantom

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 50

tumors may appear, especially in the right lung, due to

the higher hydrostatic pressure on the right side of the

lung (5). A local increase in elastic recoil by the neighbor-

ing partially atelectatic lung, which is yielding the accu-

mulation of fluid, is also considered responsible for the

pathogenesis.

In most cases, phantom tumors appear in settings of

congestive heart failure, although there may be other

conditions, such as hypoalbuminemia, renal insufficiency

or pleuritis (2). Accordingly, in the presence of accumu-

lated pleural fluid within the fissure, a differential diagno-

sis should include transudates due to renal failure, exu-

dates due to malignant effusions, benign asbestos-related

effusions, parapneumonic effusions and hemothorax,

chylothorax and fibrous tumors of the visceral pleura of

the interlobar fissure (1).

Here, we reported on a phantom tumor with a real lung

neoplasia in the same location, both of which contributed

to the complaints and the clinical picture of the patient.

As signs of congestive heart failure, the reported pericar-

dial and pleural effusions in CT-scans led us to believe

that decompensation had caused an accumulation of

pleural fluid in the vascular spaces and the appearance

of the mass. Although most case reports suggested no

further investigation, and indicate an immediate launch of

treatment (2,4), the clinical picture of the patient should

be carefully evaluated for further possible diagnoses, as

in the presented case. We found in the present study that

both pleural interlobar fluid and lung neoplasia were

causing the symptoms and the chest X-ray appearance.

The presented case demonstrates the low possibility of the

existence of lung neoplasia in an acute exacerbation of

congestive heart failure settings.

The phantom tumors seen in congestive heart failure

patients are a particularly important concern if the patient

has a concurrent disease requiring treatment. In this case,

the co-existence of heart failure and lung cancer signifi-

cantly changed the management and follow-up of the

patient. In such cases, the treatment course depends on

crucial considerations and a multidisciplinary approach

involving a pulmonologist, oncologist and cardiologist.

Furthermore, each treatment procedure should be de-

signed on a case-by-case basis. In such patients, cardio-

toxic chemotherapy agents should be avoided in the

course of treatment to prevent the progression of heart

failure. Furthermore, a critical evaluation by a cardiolo-

gist should be carried out at every stage of treatment.

CONCLUSION

Phantom tumors are pseudo-tumors of the lung that are

seen on chest X-rays in the setting of congestive heart

failure. They often require no further investigation since,

since they can resolve with appropriate diuretic treatments.

However, a tight follow-up is required to ensure the dis-

appearance of the mass. As in this the present case, there

is the possibility of a real neoplasia of the lung being

missed if the tumor appearance does not resolve follow-

ing treatment. For this purpose, further investigations and

imaging techniques, as well as biopsy procedures should

be performed to ensure an accurate diagnosis. If lung

cancer is discovered in such patients, the cardiologist and

oncologist should collaborate in the establishment of an

appropriate treatment while preventing the possible car-

diac complications of chemotherapeutic agents. The

present case confirms the rare possibility of the co-

occurrence of lung neoplasia with a phantom tumor, and

recommends the delicate management of such patients.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - D.E., İ.K., A.D., Ş.Ö.; Planning and Design -

D.E., İ.K., A.D., Ş.Ö.; Supervision - D.E., İ.K., A.D., Ş.Ö.;

Funding - A.D., Ş.Ö., D.E.; Materials - Ş.Ö., D.E.; Data

Collection and/or Processing - D.E., İ.K.; Analysis and/or

Interpretation - D.E., İ.K.; Literature Review - D.E., Ş.Ö.;

Writing - D.E., İ.K., A.D., Ş.Ö.; Critical Review - D.E., İ.K.,

A.D., Ş.Ö.

YAZAR KATKILARI

Fikir - D.E., İ.K., A.D., Ş.Ö.; Tasarım ve Dizayn - D.E.,

İ.K., A.D., Ş.Ö.; Denetleme - D.E., İ.K., A.D., Ş.Ö.; Kay-

naklar - A.D., Ş.Ö., D.E.; Malzemeler - Ş.Ö., D.E.; Veri

Toplama ve/veya İşleme - D.E., İ.K.; Analiz ve/veya Yo-

rum - E D.E., İ.K., N.K.; Literatür Taraması - D.E., Ş.Ö.;

Yazıyı Yazan - D.E., İ.K., A.D., Ş.Ö.; Eleştirel İnceleme -

D.E., İ.K., A.D., Ş.Ö.

REFERENCES

1. Lozo M, Lozo Vukovac E, Ivancevic Z, Pletikosic I. Phan-

tom tumor of the lung: localized interlobar effusion in

congestive heart failure. Case Rep Cardiol 2014;

2014:207294. [CrossRef]

Phantom Tumor with Real Tumor of The Lung: An Unreported Case | Erdem et al.

51 www.respircase.com

2. Tesloianu DN, Chioarta M, Corduneanu D, Ignat AM,

Petris AO, Tesloianu A. Does phantom tumor really exist?!

Maedica (Buchar) 2017; 12:281-5.

3. Ardic I, Yarlioglues M, Celik A, Kaya MG. Vanishing or

phantom tumor of the lung. Tex Heart Inst J 2010;

37:730-1.

4. Melo BS, Serra AC, Belo MT, Belo Neto E, Melo SM.

Phantom tumor of the lung. Rev Assoc Med Bras (1992)

2012; 58:517-8. [CrossRef]

5. Mikaeili H, Mehdizadeh Baghbani J. Multiple phantom

tumor of the lung: A complex appearance resolving with

appropriate intervention. Tanaffos 2016; 15:243-5.

6. Shaikh S, Shaikh S. Pleural effusion resembling a lung

tumor: phantom tumor of the lung. Egypt J Intern Med

2016; 28:174-5. [CrossRef]

Respir Case Rep 2020;9(2): 52-55 DOI: 10.5505/respircase.2020.82435

OLGU SUNUMU CASE REPORT

52

İbrahim Güven Çoşğun1, Düriye Öztürk

2, Çiğdem Özdemir

3, Şule Çilekar

1, Filiz Yavaşoğlu

4,

Ersin Günay1

Diffuse Large B-cell lymphoma is a subgroup of non-

Hodgkin's lymphoma. Lymphoma with endobronchial

pulmonary involvement is a rarely reported condition.

A 64-year-old male patient was admitted to our clinic

with complaints of swellings in the neck and in the

bilateral upper limbs, along with shortness of breath.

A computed tomography of the thorax revealed dif-

fuse mediastinal enlargement with a superior vena

cava obstruction, and a huge right hilar mass for-

mation obliterating the anterior segment of the right

upper lobe. A diagnostic fiberoptic bronchoscopy

showed mucosal irregularity and an endobronchial

lesion on the anterior segment of the right upper lobe.

A diagnosis of endobronchial lymphoma was made

following bronchial biopsies.

Key words: Diffuse Large B-cell Lymphoma, Endob-

ronchial, Non-Hodgkin.

Diffüz büyük B hücreli lenfomalar non-Hodgkin len-

fomaların subtipini oluştururlar. Lenfomaların endob-

ronşial invazyonu oldukça nadirdir. Nefes darlığı,

boyun ve üst ekstremitede şişlik şikayeti olan 64 ya-

şında erkek hasta kliniğimize başvurdu. Toraks to-

mografisinde üst mediastende büyümüş lenf nodları,

vena cava süperiorda daralma ve sağ hiler büyümüş

lenf nodları izlendi. Fiberoptik bronkoskopisinde sağ

akciğer üst lob anterior segmentte endobronşiyal

lezyon izlendi. Fiberoptik bronkoskopik biyopsi ile

endobronşiyal lenfoma tanısı doğrulandı.

Anahtar Sözcükler: Diffüz Büyük B-Hücreli, Lenfoma,

Endobronşiyal Non-Hodgkin.

1Department of Pulmonology, Afyonkarahisar University of Health

Sciences, Medical Faculty, Turkey

2Department of Radiation Oncology, Afyonkarahisar University of

Health Sciences, Medical Faculty, Turkey

3Department of Pathology, Afyonkarahisar University of Health

Sciences, Medical Faculty, Turkey

4Department of Hematology, Afyonkarahisar University of Health

Sciences, Medical Faculty, Turkey

1Afyonkarahisar Sağlık Bilimleri Üniversitesi, Tıp fakültesi

Hastanesi, Göğüs Hastaliklari Ana Bilim Dalı,Afyonkarahisar

2Afyonkarahisar Sağlık Bilimleri Üniversitesi, Tıp fakültesi

Hastanesi, Radyasyon Onkolojisi Ana Bilim

Dalı,Afyonkarahisar

3Afyonkarahisar Sağlık Bilimleri Üniversitesi, Tıp fakültesi

Hastanesi, Patoloji Ana Bilim Dalı,Afyonkarahisar

4Afyonkarahisar Sağlık Bilimleri Üniversitesi, Tıp fakültesi

Hastanesi, Hematoloji Ana Bilim Dalı, Afyonkarahisar

Submitted (Başvuru tarihi): 27.09.2019 Accepted (Kabul tarihi): 07.01.2020

Correspondence (İletişim): İbrahim Güven Çoşğun, Department of Pulmonology, Afyonkarahisar University of Health Sciences,

Medical Faculty, Turkey

e-mail: dr_guven@hotmail.com

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Pulmonary lymphomas may occur as a primary pulmo-

nary malignancy or as a secondary manifestation derived

from systemic lymphoma. Primary pulmonary lymphomas

are defined as malignant lymphomas that originate in the

pulmonary parenchyma or bronchi, with or without hilar

lymph node involvement and the absence of any other

tissue localization for 3 months following the diagnosis

(1). In literature, the prevalence of primary pulmonary

lymphoma has been reported in 0.5–1% of malignancies,

accounting for <1% of all malignant lymphomas (2).

Primary pulmonary lymphoma is rare, as pulmonary tissue

contains less lymphoid tissue than other sites. The most

common type of primary pulmonary lymphoma is muco-

sa-associated lymphoid tissue (MALT) lymphoma (2).

Secondary pulmonary lymphoma is more common than

primary pulmonary lymphoma. Diffuse Large B-cell lym-

phoma (DLBCL) is a subgroup of non-Hodgkin's lympho-

ma. Lymphoma with pulmonary involvement has been

rarely reported, and endobronchial lymphoma is an ex-

ceptional finding. Mediastinal lymphoma has an aggres-

sive behavior due to the close relationship with blood

vessels (3). We present here the case of a patient with

endobronchial lymphoma, diagnosed with a bronchial

biopsy.

CASE

A 64-year-old male patient presented with a history of

neck and bilateral upper limb swelling, as well as short-

ness of breath for the past 2 weeks. Upon physical exam-

ination, swelling to the neck and the bilateral upper ex-

tremities, and bilateral dilated superficial veins on the

chest were observed. The patient was using inhaler drugs

for chronic obstructive pulmonary disease (COPD). The

laboratory findings (hemogram and blood biochemistry)

were within normal limits. A postero-anterior chest X-ray

showed mediastinal and right hilar enlargement (Figure

1A). A contrast-enhanced computed tomography (CT) of

the thorax revealed diffuse mediastinal enlargement with

a superior vena cava obstruction and a right hilar mass

formation obliterating the anterior segment of the right

upper lobe (Figure 1C and D). A diagnostic fiberoptic

bronchoscopy was performed, revealing mucosal irregu-

larity and an endobronchial lesion in the anterior seg-

ment of the right upper lobe (Figure 1B). A histopatholog-

ical examination revealed diffuse large B-cell lymphoma

(DLBCL) infiltration. A bone marrow aspiration biopsy was

negative for lymphoma infiltration. Immunohistochemistry

showed an expression of vimentin, and a clustering of

differentiation 20 (CD20), CD45 and CD19, but not of

CD3, CD15 or CD30 (Figure 2). Due to the bulky lymph

node and superior vena cava syndrome, the patient ini-

tially received a 300 cGy dose fraction per day, with a

total dose of 3000cGy radiation. A chemotherapy proto-

col with rituximab, cyclophosphamide, doxorubicin hy-

drochloride, vincristine and prednisolone (R-CHOP) was

started for the treatment of the patient.

DISCUSSION

We report here on a rare case of lymphoma with endo-

bronchial involvement, diagnosed by bronchoscopic

biopsy. Lymphoma refers to malignant tumors that origi-

nate in the lymph nodes or in other lymphoid tissues (4).

DLBCL is a subgroup and an aggressive form of non-

Hodgkin lymphoma, and accounts for approximately 30%

of all lymphomas (5). In a retrospective study of 1,221

patients with extranodal DLBCL, Takahashi et al. (6) re-

ported lung involvement in 3.7% of cases. Lymphoma

may present as mediastinal lymphadenopathy and as an

isolated mediastinal mass. Pulmonary lymphoma with

endobronchial involvement usually presents as a bulky

lesion in the mediastinum, and is associated with symp-

toms related to local compression (7). The pathogenesis

of endobronchial Hodgkin lymphoma is unclear. The

mechanism is presumed to be a contiguous transmural

spread from the adjacent lymph nodes, but may also

arise in mucosa-associated lymphoid tissue (8). The dif-

ferential diagnosis of lymphoma with pulmonary involve-

ment includes mycobacterial and fungal infections, We-

gener granulomatosis, Langerhans cell histiocytosis and

other pulmonary malignancies (9). Endobronchial lym-

phoma is classified into two types, according to the pat-

tern of involvement: diffuse submucosal infiltration (type I),

and localized solitary mass (type II) (10). Type I includes

diffuse submucosal infiltrates originating from hematog-

enous or lymphangitic spread in the presence of systemic

lymphoma; while type II includes airway involvement by a

localized mass due to the direct spread of lymphoma

from the adjacent lymph nodes, or arising out of bron-

chus-associated lymphoid tissue (11). The pattern of in-

volvement in the current patient was a localized solitary

mass.

Routinely, mediastinoscopy or other invasive surgical

procedures are preferred for the obtaining of an exact

diagnosis, although Endobronchial Ultrasound-

transbronchial needle aspiration (EBUS-TBNA) can be

used as an alternative option (12). Due to the difficulties

in confirming a diagnosis of the lymphoma subtype from

a small volume specimen in EBUS-TBNA, the recommen-

Diffuse Large B-cell Lymphoma Exhibiting Endobronchial Involvement: A Case Report | Coşğun et al.

54 www.respircase.com

dation of EBUS-TBNA for the evaluation of suspected

lymphoma is controversial. The diagnostic sensitivity of

EBUS-TBNA for lymphoma is lower than for lung cancer,

although to avoid invasive surgical procedures, EBUS-

TBNA may still be considered as the initial investigative

technique for suspected lymphoma (13). Recently, EBUS-

TBNA has been reported to be useful in the diagnosis of

mediastinal lymphoma. Of 1,471 cases that underwent

EBUS-TBNA for isolated mediastinal masses and/or lym-

phadenopathy, 27 patients (1.8 %) were diagnosed with

lymphoma (14). In the present case, endobronchial lym-

phoma was diagnosed via bronchoscopic biopsy rather

than EBUS-TBNA. By performing a fiberoptic bronchos-

copy and obtaining biopsy samples showing tumor infil-

tration on the right upper lobe of the right lung, the diag-

nosis of lymphoma was made without the use of EBUS-

TBNA.

Figure 1: Chest X-ray showing enlargement of the right mediastinal and

hiler region (A), Bronchoscopic image endobronchial vegetation right

upper lobe of the right lung (B), Thorax tomography enhancing lymph

node with superior vena cava obstruction (C), Enhancing right hilar

lymph node (D)

Figure 2: Hematoxylin and eosin (H&E) staining of the lung biopsy

demonstrate diffuse proliferation (H&EX40) (A), Lung biopsy demonstrate

diffuse proliferation, high power (H&EX200) (B), Immunohistochemistry

staining of the lung biopsy shows positive for CD20 (X200) (C)

Lymphoma with endobronchial involvement is rare, and a

diagnosis of endobronchial lymphoma with bronchoscop-

ic biopsy has been rarely reported in literature. A system-

atic, careful examination of the bronchial system for a

diagnosis of mediastinal mass lesions is crucial. Broncho-

scopic biopsies for a diagnosis of endobronchial lym-

phoma with huge mediastinal lesions may contribute to

histological diagnoses through the use of minimally inva-

sive procedures, avoiding the need for EBUS or mediasti-

noscopic interventions.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.; Plan-

ning and Design - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.;

Supervision - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.;

Funding - İ.G.Ç., E.G.; Materials - İ.G.Ç., E.G.; Data

Collection and/or Processing - İ.G.Ç., E.G.; Analysis

and/or Interpretation - İ.G.Ç., E.G.; Literature Review -

İ.G.Ç., E.G.; Writing - İ.G.Ç., E.G.; Critical Review -

İ.G.Ç., E.G.

YAZAR KATKILARI

Fikir - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.; Tasarım ve

Dizayn - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.; Denet-

leme - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.; Kaynaklar

- İ.G.Ç., E.G.; Malzemeler - İ.G.Ç., E.G.; Veri Toplama

ve/veya İşleme - İ.G.Ç., E.G.; Analiz ve/veya Yorum -

İ.G.Ç., E.G.; Literatür Taraması - İ.G.Ç., E.G.; Yazıyı

Yazan - İ.G.Ç., E.G.; Eleştirel İnceleme - İ.G.Ç., E.G.

REFERENCES

1. Dong Y, Zeng M, Zhang B, Han L, Liu E, Lian Z, et al.

Significance of imaging and clinical features in the differ-

entiation between primary and secondary pulmonary

lymphoma. Oncol Lett 2017; 14:6224-30. [CrossRef]

2. Pajares V, Torrego A, Granell M, Szafranska J, Mozos A,

Puzo C. Recurrent endobronchial diffuse large B-cell

lymphoma. Diagnosed by cryoprobe. Arch Bron-

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of superior vena cava syndrome: diagnosis with percuta-

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Kartaloğlu Z. Hodgkin's lymphoma with endobronchial

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 55

involvement: a case report. Respir Case Rep 2012; 1:19-

23. [CrossRef]

5. Cabuk D, Gullu YT, Basyigit I, Acikgoz O, Uygun K, Yild-

iz K, et al. Multifocal extranodal involvement of diffuse

large B-cell lymphoma. Case Rep Pulmonol 2013;

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6. Takahashi H, Tomita N, Yokoyama M, Tsunoda S, Yano

T, Murayama K, et al. Prognostic impact of extranodal

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imab era. Cancer 2012; 118:4166-72. [CrossRef]

7. Shimada M, Fukuda M, Horio K, Suyama T, Kitazaki T,

Hashiguchi K, et al. Primary Mediastinal Large B-cell

Lymphoma Exhibiting Endobronchial Involvement. Intern

Med. 2016; 55:3147-50. [CrossRef]

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dobronchial Hodgkin's disease with pneumomediastinum.

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Rowe JM, Yigla M. Non-Hodgkin's lymphoma presenting

as an endobronchial tumor: report of eight cases and lit-

erature review. Am J Hematol 2008; 83:416-9.

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Igawa S, et al. Endobronchial involvement of mantle cell

lymphoma: A case report. Respir Med Case Rep 2016;

19:77-9. [CrossRef]

12. Yang H, Zhao H, Garfield DH, Teng J, Han B, Sun J. En-

dobronchial ultrasound-guided transbronchial needle as-

piration in the diagnosis of non-lymph node thoracic le-

sions. Ann Thorac Med 2013; 8:14-21. [CrossRef]

13. Steinfort DP, Conron M, Tsui A, Pasricha SR, Renwick WE,

Antippa P, et al. Endobronchial ultrasound-guided trans-

bronchial needle aspiration for the evaluation of suspect-

ed lymphoma. J Thorac Onchol 2010; 5: 804-9.

[CrossRef]

14. Yilmaz A, Ozturk A. Endobronchial ultrasound-guided

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of lymphoma: don't give up! Bratisl Lek Listy 2018;

119:503-8. [CrossRef]

Respir Case Rep 2020;9(2): 56-59 DOI: 10.5505/respircase.2020.68542

OLGU SUNUMU CASE REPORT

56

Emine Afşin1, Ayperi Ozturk

2

Karsinoid tümörler nöroendokrin tümör grubunda

değerlendilirler. Gastrointestinal traktusta yerleşimi

daha sık iken nadiren bronşial orjinli de olabilir.

Akciğer kanserlerinin %0,5-1’ini oluşturur. Genellikle

genç yaş grubunda görülür. Yetmiş sekiz yaşında,

erkek hastada rastlantısal olarak istenilen toraks

bilgisayarlı tomografide trakeal polipoid lezyon izlen-

di. Atipik karsinoid tümör olarak tanı alan hasta ileri

yaşı ve ağır KOAH' ı (FEV1 %25) olması nedeniyle

inoperabl kabul edildi. Radyolojik olarak ekstralumi-

nal uzanımı ve metastatik lenf nodu olmaması üzerine

Nd: YAG lazer ve ardından lezyon köküne koterizas-

yon uygulandı. Endobronşial tedavi yaklaşımı ile tam

kür sağlandı.

Anahtar Sözcükler: Atipik karsinoid tümör, endobron-

şial tedavi, trakea.

Carcinoid tumors are evaluated in the

neuroendocrine tumor group. While they are more

common in the gastrointestinal tract, they may rarely

be of bronchial origin. They account for 0.5–1% of

all lung cancers, and are usually seen in the young

age group. A 78-year-old male patient with a

tracheal polypoid lesion was observed at thorax CT.

The patient was diagnosed with an atypical carcinoid

tumor, and was considered to be inoperable due to

his advanced age and severe COPD (FEV1 25%).

Due to the extraluminal extension and no metastatic

lymph nodes identified radiologically, an Nd: YAG

laser was applied and the root of the lesion was

cauterized. Complete cure was achieved with an

endobronchial treatment approach.

Key words: Atypical carcinoid tumor, endobronchial

therapy, trachea.

1İzzet Baysal Devlet Hastanesi, Göğüs Hastalıkları Kliniği, Bolu

2Ankara Atatürk Göğüs Hastalıkları ve Göğüs Cerrahisi Eğitim ve

Araştırma Hastanesi, Girişimsel Pulmonoloji Kliniği, Ankara

1Department of Chest Diseases, İzzet Baysal State Hospital,

Bolu, Turkey

2Department of Interventional Pulmonology, Ankara Ataturk

Chest Diseases and Chest Surgery Training and Research

Hospital, Health Science University, Ankara, Turkey

Başvuru tarihi (Submitted): 06.10.2019 Kabul tarihi (Accepted): 06.12.2019

İletişim (Correspondence): Emine Afşin, İzzet Baysal Devlet Hastanesi, Göğüs Hastalıkları Kliniği, Bolu

e-mail: emineafsin@yahoo.com

RES

PIR

ATO

RY

CA

SE R

EPO

RTS

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 57

Bronşial karsinoid tümörler bronş muköz bezlerindeki

Kultchitsky hücrelerinden köken alırlar. Düşük malignite

potansiyeline sahip, yavaş büyüyen, lokal gelişme göste-

ren, nadiren lenf nodu ve uzak organ metastazı yapan ve

bazen hormonal aktivite gösteren bir tümör grubudur (1).

Karsinoid tümörlerin çoğu ana bronş veya lob bronşu gibi

büyük solunum yollarından köken alır. Atipik ve tipik kar-

sinoid tümör olarak 2 grupta incelenir. Karsinoid tümörle-

rin %10-20’si atipik, %80-90'ı tipik karsinoiddir (2). Genç

yaş grubunda daha sık görülen bu tümörler bizim olgu-

muzda olduğu gibi nadiren ileri yaşta rastlantısal olarak

da görülebilir. Yazımızda atipik karsinoid tümör tanısı

koyup endobronşial tedavi uyguladığımız bir hasta su-

nulmaktadır. Hastamızı sunma amacımız; ileri yaşta nadir

karşılaşılan bir olgu olması ve eşlik eden hastalıkları ne-

deniyle cerrahi yapılamayıp endobronşial tedavi uygu-

lanması nedeniyle literatür eşliğinde tartışmaktır.

OLGU

Yetmiş sekiz yaşında erkek hasta, ağır KOAH, opere la-

rinks karsinomu (skuamöz hücreli karsinom) tanısıyla

polikliniğimizde takip edilmekteydi. Elli paket/yıl sigara

öyküsü vardı. Fizik muayenede ekspiryum uzun olup solu-

num sesleri azalmıştı. Oda havasında oksijen saturasyo-

nu %80 idi. İki yıl önceki toraks bilgisayarlı tomografisin-

de sağ paratrakeal lenfadenomegali olması nedeniyle

kontrol amaçlı toraks bt istendiğinde: trakeal polipoid

lezyon izlendi (Şekil 1 ve 2). Lokal anestezi altında yapılan

fiberoptik bronkoskopisinde: trakeada karinaya 3 cm

mesafede, sağ lateral duvarda, havayolunu %80 oranın-

da daraltan polipoid lezyonlar izlendi (Şekil 3) ve buradan

biyopsi alındı. Patoloji sonucu: nöroendokrin tümör ola-

rak raporlandı. Hastanın solunum sıkıntısı olup havayolu

açıklığını sağlamak üzere endonronşial tedavi planlandı.

Genel anestezi altında rijid bronkoskop ile Nd:YAG lazer

ile lezyonun eksizyonu ve ardından lezyonun köklerine

koterizasyon yapıldı. Patoloji sonucu atipik karsinoid tü-

mör olarak raporlandı. İleri yaşı, ağır KOAH ve opere

larenks karsinomu olmasından dolayı cerrahi tedavi dü-

şünülmedi.

TARTIŞMA

Primer trakeobronşiyal tümörler tüm pulmoner tümörlerin

yaklaşık %0,1’ni oluşturmaktadırlar (3). Karsinoid tümör,

epitel hücrelerinden köken alan akciğerin nöroendokrin

tümörleri arasındadır. Dünya Sağlık Örgütü akciğerin

nöroendokrin tümörlerini: karsinoid tümör (tipik/atipik),

büyük hücreli nöroendokrin karsinom (BHNEK) ve küçük

hücreli karsinom (KHK)olarak sınıflandırmıştır (4). Akciğer

karsinomlarının yaklaşık %2’ sini oluştururlar (5,6). Bron-

kopulmoner karsinoidlerin %75-90’ı santral %10-25’i

periferik yerleşimlidir. Pulmoner karsinoidler çocuklarda

ve gençlerde en sık görülen primer akciğer tümörleridir

(7).

Karsinoid tümörler genellikle 45-55 yaş grubu arasında

görülmektedir. Tipik karsinoidler (TK) atipik (AK) olanlara

göre daha erken yaş grubunda görülme eğilimi gösterirler.

Genel olarak erkek ve kadın cinsiyet için eşit dağılım

izlenmektedir.

Şekil 1: Axial kesitte trakeadaki polipoid lezyonlar.

Şekil 2: Coronal kesitte trakeadaki polipoid lezyonlar

Şekil 3: Bronkoskopide lümeni daraltan polipoid lezyonlar

Trakeal Yerleşimli Atipik Karsinoid Tümör | Afşin et al.

58 www.respircase.com 58

Atipik karsinoid tümörler malign histolojik özellikler ve

agresif tavır gösteren tümörlerdir. TK’in tersine bu tümör-

ler sıklıkla periferal yerleşimli olup daha ileri yaş grubun-

da görülür. Bizim olgumuz ileri yaşta (78 yaşında) olup

lezyonu santral yerleşimli idi. Atipik karsinoidli hastalarda

yüksek oranda metastaz gelişme riski vardır (8,9).

Proksimal lokalizasyon gösteren tümörler kısmi veya tam

bronş obstrüksiyonu oluşturabilirler. Bizim olgumuzda da

bronkoskopik olarak trakea lümeni %80 oranında daral-

mış olarak izlendi. Öksürük, hemoptizi, tekrarlayan enfek-

siyon bulguları klasik semptomlarındandır. Trakea veya

ana bronş yerleşimi olan olgularda stridor gelişebilir.

Pulmoner karsinoid olgularında tanı anında karsinoid

sendrom görülmesi oldukça nadirdir ancak; büyük tümö-

rü olan veya yaygın karaciğer metastazı olan olgularda

izlenebilmektedir (10,11). Olgumuzda olduğu gibi karsi-

noid tümörlerin yaklaşık %40’ı belirgin bir klinik bulgu

olmadan insidental olarak radyolojik bulgularla saptan-

maktadır (12). Radyolojik olarak iyi sınırlı, yuvarlak veya

ovoid, hafifçe lobüle nodüller olarak tomografilerde izle-

nebildiği gibi sadece hava yolunda saplı polipoid lezyon-

lar olarak da izlenebilir (13).

Tanı için günümüzde nöroendokrin tümörlerde somatos-

tatin analogları ile yapılan PET görüntülemede sıklıkla

Galyum-68 kullanılır (14). Tüm karsinoidlerin yakla-

şık %75’i bronkoskopik olarak görülebilir. Bronkoskopik

olarak vaskülaritesi fazla, pembe-mor intakt epitel ile

örtülü polipoid lezyonlar şeklinde izlenirler. Bazı karsinoid

tümörler polipoid ve saplı iken bazıları da sapsız olarak

izlenirler. Tümörün çoğunluğu ekstraluminal yerleşimli

olabileceği için “buz dağı tümörler” olarak da isimlendiri-

lirler (10). Vaskülaritesi oldukça fazla olan ve submukozal

yerleşimli olan bu tümörlerde derin biyopsiler tanı koya-

bilmek için gerekli olup, sıklıkla ciddi kanamalara yol

açabilir. Ancak olgumuzda biyopsi sonrasında kanama

sorunu yaşanmadı. Bazı olgularda genel anestezi altında

rijid bronkoskopi ile işlemin yapılması kanama kontrolünü

sağlamak için tercih edilebilir (15). AK ile TK ayırımı an-

cak cerrahi ve bronkoskopik olarak çıkarılan kitleden

yapılabilir. Karsinoid tümörlerde evreleme için TNM sis-

temi kullanılmaktadır. Ancak multipl karsinoid tümör

mevcudiyetinde metastaz yerine senkron primer tümörler

olarak düşünmek gerekir (16). TK’ler düşük dereceli ma-

lign davranış gösterirler ve beş yıllık sağkalım %87-

100’dür. AK’ler ise daha fazla malign potansiyele sahip-

tirler ve beş yıllık sağkalım oranı %56-75’dir (17,18).

Pulmoner karsinoidlerde tedavinin esası; akciğer dokusu

korunarak tümörün komplet rezeksiyonudur. Ancak tümö-

rün distalinde geri dönüşümsüz değişikliklerin saptandığı

olgularda akciğer rezeksiyonu da gerekebilmektedir.

Ayrıca, cerrahi olarak komple lenfadenektomi yapılması

da önerilmektedir (16).

Son yıllarda doğru seçilmiş olgularda yapılan bronkosko-

pik rezeksiyon sonuçlarının da iyi olduğu bildirilmektedir

(17,19). Bronkoskopik olarak Argon plazma koagülasyo-

nu, elektrokoter, kriyoterapi ya da lazer teknikleri kulla-

nılmaktadır (20). Santral yerleşimli pulmoner karsinoidli

hastalara başlangıçta endobronşiyal tedavi yapılması,

ekstralüminal komponenti belirgin olanlarda da tedavinin

cerrahi rezeksiyon ile tamamlanmasının uygun bir yakla-

şım olacağı belirtilmektedir. Endobronşiyal tedavi sonrası

altıncı haftada bronkoskopik ve görüntüleme yöntemleri

ile lezyonun (özellikle ekstralüminal komponentin) yeniden

değerlendirilmesi önerilir. Ekstralüminal komponenti be-

lirgin olan, tümörün distalinde geri dönüşümsüz paranki-

mal lezyonu olan, endobronşiyal tedavi sonrası nüks olan,

atipik karsinoid histolojisi olan veya belirgin mediastinal

lenfadenopatileri olan olgularda cerrahi tedavi uygulan-

ması önerilir (17). Yirmi yıl sonra bile nüks olabileceğin-

den 20 yıla kadar takip önerilir.

Bizim olgumuzun radyolojik olarak ekstraluminal uzanımı

ve metastatik lenf nodu olmaması üzerine, Nd:YAG lazer

ve ardından lezyon köküne kriyoterapi uygulandı. Hasta-

nın ileri yaşı ve ağır KOAH'ı (FEV1: %25) olması nedeniy-

le inoperabl kabul edildi. Postop bronkoskopik kontro-

lünde lezyon izlenmedi (Şekil 4) ve alınan kontrol biyopsi-

sinde malign hücre izlenmedi.

Şekil 4: Tedavi sonrası altıncı hafta bronkoskopik görünüm

Sonuç olarak; karsinoid tümörler düşük gradlı malign ve

yavaş büyüyen lokal invazif tümörlerdir. Endobronşial

tedavi ile başarılı sonuçlar alınmaktadır. Bizim hastamız

ileri yaşta rastlantısal olarak atipik karsinod tümör tanısı

konulması ve endobronşial rezeksiyon ile tam kür sağ-

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 59

lanmış olması nedeniyle sunularak ilgili literatür gözden

geçirilmiştir.

ÇIKAR ÇATIŞMASI

Bu makalede herhangi bir çıkar çatışması bildirilmemiştir.

YAZAR KATKILARI

Fikir - E.A., A.O.; Tasarım ve Dizayn - E.A., A.O.; Denet-

leme - E.A., A.O.; Kaynaklar - E.A.; Malzemeler - E.A.;

Veri Toplama ve/veya İşleme - E.A.; Analiz ve/veya Yo-

rum - E.A.; Literatür Taraması - E.A.; Yazıyı Yazan - E.A.;

Eleştirel İnceleme - E.A.

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15. Harpole DH, Feldman JM, Buchanan S, Young WG,

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16. Caplin ME, Baudin E, Ferolla P, Filosso P, Garcia-Yuste

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Respir Case Rep 2020;9(2): 60-65 DOI: 10.5505/respircase.2020.01328

OLGU SUNUMU CASE REPORT

60

Yasemin Arı Yılmaz, Meral Gulhan

Pulmoner tromboemboli daha çok alt ekstremite

venlerinden kaynaklanmaktadır. Ana pulmoner yata-

ğın aniden tıkanması kardiyak dekompansasyona

neden olabilir. Elektrokardiyografi (EKG)de sağ vent-

rikül yüklenme bulguları olan V1-4 T negatifliği,

V1’de QR paterni, komplet veya inkomplet sağ dal

bloğu ve S1Q3T3 görülebilir. Acil ve hızlı tedavi

hayat kurtarıcı olabilmektedir. Streptokinaz ve alteplaz

verilişi saatler sürerken tenekteplaz verilişi puşe şek-

linde olmaktadır. Bu da dakikaların önemli olduğu

acil müdahale gerektiren durumlarda hayati önem

arzetmektedir. Ayrıca riskli ilaçların uygulanması

esnasında sorumlu doktorun hasta başında bekleme

süresini ve iş gücü kaybını kısaltmaktadır. Hastanın

daha kısa sürede normale dönmesini sağlamaktadır.

Bu konuda literatürde çok sayıda çalışma olmasına

rağmen ülkemizde kullanım endikasyonu olmadığın-

dan verilerimiz yetersizdir. Bu olguda alternatif ilaçlar

elde olmadığı için tenekteplaz kullanılmak zorunda

kalınmıştır.

Anahtar Sözcükler: Pulmoner tromboemboli, tromboli-

tik Tedavi, tenekteplaz.

Pulmonary thromboembolisms occur mostly in the

lower extremity veins. Sudden occlusions of the main

pulmonary bed may result in cardiac

decompensation. Electrocardiography (ECG) shows

right ventricular overload findings, V1-4 T negativity,

Q1 pattern in V1, complete or incomplete right

bundle branch block and accompanying S1Q3T3.

Emergency and rapid treatment can be life-saving.

Streptokinase and alteplase administration lasts for

hours while tenecteplase administration is in the form

of push. This is vital in patients requiring immediate

interventions, where minutes can be critical. It also

reduces the waiting time of the responsible doctor

and loss of labor during the administration of risky

drugs, and enables the patient to return to normal in

a shorter time. Although there have been many

studies in the literature addressing this subject, the

available data is insufficient, since there is no

indication for use in our country. In the present case,

as no alternative drugs were available, tenecteplase

had to be used.

Key words: Pulmonary thromboembolism, thrombolyt-

ic therapy, tenecteplase.

Hitit Üniversitesi Göğüs Hastalıkları Ana Bilim Dalı, Çorum Department of Chest Diseases, Hitit University, Çorum, Turkey

Başvuru tarihi (Submitted): 01.10.2019 Kabul tarihi (Accepted): 10.02.2020

İletişim (Correspondence): Yasemin Arı Yılmaz, Hitit Üniversitesi Göğüs Hastalıkları Ana Bilim Dalı, Çorum

e-mail: yasminbee07@gmail.com

RES

PIR

ATO

RY

CA

SE R

EPO

RTS

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 61

Pulmoner tromboemboli daha çok alt ekstremite venle-

rinden kaynaklanmaktadır. Ana pulmoner yatağın aniden

tıkanması kardiyak dekompansasyona neden olabilir.

Elektrokardiyografi (EKG)’de sağ ventrikül yüklenme bul-

guları olan V1-4 T negatifliği, V1’de QR paterni, komplet

veya inkomplet sağ dal bloğu ve S1Q3T3 görülebilir (1-

3). Yüksek riskli emboli olarak adlandırılan bu duruma

hipotansiyon hipoksi gibi şok bulguları eşlik eder. Acil ve

hızlı tedavi hayat kurtarıcı olabilmektedir. Streptokinaz ve

alteplaz verilişi saatler sürerken tenekteplase verilişi ise

puşe şeklinde olmaktadır. Bu da dakikaların önemli oldu-

ğu acil müdahale gerektiren durumlarda hayati önem arz

etmektedir. Ayrıca riskli ilaçların uygulanması esnasında

sorumlu doktorun hasta başında bekleme süresini ve iş

gücü kaybını da kısaltmaktadır. Hastanın daha kısa süre-

de normale dönmesini sağlamaktadır. Bu konuda litera-

türde çok sayıda çalışma olmasına rağmen ülkemizde

kullanım endikasyonu olmadığından bizim verilerimiz

yetersizdir. Bu olguda, alternatif ilaçlar elde olmadığı için

pulmoner embolide tenekteplaz kullanılmak zorunda

kalınmıştır ve yaşanan deneyimin ülke verisi olarak payla-

şılması amaçlanmıştır.

OLGU

Göğüs ağrısı ve nefes darlığı şikâyeti ile acil servisimize

başvuran 65 yaşındaki kadın hastanın özgeçmişinde di-

yabetes mellitus ve obezite dışında bilinen hastalığı yoktu.

Hastanın oda havasında geliş satürasyonu (SaO2) %80

idi. Laboratuvar parametreleri tablo 1 de görülmektedir.

Akciğer grafisinde, (film kalitesi kötü olmakla beraber)

ekspiryum filmi olup mediastende hafif dolgunluk vardı.

Yatarak hasta başı çekilmişti. Belirgin parankimal patoloji

izlenmedi (Şekil 2).

Hasta bu bulgularla miyokard enfaktüsü açısından kardi-

yolojiye danışıldı. Hastanın 1 hafta öncesinde de göğüs

ağrısı ve nefes darlığı ile kardiyoloji tarafından değerlen-

dirildiği ve yapılan koroner anjiografi ve ekokardiyografi

(eko) bulgularının normal olduğu öğrenildi. Yapılan eko-

sunda daha önceki ekosunda olmayan ileri triküspit yet-

mezliği izlendi. Sağ boşluklarda genişleme olduğu belir-

tildi. Pulmoner arter basıncı ölçülemedi. Hastada mevcut

bulgular ile miyokard enfarktüsü düşünülmedi.

Hasta tarafımıza konsülte edildi. Hastanın başvuru saati

gece olduğundan o sırada nöbetçi radyoloji uzmanı ol-

madığından alt ekstremite venöz dopler ultrasonografi

(USG) çekilemedi. Ancak sol bacakta derin ven trombo-

zunu düşündürür şekilde, çap farkı, şişlik ve kızarıklık

mevcuttu. Geliş öyküsünde daha önce nefes darlığı ve

göğüs ağrısının olmadığını, şikâyetlerinin bir hafta önce

başladığını ve giderek ciddi şekilde arttığını ifade etti.

Belirgin öksürük ve balgamı yoktu. Kreatinin yüksek oldu-

ğundan kontrastsız toraks BT çekilebildi. BT'de belirgin

parankimal patoloji saptanmadı. EKO bulguları ile uyum-

lu olarak kardiyak şift, sağ boşluklarda genişleme ve

pulmoner arterin dilate olduğu gözlendi (Şekil 3, 4 ve 5).

Şekil 1: Tedavi öncesi EKG (atriyal fibrilasyon).

Tablo 1: Hastanın başvurudaki laboratuvar parametreleri

Parametre Ölçüm Normal

Aralık

Beyaz küre 13850 10⁹/ul 4-10

Hemoglobin 12,6 gr/dl 11-16

Trombosit 193 103/ul 100-300

Glukoz 214 mg/dl 74-106

Kreatin 1,7 mg/dl 0,5-0,9

C-Reaktif Protein 23 mg/l 0-5

D-Dimer: 5 mg/l 0,063-0,701

Troponin 0,074 ng/ml 0,0-0,02

Pro-BNP Çalışılmadı

Venöz kan gazı

• PO2 22,4 mmHg 83-108

• PCO2 40,4 mmHg 32-45

• ph 7,28 7,35-7,45

• O2 sat 21,6 % 95-99

Kardiyak Arrestle Seyreden ve Tenekteplaz Kullanılan bir Masif Pulmoner Tromboemboli Olgusu | Arı Yılmaz et al.

62 www.respircase.com 62

Şekil 2: Akciğer grafisi, ekspiryum filmi olup mediastende hafif dolgun-

luk vardı. Belirgin parankimal patoloji izlenmedi.

Şekil 3: Tedavi öncesi kontrastsız toraks BT.

Şekil 4: Tedavi öncesi kontrastsız toraks BT.

Şekil 5: Tedavi öncesi kontrastsız toraks BT (Sağ ventriküler genişleme

ve kardiyak shift kırmızı ok).

Hasta klinik laboratuvar ve tetkik sonuçları ile rehberlere

göre yüksek riskli pulmoner tromboemboli olarak kabul

edildi. Trombolitik tedavi amaçlı kardiyoloji yoğun bakım

ünitesine göğüs hastalıkları adına yatırıldı. Ancak hasta

yatağına alındıktan hemen sonra kardiyak arrest oldu. Bu

nedenle hastaya transözefajial eko ya da dopler ultraso-

nografi için beklenilemedi. Acil karar verilmesi gereken

bir durumdu. Elimizde diğer trombolitik ajanlar yoktu.

Sistemden istem yapılması, eczaneden alınıp gelinmesi

yaklaşık 30 dakikalık bir zaman kaybı olacağından ve

hastanın bu süreyi bekleyecek zamanı olmadığından,

hasta yakınlarına durum ile ilgili bilgi verilip onamları

alındıktan sonra koroner yoğun bakım ünitesinde hazır

bulunan tenekteplaz hastaya puşe olarak verildi. Hastanın

yaklaşık 90-100kg civarı olduğu tahmin edilerek

50mg=10ml dozunda uygulandı Resüsitasyona devam

edilen hasta bir süre sonra sinüs ritminde döndü (Şekil 6).

Satürasyonları takibinde 10 dk. içinde %90'a kadar yük-

seldi. Koroner anjio yapılan bölgeye kum torbası ile bası

uygulandı. Takipte düşük moleküler ağırlıklı heparin

(DMAH) tedavisine geçildi. Koroner anjio yapılan bölgede

hematom gelişti. Hb:12,6 gr/dl’den 9,6 gr/dl'e geriledi.

Hastaya 1 ünite eritrosit replasmanı yapıldı. Kanaması

duran, satürasyonu %99'a kadar yükselen hasta servise

alındı. Serviste 1 ünite daha eritrosit replase edildi. War-

farin başlandı. INR'si 2-3 arasına gelen ve ek problem

saptanmayan hasta toplam 1 hafta sonrasında yürüyerek

taburcu edildi. Kontrolünde alt ekstremite venöz dopler

USG'de sol popliteal vende trombüs izlendi. Kreatinin

normale gerileyen hastanın Spiral Toraks BT anjiografi-

sinde tamamen düzelme saptandı (Şekil 7 ve 8). Warfarin

ile takibine devam edilmektedir.

Şekil 6: Tedavi sonrası EKG’si (Sinüs ritmi).

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 63

TARTIŞMA

Bir doku plazminojen aktivatörü olan tenekteplazın miyo-

kard enfaktüsünde kullanım onayı olmasına rağmen he-

nüz pulmoner tromboembolide kullanım onayı mevcut

değildir. ST elevasyonu olan miyokard enfarktüsünde ilaç

dozu vücut ağırlığı baz alınarak hesaplanmaktadır. Öne-

rilen dozlar; <60 kg: 6.000 U, 30 mg, 6 ml. >=60-

<70 kg: 7.000 U, 35 mg, 7 ml. >=70-<80 kg: 8.000

U, 40 mg, 8 ml. >=80-<90: 9.000 U, 45 mg, 9

ml. >=90: 10.000 U, 50 mg, 10 ml’dir. Gerekli doz, 5-

10 saniye içerisinde, tek intravenöz bolus şeklinde uygu-

lanmaktadır (4). Pulmoner tromboembolide (PTE) de

kullanım dozu da benzerdir (5,6).

Pulmoner tromboembolide tenekteplaz kullanımı ile ilgili

literatürdeki ilk veriler 2001 yılına dayanmaktadır (7,8).

O dönemden beri tenekteplazın pulmoner embolideki

kullanımı olgu sunumları ve çalışmalara konu olmaktadır.

Akut miyokard enfarktüsünde alteplaz ve streptokinaz

karşısındaki güvenilirliğini ve etkinliğini kanıtlamış ve onay

almıştır. Aktif olarak kullanılmaktadır. Majör kanamaların

daha az olduğuna yönelik yayınlarda mevcuttur (9).

Şekil 7: Tedavi sonrası Kontrastlı Toraks BT Anjio (Sağ boşluklar ve

kardiyak septum normal).

Şekil 8: Tedavi sonrası Kontrastlı Toraks BT Anjio (Sağ boşluklar ve

kardiyak septum normal).

Ancak yüksek riskli pulmoner embolide kullanıma ilişkin

halen yeterli sayıda çalışma olmadığından rehberlere

girmeyi başaramamıştır ve çalışmalar devam etmektedir.

Nitekim 2019 ERS raporunda da çalışılmaya devam edi-

len ajanlar arasında gösterilmiştir (2).

Literatürde yüksek riskli pulmoner embolide alteplaz ile

tenekteplazı karşılaştıran bir çalışmaya rastlanılamadı.

Ancak orta ve yüksek riskli pulmoner embolide tenektep-

lazın plesebo heparin ya da streptokinaz ile karşılaştırmalı

yayınları mevcuttur:

Bu yayınların en büyüğü Meyer ve ark. (10) tarafından

yapılan orta riskli pulmoner embolili hastalarda tenektep-

laz+heparin ile plasebo+heparini karşılaştıran çok mer-

kezli, çift kör, randomize kontrollü çalışmadır. Trombolitik

tedavinin ilk 7. veya 30. günde mortaliteyi önemli ölçüde

azaltmadığı, ancak hemodinamik bozulmayı önlediği

bulunmuştur. Ancak, majör kanama tenekteplaz grubun-

da anlamlı olarak daha sıktı. Kline ve ark. (11) tarafından

yapılan bir başka randomize çalışmada da düşük mole-

küler ağırlıklı heparin (DMAH) ve DMAH + tenekteplaz

karşılaştırılmıştır. Üç aylık takipte tenekteplaz grubu daha

iyi prognoz, yaşam kalitesi ve fonksiyonel kapasite de artış

göstermiştir.

Becattini ve ark. (12) ise sağ ventrikül disfonksiyonu olan

hemodinamik olarak stabil pulmoner embolili hastalarda

tenekteplaz+heparin ile plesebo+heparini karşılaştırmıştır.

Miyokard enfarktüsü için kullanılan dozlarda kullanıldı-

ğında ciddi kanamalara neden olmadan sağ ventrikül

disfonksiyonunu 24 saatte belirgin şekilde düzelttiği gös-

terilmiştir.

Agrawal ve ark. (13) yaptıkları çalışmada 33 masif, 50

submasif, 20 nonmasif pulmoner embolili, 103 hastanın

62’sine tenekteplaz, 17’sine streptokinaz, 24’üne heparin

uygulanmıştır. Tenekteplaz’ın dispneyi daha belirgin azalt-

tığı ve hatta sağ dal bloğunu geri çevirmede %100 başa-

rılı olduğu saptanmıştır. Tenekteplaz grubunda tedavi

öncesi satürasyonu (SaO2) %88,79’dan tedavi sonra-

sı %96,90, 6 ay sonra %97,91’e çıkarken; streptokinaz

grubunda, tedavi öncesi SaO2 %91,90 tedavi sonra-

sı %94,90 6 ay sonra %90,09; heparin grubunda ise

tedavi öncesi SaO2 % 91,4 tedavi sonrası %92,75 6 ay

sonra %91,83 olarak saptanmıştır (13).

Shukla ve ark. (14), 30 pulmoner embolili hastanın dâhil

edildiği çalışmalarında, tenekteplaz’ı etkili ve güvenilir

olarak saptamışlardır. Yine bu çalışmada 4 sağ dal bloğu

olan hastanın 4’ününde dal blokları düzelmiş olarak

taburcu edilmiştir. Nitekim bizim olgumuzda da sağ dal

bloğu düzeldi ve entübasyona gerek kalmadan sinüs

ritmine döndü.

Kardiyak Arrestle Seyreden ve Tenekteplaz Kullanılan bir Masif Pulmoner Tromboemboli Olgusu | Arı Yılmaz et al.

64 www.respircase.com 64

Tenekteplaz kullanımı inravenöz bolus şeklinde olduğun-

dan ve oda sıcaklığında stabil olup sulandırılmadan kul-

lanılabildiğinden çoğu kez arrest vakalarında da kullanıl-

mış olup çok sayıda başarılı sonuçlar alınmıştır (15,16).

İleri yaş hastalarda ve gebelerde kullanımında bile ciddi

komplikasyon saptanmamıştır (17,18). Alteplaz ile karşı-

laştırıldığında, etkide azalma olmaksızın kanama riskinin

daha az olduğuna dair yayınlar mevcuttur (9,19).

Yüksek ve orta riskli pulmoner embolilerde kullanımına

ilişkin yurtdışı yayınlar giderek artmakta olmasına karşın,

ülkemizde pulmoner tromboembolide kullanımına yönelik

yayın bulunamamıştır. Sadece yurtdışı benzer olgu su-

numları mevcuttur (20-22). Bu nedenle olgumuzun litera-

türe katkı sağlayacağı düşünülmüştür.

Sonuç olarak; bizim olgumuzda elde edilen başarılı yanıt

ve diğer çalışmalara bakıldığında, tenekteplazın ileride

rehberlere girmesi için randomize kontrollü çalışmalara

ihtiyaç olduğu düşünülmektedir.

ÇIKAR ÇATIŞMASI

Bu makalede herhangi bir çıkar çatışması bildirilmemiştir.

YAZAR KATKILARI

Fikir - Y.A.Y., M.G.; Tasarım ve Dizayn - Y.A.Y., M.G.;

Denetleme - Y.A.Y., M.G.; Kaynaklar - Y.A.Y., M.G.;

Malzemeler - Y.A.Y., M.G.; Veri Toplama ve/veya İşleme

- Y.A.Y., M.G.; Analiz ve/veya Yorum - Y.A.Y., M.G.;

Literatür Taraması - Y.A.Y., M.G.; Yazıyı Yazan - Y.A.Y.,

M.G.; Eleştirel İnceleme - Y.A.Y., M.G.

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Respir Case Rep 2020;9(2): 66-69 DOI: 10.5505/respircase.2020.81489

OLGU SUNUMU CASE REPORT

66

Halil İbrahim Yakar1, Asiye Kanbay

2

Pulmonary endarterectomy (PEA) is a surgical treat-

ment approach for the treatment of patients with

chronic thromboembolic pulmonary hypertension

(CTEPH). The surgery is considered high risk in terms

of mortality and morbidity. We present here a patient

at high operative risk due to severe pulmonary hyper-

tension (PH) and chronic renal failure (CKD). The 22-

year-old male patient was undergoing hemodialysis

treatment for three years due to bilateral hydro-

nephrosis-induced CKD, and was admitted to the

hospital with increasing dyspnea. The diagnosis of

CTEPH was made clinically and radiologically. Dur-

ing the follow-up period, an endarterectomy was

planned, although the patient was considered high

risk due to insufficient clinical response, despite med-

ical therapy. Bilateral PEA was applied by thoracic

surgery team. sPAP decreased from 105 to 35 mmHg

on echocardiography, and dyspnea was improved

and functional capacity recovered after the PEA sur-

gery. A renal transplantation was contraindicated in

the preoperative period due to severe pulmonary

arterial hypertension. PEA surgery was enabled to

patient kidney transplantation due to an improvement

in pulmonary functions and pulmonary artery pres-

sure. We present here a case of CTEPH who had a

chance of kidney transplantation after undergoing

successful PEA surgery.

Key words: Chronic Thromboembolic Pulmonary

Hypertension, Chronic Renal Failure, Pulmonary En-

darterectomy.

Pulmoner endarterektomi (PEA), kronik tromboembo-

lik pulmoner hipertansiyon (KTEPH) hastalarında

cerrahi tedavi yöntemi olarak kullanılmaktadır. Diğer

yandan mortalite ve morbiditesi yüksek bir cerrahidir.

Burada ileri derece pulmoner hipertansiyonu (PH)

bulunan, kronik böbrek yetmezliği (KBY) nedenli

hemodialize giren ve bu nedenle yüksek riskle PEA

yapılan bir olgu sunuldu. Yirmi iki yaşında erkek

hasta, bilateral hidronefroza bağlı KBY nedeni ile 5

yıldır haftada 3 gün hemodialize girmekteydi. Gide-

rek artan nefes darlığı nedeniyle hastaneye başvurdu.

Klinik ve radyolojik olarak KTEPH tanısı konuldu.

Takiplerinde medikal tedaviye rağmen yeterli klinik

yanıt alınamaması nedeniyle hastaya yüksek riske

rağmen pulmoner endarterektomi planlandı. Göğüs

cerrahisi tarafından bilateral PEA operasyonu uygu-

landı. Hastanın kontrol ekokardiografisinde sPAP:

105 mmHg’dan 35 mmHg’ a geriledi. Cerrahi son-

rası takiplerinde hastanın istirahat dispnesi kayboldu,

eforla dispne şikâyeti belirgin azaldı ve fonksiyonel

kapasitesi arttı. Operasyon öncesi pulmoner hiper-

tansiyon nedeniyle hastada renal transplantasyon

kontrendike iken, postoperatif şikayetlerinin gerileme-

si ve pulmoner arter basıncı düşmesi nedeniyle hasta-

ya transplantasyon imkânı oluştu. Bu olgu sunumun-

da, başarılı bir PEA geçirdikten sonra böbrek nakli

imkânı oluşan bir KTEPH hastası sunuldu.

Anahtar Sözcükler: Kronik Tromboembolik Pulmoner

Hipertansiyon, Kronik Böbrek Yetmezliği, Pulmoner

Endarterektomi.

1Tokat Gaziosmanpaşa Üniversitesi Tıp Fakültesi Göğüs Has-

talıkları Anabilim Dalı, Tokat

2İstanbul Medeniyet Üniversitesi Tıp Fakültesi Göğüs Hastalıkları

Anabilim Dalı, İstanbul

1Department of Chest Disease, Tokat Gaziosmanpasa

University, Faculty of Medicine, Tokat, Turkey

2Department of Chest Disease, İstanbul Medeniyet University,

Faculty of Medicine, İstanbul, Turkey

Submitted (Başvuru tarihi): 15.12.2019 Accepted (Kabul tarihi): 04.02.2020

Correspondence (İletişim): Halil İbrahim Yakar, Tokat Gaziosmanpaşa Üniversitesi Tıp Fakültesi Göğüs Hastalıkları

Anabilim Dalı, Tokat

e-mail: halil_yakar@hotmail.com

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Chronic thromboembolic pulmonary hypertension

(CTEPH) is a disease with poor survival rates and increas-

ing frequency (1). Recurrent and organized thrombi oblit-

erate the pulmonary vascular system and cause progres-

sive pulmonary hypertension and right heart failure. Pul-

monary thromboendarterectomy (PEA) is the leading cu-

rative approach to CTEPH, and can be life-saving in

patients with severe pulmonary hypertension and proximal

thrombus in the pulmonary arteries. In pulmonary

endarterectomy operations, residual thrombus and fi-

brous obstructive tissue in the pulmonary arteries can be

extracted under conditions of deep hypothermic circulato-

ry arrest (2). In the postoperative period, pulmonary re-

vascularization, a decrease in pulmonary artery pressure

and clinical improvement can be expected due to the

removal of thrombus tissue. That said, it is a surgical

method associated with a high rate of mortality and mor-

bidity (3,4). We present here a patient considered a high

surgical risk due to CRF prior to PEA, and who gained the

possibility of renal transplantation after successful PEA

surgery.

CASE

A 22-year-old male patient who had undergone hemodi-

alysis three times a week for five years due to bilateral

hydronephrosis, was admitted to our hospital with exer-

cise dyspnea that had developed over the prior three

months (Functional Capacity 3-4). Prominent pulmonary

arteries and cardiomegaly were seen on chest X-ray (Fig-

ure 1), while p-pulmonale, right ventricular hypertrophy

and right bundle branch block were seen on ECG (Figure

2). Echocardiography (ECO) revealed enlargement of the

right heart cavity, severe pulmonary hypertension (sPAP:

105 mmHg) and paradoxical movement due to increased

pressure in the interventricular septum. A Thorax CT an-

giography showed an enlargement of the main pulmo-

nary artery and its branches, a nearly complete thrombus

that obstructed the lumen, extending from the bilateral

main pulmonary arteries to the segment branches, and

also pneumonic infiltration in the right upper lobe (Figure

3). Anticoagulant therapy and antibiotherapy were initiat-

ed for CTEPH and pneumonia.

As a result of the insufficient clinical response to antico-

agulant therapy, despite the three months of anticoagu-

lant therapy, no clinical improvement was observed, and

a PEA operation was planned, despite the high surgical

risk. A bilateral pulmonary endarterectomy with median

sternotomy under systemic hypothermia was successfully

performed by the thoracic surgery team (Figure 4). In the

control ECO, sPAP decreased to 35 mmHg.

In the postoperative follow-up, the patient's dyspnea

complaint reduced considerably and functional capacity

increased. The patient was referred to the Nephrology

Unit for renal transplantation for CRF.

DISCUSSION

CTEPH is one of the leading causes of PHT (5). In pa-

tients with pulmonary artery pressure (PAP) exceeding 50

mmHg, average five-year survival is reported to be 10%

(6). CTEPH should be considered in the differential diag-

nosis of patients presenting with chronic dyspnea and/or

chest pain. CTEPH should be investigated with a CT thor-

ax angiography and echocardiography in such cases.

Surgery is the optimum treatment for patients diagnosed

with CTEPH (7), although PEA has a high risk of morbidity

and mortality (8-10). Accordingly, medical treatment (oral

anticoagulant, CCB, ERA, Riociguat, etc.) is preferred in

patients with PAP> 50mmHg and in the presence of an

additional disease that may increase the risk of postoper-

ative mortality and morbidity (11,12). OAC was initiated

in our case due to sPAP: 105 (> 50) mmHg and CRF.

However, the PEA operation was planned due to the

patient’s inability to respond to medical treatment and the

patient's young age. While a renal transplant was contra-

indicated in the preoperative period due to PHT, a renal

transplantation was possible due to the regression of the

complaints, and the decrease in pulmonary artery pres-

sure in the postoperative period.

Figure 1: Initial Posteroanterior Chest X-ray (Bilateral pulmonary arteries

were prominent, and pneumonic infiltration surrounded the vascular

structures in the right upper lobe)

Pulmonary Thromboendarterectomy Enabling Renal Transplantation in Patient of Chronic Kidney Disease: A Case Report | Yakar et al.

68 www.respircase.com

Figure 2: ECG: Right bundle branch block and right ventricular hyper-

trophy (V1-V2 RSR pattern, ST depression and T wave inversion on right

precordial leads [V1-3])

Figure 3: Axial and coronal section on thorax CT angiography. En-

largement of the main pulmonary artery and its branches, widespread

thrombus obstructing the lumens, extending from the bilateral pulmonary

arteries to the segment branches, and pneumonic infiltration of the right

upper lobe were also observed

Figure 4: Postoperative view of thrombus material in the pulmonary

artery branches

CONCLUSION

Pulmonary endarterectomy is a curative treatment for

CTEPH. While renal transplantation was contraindicated

in the preoperative period due to PHT, it became possible

due to PEA in the postoperative period in our case. As

such, PEA is thought to be important for survival in CTEP

patients, and so all patients should carefully be evaluated

for PEA.

ABBREVIATIONS

CTEPH: Chronic thromboembolic pulmonary hyperten-

sion, sPAB: systolic pulmonary artery pressure, PTE: Pul-

monary thromboendarterectomy, CRF: Chronic renal

failure, ECO: Echocardiography, ECG: Electrocardiog-

raphy, OAC: Oral anticoagulant, PHT: Pulmonary

Hipertension, ERA: Endothelin receptor antagonist, CKB:

blockers.

ACKNOWLEDGEMENT

We thank Mr. Bedrettin Yildizeli for the patient's successful

surgical operation.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - H.İ.Y., A.K.; Planning and Design - H.İ.Y., A.K.;

Supervision - H.İ.Y., A.K.; Funding - H.İ.Y.; Materials -

H.İ.Y.; Data Collection and/or Processing - H.İ.Y., A.K.;

Analysis and/or Interpretation - H.İ.Y., A.K.; Literature

Review - H.İ.Y.; Writing - H.İ.Y.; Critical Review - H.İ.Y.,

A.K.

YAZAR KATKILARI

Fikir - H.İ.Y., A.K.; Tasarım ve Dizayn - H.İ.Y., A.K.; De-

netleme - H.İ.Y., A.K.; Kaynaklar - H.İ.Y.; Malzemeler -

H.İ.Y.; Veri Toplama ve/veya İşleme - H.İ.Y., A.K.; Analiz

ve/veya Yorum - H.İ.Y., A.K.; Literatür Taraması - H.İ.Y.;

Yazıyı Yazan - H.İ.Y.; Eleştirel İnceleme - H.İ.Y., A.K.

REFERENCES

1. Hyduk A, Croft JB, Ayala C, Zheng K, Zheng ZJ, Mensah

GA. Pulmonary hypertension surveillance--United States,

1980-2002. MMWR Surveill Summ 2005; 54:1-28.

2. Sunar H, Yıldızeli B, Taş S, Yanartaş M, saçlı H, Kış M, et

al. Pulmonary endarterectomy in chronic thromboembolic

pulmonary hypertension. Turk Gogus Kalp Damar 2013;

21:7-13. [CrossRef]

3. Olman MA, Auger WR, Fedullo PF, Moser KM. Pulmo-

nary vascular steal in chronic thromboembolic pulmonary

hypertension. Chest 1990; 98:1430-4. [CrossRef]

4. Lee KC, Cho YL, Lee SY. Reperfusion pulmonary edema

after pulmonary endarterectomy. Acta Anaesthesiol Sin

2001; 39:97-101.

5. Simonneau G, Robbins IM, Beghetti M, Channick RN,

Delcroix M, Denton CP, et al. Updated clinical classifica-

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 69

tion of pulmonary hypertension. J Am Coll Cardiol 2009;

30 (1 Suppl):S43-54. [CrossRef]

6. Riedel M, Stanek V, Widimsky J, Prerovsky I. Longterm

follow-up of patients with pulmonary thromboembolism.

Late prognosis and evolution of hemodynamic and res-

piratory data. Chest 1982; 81:151-8. [CrossRef]

7. Galié N, Humbert M, Vachiery JL, Gibbs S, Lang

I,Torbicki A, et al. 2015 ESC/ERS Guidelines for the di-

agnosis and treatment of pulmonary hypertension: The

Joint Task Force for the Diagnosis and Treatment of Pul-

monary Hypertension of the European Society of Cardi-

ology (ESC) and the European Respiratory Society (ERS):

Endorsed by: Association for European Paediatric and

Congenital Cardiology (AEPC), International Society for

Heart and Lung Transplantation (ISHLT) Eur Heart J

2016;37:67-119. [CrossRef]

8. D'Armini AM, Zanotti G, Viganò M. Pulmonary endarter-

ectomy: the treatment of choice for chronic thromboem-

bolic pulmonary hypertension. Ital Heart J 2005; 6:861-

8.

9. Hagl C, Khaladj N, Peters T, Hoeper MM, Logemann F,

Haverich A, et al. Technical advances of pulmonary

thromboendarterectomy for chronic thromboembolic

pulmonary hypertension. Eur J Cardiothorac Surg 2003;

23:776-81. [CrossRef]

10. Mellemkjaer S, Ilkjaer LB, Klaaborg KE, Christiansen CL,

Severinsen IK, Nielsen-Kudsk JE, et al. Pulmonary

endarterectomy for chronic thromboembolic pulmonary

hypertension. Ten years experience in Denmark. Scand

Cardiovasc J 2006; 40:49-53. [CrossRef]

11. Rubin LJ, Hoeper MM, Klepetko W, Galiè N, Lang IM,

Simonneau G. Current and future management of chron-

ic thromboembolic pulmonary hypertension: from diag-

nosis to treatment responses. Proc Am Thorac Soc 2006;

3:601-7. [CrossRef]

12. Ghofrani HA, D'Armini AM, Grimminger F, Hoeper MM,

Jansa P, Kim NH, et al. Riociguat for the treatment of

chronic thromboembolic pulmonary hypertension. N Engl

J Med 2013; 369:319-29. [CrossRef]

Respir Case Rep 2020;9(2): 70-73 DOI: 10.5505/respircase.2020.13540

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70

Derya Yenibertiz, Berna Akıncı Özyürek, Yurdanur Erdoğan

The most common and important side-effect of war-

farin treatment is bleeding. Erythema nodosum, in the

form of painful, erythematous nodules in the dermal

and subcutaneous tissues, is not a known side-effect

of warfarin. In this case, we report on erythema

nodosum occurring as a side-effect of warfarin treat-

ment. A 49-year-old female patient was treated for

pulmonary thromboembolism after multiple lesions

emerged identified as erythema nodosum in a der-

matology consultation that were interpreted as a

possible side-effect of warfarin treatment. It was con-

cluded that erythema nodosum may present as a

side-effect of warfarin.

Key words: Pulmonary thromboembolism, warfarin,

erythema nodosum.

Warfarin tedavisine bağlı en sık ve en önemli yan etki

kanamadır. Dermal ve subkutan dokularda ağrılı,

eritemli nodüllerden oluşan eritema nodozum warfa-

rinin bilinen bir yan etkisi değildir. Warfarine tedavisi-

ne bağlı gelişen eritema nodozum olgumuzu sunmayı

amaçladık. Pulmoner tromboemboli tedavisi başla-

nan 49 yaşında kadın hastada warfarin kullanımına

bağlı dermatoloji konsültasyonu ile eritema nodozum

olarak tanımlanan çok sayıda lezyon saptandı. War-

farin tedavisine bağlı bir yan etki olarak eritema no-

dozum görülebileceğini vurgulamak istedik.

Anahtar Sözcükler: Pulmoner tromboemboli, warfarin,

eritema nodozum.

Sağlık Bilimleri Üniversitesi, Atatürk Göğüs Hastalıkları ve Göğüs

Cerrahisi Eğitim Araştırma Hastanesi, Ankara

University of Health Sciences, Ataturk Chest Diseases and

Chest Surgery Training and Research Hospital, Ankara, Turkey

Submitted (Başvuru tarihi): 21.09.2019 Accepted (Kabul tarihi): 16.01.2020

Correspondence (İletişim): Berna Akıncı Özyürek, Sağlık Bilimleri Üniversitesi, Atatürk Göğüs Hastalıkları ve Göğüs Cerrahisi Eğitim

Araştırma Hastanesi, Ankara

e-mail: drberna_1982@yahoo.com

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Pulmonary thromboembolism (PTE) is the result of a clot

in the pulmonary artery or in one of its branches, and is

associated with high morbidity and mortality. Patient-

specific treatment is guided by signs and symptoms,

bleeding risk and comorbidities. Warfarin sodium – a

vitamin K antagonist – is an effective option for the treat-

ment of PTE, despite its narrow therapeutic index, its wide

inter-patient dosing variability, its predisposition to drug

and food interactions, and the need for close monitoring

of the intensity of the anticoagulation effect using the

international normalized ratio (INR). The most common

and important side effect of warfarin treatment is bleeding

(1). Several adverse skin manifestations have been asso-

ciated with the use of oral anticoagulants, ranging from

ecchymosis and purpura, hemorrhagic necrosis and

maculopapular vesicular urticarial eruptions, to purple

toes (2). While skin necrosis has been mentioned as the

most common dermatological side-effect of warfarin in

literature (2-7), we could find no studies in literature iden-

tifying erythema nodosum as a warfarin-related side-

effect.

In this case, we report that erythema nodosum may occur

as a side effect of warfarin treatment.

CASE

A 49-year-old, non-smoking, female patient applied to

our hospital emergency department with a sudden onset

of dyspnea and cough. The patient reported no other

symptoms, such as fever, malaise, fatigue, weight loss,

dysuria or sputum, and there were no signs of infection in

the patient. Upon hospitalization, thrombus was detected

in the subsegmentary arteries at the level of both postero-

basal segments in the lungs in a thorax computed tomog-

raphy angiography (Figure 1). The patient had neither

identified history of chronic medical disease nor a re-

markable family medical history. Her physical examina-

tion was normal and all laboratory parameters, include

C-reactive protein and white blood cells, were normal

with the exception of a raised d-dimer. Low molecular

weight heparin and warfarin treatment were started simul-

taneously; the patient was treated with no other drugs.

When the INR value reached the desired range due to the

effective dose of warfarin, the low molecular weight

heparin was stopped and treatment continued with warfa-

rin. On the 7th day of warfarin treatment, multiple painful,

swollen, nodular indurations emerged all over the body

(Figure 2 and 3). The patient was passed to the derma-

tology and allergy clinic, and the lesions were linked to

the warfarin treatment. Warfarin was stopped, and treat-

ment for erythema nodosum was carried out, as per the

dermatologist’s suggestion. The skin lesions regressed on

follow-up.

DISCUSSION

Erythema nodosum is a form of acute nodular septal

panniculitis, characterized by the sudden onset of ery-

thematous, firm, solid, deep nodules or plaques that are

painful on palpation, and localized mainly on the exten-

sor surfaces of the legs. It occurs more often in women

aged 25–40 years, but can be observed at any age (8).

Erythema nodosum may be linked to a variety of causes,

such as infection, medications, sarcoidosis, pregnancy,

inflammatory bowel disease, vaccination, autoimmune

disease and malignancy, among others. The condition is

idiopathic in approximately 50% of cases. Diagnosis is

generally made clinically, but a biopsy may be required in

atypical cases (9). A skin biopsy is generally not necessary

if the history and physical signs are suggestive of EN, and

the treatment of EN depends on the suspected or docu-

mented etiology, if known (10).

Our patient, a woman aged 49-year-old, presented with

lesions that were diagnosed as erythema nodosum by a

dermatologist. No biopsy was performed as the lesions

were considered typical. Most cases of erythema

nodosum are self-limited and require no treatment. Bed

rest and leg elevation are generally recommended to

reduce discomfort. Nonsteroidal anti-inflammatory drugs

are the first-line treatment for pain management. The

dermatologist started the patient on Colchium Dispert

and nonsteroidal anti-inflammatory drugs.

The present study emphasizes the rarity of erythema

nodosum as a side-effect of warfarin.

CONCLUSION

This article reports on a case of erythema nodosum that

emerged due to warfarin treatment for a pulmonary

thromboembolism.

Figure 1: Computed tomography angiography of the thorax

Erythema Nodosum due to Warfarin Treatment in Pulmonary Thromboembolism: A Case Report | Akıncı Özyürek et al.

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Figure 2: Erythema nodosum on the leg

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - D.Y., B.A.Ö., Y.E.; Planning and Design - D.Y.,

B.A.Ö., Y.E.; Supervision - D.Y., B.A.Ö., Y.E.; Funding -

D.Y., B.A.Ö.; Materials - D.Y., B.A.Ö.; Data Collection

and/or Processing - D.Y., B.A.Ö.; Analysis and/or Inter-

pretation - E D.Y., B.A.Ö.; Literature Review - D.Y.,

B.A.Ö.; Writing - D.Y.; Critical Review - D.Y., B.A.Ö.

YAZAR KATKILARI

Fikir - D.Y., B.A.Ö., Y.E.; Tasarım ve Dizayn - D.Y.,

B.A.Ö., Y.E.; Denetleme - D.Y., B.A.Ö., Y.E.; Kaynaklar -

D.Y., B.A.Ö.; Malzemeler - D.Y., B.A.Ö.; Veri Toplama

ve/veya İşleme - D.Y., B.A.Ö.; Analiz ve/veya Yorum -

D.Y., B.A.Ö.; Literatür Taraması - D.Y., B.A.Ö.; Yazıyı

Yazan - D.Y.; Eleştirel İnceleme - D.Y., B.A.Ö.

Figure 2: Erythema nodosum on the arm

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REFERENCES

1. Ageno W, Gallus AS, Wittkowsky A, Crowther M, Hylek

EM, Palareti G. Oral anticoagulant therapy: antithrom-

botic therapy and prevention of thrombosis, 9th ed:

American College Of Chest Physicians Evidence- Based

Clinical Practice Guidelines. Chest 2012; 141: e44S-

e88S. [CrossRef]

2. Chan YC, Valenti D, Mansfield AO, Stansby G. Warfarin

induced skin necrosis. Br J Surg 2000; 87: 266-72.

[CrossRef]

3. Eichhoff G. Warfarin induced skin necrosis within psoriat-

ic plaques Dermatol Online J 2019; 25(6): pii:

13030/qt4gf5r5qk.

4. Tilton C, Livengood S, Hodges J, Marshall J. Warfarin-

induced skin necrosis in the presence of acute hepatic in-

jury and May-Thurner Syndrome. Hosp Pharm 2019; 54:

130- 4. [CrossRef]

5. Fraga R, Diniz LM, Lucas EA, Emerich PS. Warfarin-

induced skin necrosis in a patient with protein S deficien-

cy. An Bras Dermatol 2018; 93: 612- 3. [CrossRef]

6. Sklar LR, Messman A. An atypical case of warfarin-

induced skin necrosis. Clin Pract Cases Emerg Med 2017;

1: 359-61. [CrossRef]

7. Hamada T, Miyake T, Otsuka M, Iwatsuki K. Warfarin-

induced skin necrosis accompanied by aggravation of

vasculitis in a patient with cutaneous arteritis. Int J Der-

matol 2017; 56:779-81. [CrossRef]

8. Hafsi W, Badri T. Erythema Nodosum. [Updated 2019

Dec 20]. In: StatPearls [Internet]. Treasure Island (FL):

StatPearls Publishing; 2020 Jan-. Available from:

https://www.ncbi.nlm.nih.gov/books/NBK470369/

9. Leung AKC, Leong KF, Lam JM. Erythema nodosum.

World J Pediatr 2018; 14:548-54. [CrossRef]

10. Allen RA. Erythema Nodosum. Clinical Gate Dermatolo-

gy 18/ 03/ 2015. Available from:

https://clinicalgate.com/erythema-nodosum-2/

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Ezgi Çimen Çelik1, Serkan Yazgan

1, Soner Gürsoy

1, Ahmet Ucvet

1, Zekiye Aydoğdu

2

Amyloidosis is a disease that is characterized by an

extracellular accumulation of fibril proteins called

amyloid in tissues, and organ dysfunction. There are

various types of amyloidosis, with nodular pulmonary

amyloidosis usually considered a subtype of AL amy-

loidosis. Surgical excision is usually curative and the

prognosis is excellent. This case is presented to em-

phasize the rare occurrence of pulmonary amyloido-

sis and the need to keep malignancies in mind in

differential diagnosis.

Key words: Amiloidosis, breast cancer, metastasis.

Amiloidoz, dokularda amiloid adı verilen fibril yapı-

sındaki proteinlerin ekstrasellüler birikimi ve organ-

larda işlev bozukluğu ile seyreden hastalıktır. Çeşitli

tipleri olup, nodüler pulmoner amiloidoz, genellikle

AL amiloidozun bir subtipi olarak görülür. Cerrahi

eksizyon genellikle küratiftir ve prognoz mükemmel-

dir. Bu olgu, pulmoner amiloidozun nadiren görül-

mesi, ayırıcı tanıda malignitelerin akılda tutulması

gerekliliğini vurgulamak amacıyla sunulmuştur.

Anahtar Sözcükler: Amiloidoz, meme karsinomu,

metastaz.

1Department of Thoracic Surgery, University of Health Sciences,

İzmir Dr Suat Seren Chest Diseases and Surgery Medical Practice

and Research Center, İzmir, Turkey

2Department of Pathology, University of Health Sciences, İzmir Dr

Suat Seren Chest Diseases and Surgery Medical Practice and Re-

search Center, İzmir, Turkey

1SBÜ İzmir Dr. Suat Seren Göğüs Hasalıkları ve Cerrahisi

SUAM, Göğüs Cerrahisi Kliniği Kliniği, İzmir

2SBÜ İzmir Dr. Suat Seren Göğüs Hasalıkları ve Cerrahisi

SUAM, Patoloji Bölümü, İzmir

Submitted (Başvuru tarihi): 24.12.2019 Accepted (Kabul tarihi): 24.02.2020

Correspondence (İletişim): Ezgi Çimen Çelik, Department of Thoracic Surgery, University of Health Sciences, İzmir Dr Suat Seren

Chest Diseases and Surgery Medical Practice and Research Center, İzmir, Turkey

e-mail: ezgi.guvenc@yahoo.com

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Amyloidosis is characterized by an accumulation of con-

gophilic amyloid fibrin deposits in the extracellular matrix

or organs (1). The most common forms are systemic AL

amyloidosis, systemic AA amyloidosis, systemic wild-type

ATTR amyloidosis, systemic hereditary ATTR amyloidosis

and localized AL amyloidosis (2). Nodular pulmonary

amyloidosis is a usual subtype of localized AL amyloidosis

(3) that may be associated with underlying inflammatory

or malignant conditions. Patients are generally asympto-

matic (4,5). Lesions are usually solitary, and may be mis-

taken for pulmonary malignancies. Biopsy material is

stained with Congo-Red to give a green color under a

polarized microscope, which is diagnostic (6). Excision of

the nodules is usually curative and the prognosis is excel-

lent (7). The case we present here is intended to empha-

size that pulmonary amyloidosis is rare and may be mis-

taken for malignancies.

CASE

A 76-year-old female patient who underwent a right mas-

tectomy 10 years previously for breast apocrine carcino-

ma followed by chemotherapy and radiotherapy, was

diagnosed with pulmonary nodules in radiological follow-

up and referred to us. No pathology was detected aside

from the old operation scar on the right breast upon

physical examination. Increased opacity was noted in the

left paracardiac area on chest radiography. A thorax

computed tomography (CT) showed a 13x15 mm solid

nodule in the left upper lobe (Figure 1 and 2), a 15x20

mm nodule in the lingual (Figure 3 and 4), and an 6x8

mm nodule in the left lower lobe (Picture 5). A positron

emission tomography/computed tomography (PET-CT)

revealed a 1.5x1 cm nodule (SUVmax: 1.6) in the left

upper lobe and a nodule of 2.2x1.4 cm (SUVmax: 1.4) in

the lingula (Figures 6, 7 and 8). Since the patient could

not be diagnosed through bronchoscopy and transthorac-

ic, a fine needle aspiration biopsy (TTFNAB), operation

was selected for diagnosis and treatment. In an explora-

tion with a left thoracotomy, four nodules in the left upper

lobe and one nodule in the lower lobe were palpated. A

wedge resection was performed on the three peripheral

nodules in the left upper lobe and in the lower lobe, and

the frozen section was examined. The frozen result report

read “The nodules may be breast carcinoma metastasis,

but a definitive diagnosis should be confirmed with further

investigations.” A left upper lobectomy was performed,

since the other central lesion in the upper lobe could not

be completely removed due to its closeness to the superi-

or pulmonary vein. The final pathology was reported as

nodular pulmonary amyloidosis (Figures 9, 10 and 11).

The patient was discharged on the 7th postoperative day

and is now in the 4th postoperative month.

Figure 1: Thorax computed tomography showing a 13x15 mm solid

nodule in left lung upper lobe

Figure 2: Thorax computed tomography showing a 13x15 mm solid

nodule in left lung upper lobe (Coronal section)

Figure 3: Thorax computed tomography showing a 15x20 mm nodule

in left lung lingual lobe

Nodular Pulmonary Amyloidosis Mimicking Breast Carcinoma Metastasis | Çimen Çelik et al.

76 www.respircase.com

Figure 4: Thorax computed tomography showing a 15x20 mm nodule

in left lung lingual lobe (sagittal section)

Figure 5: Thorax computed tomography showing a 6x8 mm nodule in

left lung lower lobe

Figure 6: PET /CT showing a 1.5x1 cm nodule (SUVmax: 1.6) in left

lung upper lobe

DISCUSSION

Nodular pulmonary amyloidosis, known also as nodular

parenchymal amyloidosis or nodular amyloidoma, is a

disease that is characterized by the presence of one or

more tumor-like amyloid deposits in the lung (1). It was

first described by Virchow in 1857 (7). It is rarely seen

among the amyloid diseases (3,6), and is characterized

primarily by a congophilic light chain amyloid (AL) depo-

sition in the extracellular matrix, and may be solitary or,

as with tour patient, multiple (1,8). It is usually diagnosed

in the sixth decade and is more common in men (1,9).

Patients are generally asymptomatic, as in our patient

(4,5), although it may cause cough, hemoptysis, dyspnea,

pleural effusion and pulmonary arterial hypertension,

depending on the location (10). It has no specific radio-

logical findings and mimics malignancies radiologically.

The present case emphasizes that pulmonary amyloidosis

is rare and may be mistaken for malignancies. Our pa-

tient was operated on due to the suspicion of breast car-

cinoma metastasis.

18-Fluoro-deoxyglucose (18F-FDG) PET-CT has emerged

as a tool for the diagnosis of pulmonary nodules to re-

duce invasive diagnostic examination. However, 18F-

FDG shows a small amount of uptake in malignancies

with low metabolic activity, such as bronchoalveolar can-

cer, carcinoid tumor and mucinous adenocarcinoma. As

such, despite low involvement on PET-CT (SUVmax: 1,6),

malignancies can be considered in a pre-diagnosis. Fur-

thermore, it has high metabolic rates alongside such non-

malignant conditions as tuberculosis, sarcoidosis and

rheumatoid nodules. In our case, and as in other rarely

reported cases, pulmonary nodular amyloidosis with low

or moderate 18F-FDG involvement can be seen (11). As

such, the results of an 18F-FDG PET-CT should be inter-

preted with caution in the differentiation of pulmonary

amyloidosis from other malignant or benign lesions. A

definitive diagnosis of localized pulmonary amyloidosis

requires histological confirmation (12). In cases in which

patient cannot be diagnosed via a CT-guided fine needle

aspiration biopsy, an invasive surgical resection may be

necessary. The patient in the present study could not be

diagnosed via TFNAB, and so exploratory thoracotomy

was decided upon. Furthermore, in the frozen examina-

tion, no differential diagnosis from malignancies could be

made.

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 77

Figure 7: PET /CT showing a calcific nodul in left lung upper lobe

Figure 8: PET /CT showing a 2.2x1.4 cm nodule (SUVmax: 1.4) in left

lung lingula lobe

Figure 9: Congo red X 200

Figure 10: Acellular hyalinized material (H&E X200)

Figure 11: Acellular eosinophilic material (H&E X200)

Pulmonary amyloidosis may occur as a component of

localized or systemic amyloidosis (12,13). Localized AL

amyloidosis has a better prognosis than systemic amyloi-

dosis. The 10-year survival rate after surgery is reported

to be 97%. A surgical resection can be performed safely,

and the prognosis is excellent (13). Our patient continues

to be followed-up without problem in the postoperative

4th month.

In conclusion, local nodular pulmonary amyloidosis is a

rare and unusual tumor of the lung, and surgical treat-

ment is curative. Nodules may be solitary or multiple. It

should be kept in mind that amyloidosis may mimic both

benign and malignant pathologies, and should be con-

sidered in a differential diagnosis.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Planning and

Design - E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Supervision -

E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Funding - E.Ç.Ç., Z.A.;

Materials – S.Y., E.Ç.Ç., Z.A.; Data Collection and/or

Processing - E.Ç.Ç., Z.A.; Analysis and/or Interpretation -

S.Y., S.G., E.Ç.Ç.; Literature Review - E.Ç.Ç.; Writing -

E.Ç.Ç.; Critical Review - S.Y., A.U., S.G.

YAZAR KATKILARI

Fikir - E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Tasarım ve Dizayn -

E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Denetleme - E.Ç.Ç., S.Y.,

S.G., A.U., Z.A.; Kaynaklar - E.Ç.Ç., Z.A.; Malzemeler -

S.Y., E.Ç.Ç., Z.A.; Veri Toplama ve/veya İşleme - E.Ç.Ç.,

Z.A.; Analiz ve/veya Yorum - S.Y., S.G., E.Ç.Ç.; Literatür

Taraması - E.Ç.Ç.; Yazıyı Yazan - E.Ç.Ç.; Eleştirel İnce-

leme - S.Y., A.U., S.G.

Nodular Pulmonary Amyloidosis Mimicking Breast Carcinoma Metastasis | Çimen Çelik et al.

78 www.respircase.com

REFERENCES

1. Khoor A, Colby TV. Amyloidosis of the Lung. Arch Pathol

Lab Med 2017; 141:247-54. [CrossRef]

2. Sipe JD, Benson MD, Buxbaum JN, Ikeda S, Merlini G,

Saraiva MJ, et al. Nomenclature 2014: amyloidfibril pro-

teins and clinical classification of the amyloidosis. Amy-

loid 2014; 21:221-4. [CrossRef]

3. Kaplan B, Martin BM, Boykov O, Gal R, Pras M, Shecht-

man I, et al. Co-deposition of amyloidogenic immuno-

globulin light and heavy chains in localized pulmonary

amyloidosis. Virchows Arch 2005; 447:756-61.

[CrossRef]

4. Beer TW, Edwards CW. Pulmonary nodules due to reac-

tive systemic amyloidosis (AA) in Crohn's disease. Thorax

1993; 48:1287-8. [CrossRef]

5. Roden AC, Aubry MC, Zhang K, Brady JO, Levin D,

Dogan A, et al. Nodular senile pulmonary amyloidosis: a

unique case confirmed by immunohistochemistry, mass

spectrometry, and genetic study. Human Pathology 2010;

41:1040-5. [CrossRef]

6. Howie AJ, Brewer DB. Optical properties of amyloid

stained by Congo red: history and mechanisms. Micron

2009; 40:285-301. [CrossRef]

7. Utz JP, Swensen SJ, Gertz MA. Pulmonary amyloidosis

The Mayo Clinic experience from 1980 to 1993. Ann In-

tern Med 1996; 124;4, 407-13. [CrossRef]

8. Milani P, Basset M, Russo F, Foli A, Palladini G, Merlini

G. The lung in amyloidosis. Eur Respir Rev 2017;

26(145): pii: 170046. [CrossRef]

9. Yang MC, Blutreich A, Das K. Nodular pulmonary amy-

loidosis with an unusual protein composition diagnosed

by fine-needle aspiration biopsy: a case report. Diagn

Cytopathol 2009; 37:286-9. [CrossRef]

10. Scala R, Maccari U, Madioni C, Venezia D, La Magra LC.

Amyloidosis involving the respiratory system: 5-year's ex-

perience of a multi-disciplinary group's activity. Ann

Thorac Med 2015, 10:212-6. [CrossRef]

11. Standaert C, Herpels V, Seynaeve P. A solitary pulmonary

nodule: pulmonary amyloidosis. J Belg Soc Radiol 2018;

102:20. [CrossRef]

12. Chen KT. Amyloidosis presenting in the respiratory tract.

Pathol Annu 1989; 24:253-273.

13. Baumgart JV, Stuhlmann-Laeisz C, Hegenbart U, Nat-

tenmüller J, Schönland S, Krüger S, et al. Local vs. sys-

temic pulmonary amyloidosis-impact on diagnostics and

clinical management. Virchows Arch 2018; 473:627-37.

[CrossRef]

Respir Case Rep 2020;9(2): 79-82 DOI: 10.5505/respircase.2020.99267

OLGU SUNUMU CASE REPORT

79

Ayse Baccioglu1, Ayse Füsun Kalpaklioglu

1, Tuba Devrim

2

Ultraviolet recall is a photodermatitis reaction that

can occur in prior ultraviolet burned skin during the

administration of systemic medication. No such reac-

tion has been reported with pirfenidone. We report

here on a 75-year-male patient who developed acute

erosive erythema on his face, forearms and hands

under pirfenidone treatment for idiopathic pulmonary

fibrosis after 4 months. The initial diagnosis was drug

eruption, since it developed after the initiation of

pirfenidone, in accordance with hypereosinophilia

and solar dermatitis on a skin biopsy, all of which

improved with discontinuation. However, the patient

tolerated the rechallange test with pirfenidone. The

presence of necrotic keratinocytes in a skin biopsy

and exaggerated dermatitis was unlikely for photo-

dermatitis, but supported an ultraviolet recall reaction.

Pirfenidone was resumed in a tapered dose, and the

patient was successfully followed up for 5 months for

a relapse of skin reaction, as well as IPF disease

activity. This case is important in indicating that the

drug can be tolerated with dose adjustment in the

presence of an ultraviolet recall reaction in contrast

to discontinuation need in drug allergy.

Key words: Drug eruption, phototoxicity, pirfenidone,

ultraviolet recall phenomen.

Ultraviyole hatırlama reaksiyonu, önceden ultraviyole

ışınına bağlı yanık gelişmiş kişinin sistemik ilaç kulla-

nımı sonrası benzer kliniğin gelişmesiyle karakterize

bir fotodermatittir. Pirfenidonla bu reaksiyon daha

önce bildirilmemiştir. Yetmiş beş yaşındaki erkek

hastada idiopatik pulmoner fibrozis için pirfenidon

başlandıktan 4 ay sonra güneş gören deri alanların-

da akut erosiv eritem gelişmişti. Önce pirfenidona

bağlı ilaç erüpsiyonu olduğu düşünüldü, çünkü; eşlik

eden hipereozinofilisi, deri biyopsisinde solar dermati-

tis sonucu vardı ve belirtiler pirfenidondan sonra

gelişmiş ve ilacın kesilmesinden sonra düzelmişti.

Ancak, oral provokasyon testinde hastanın ilacı tolere

etmesi, deri lezyonlarının basit bir solar dermatitis için

çok şiddetli olması ve deri biyopsisinde nekrotik kera-

tinositlerin görülmesi ultraviyole hatırlama reaksiyo-

nunu düşündürdü. Pirfenidon dozu düşürülerek yeni-

den başlandı, 5 aylık takip süresinde deri lezyonları

nüks etmedi ve İPF hastalığında atak gelişmedi. İlaç

allerjisinden farklı olarak pirfenidon ultraviyole hatır-

lama reaksiyonunda, tedaviye doz ayarlamasıyla

devam edilebileceğini göstermesi açısından bu olgu

önemlidir.

Anahtar Sözcükler: İlaç erupsiyonu, fototoksisite,

pirfenidon, ultraviyole hatırlama reaksiyonu.

1Department of Pulmonary Diseases, Kırıkkale University Faculty of

Medicine, Division of Immunology and Allergy, Kırıkkale, Turkey

2Department of Pathology, Kırıkkale University Faculty of Medi-

cine, Kırıkkale, Turkey

1Kırıkkale Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları

Anabilim Dalı, İmmünoloji ve Allerji Bilim Dalı, Kırıkkale

2Kırıkkale Üniversitesi Tıp Fakültesi, Patoloji Anabilim Dalı,

Kırıkkale

Submitted (Başvuru tarihi): 04.12.2019 Accepted (Kabul tarihi): 25.02.2020

Correspondence (İletişim): Ayse Baccioglu, Department of Pulmonary Diseases, Kırıkkale University Faculty of

Medicine, Division of Immunology and Allergy, Kırıkkale, Turkey

e-mail: aysebaccioglu@gmail.com

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Ultraviolet recall reaction is a type of photodermatitis that

occurs in ultraviolet-burned skin after the administration

of systemic medications (1). Such reactions are rare, and

have no report by pirfenidone, which is an oral antifibrot-

ic agent approved for patients with idiopathic pulmonary

fibrosis (IPF) (2). We present here a case of ultraviolet

recall photodermatitis by pirfenidone.

CASE

A 75-year-old Caucasian man was admitted with an

extraordinary sunburn like scaly erythema on his face,

forearms and hands that had emerged a few days earlier.

He had no recent sun exposure, and limited physical

movement that made it almost impossible for him to go

outside. He had been undergoing treatment with

pirfenidone (2403 mg/day) for IPF besides inhaled tiotro-

pium, and budesonide/formoterol for 4 months, and

subsequently developed a desquamative, burning and

erosive erythema limited on sun exposed areas such as

the forehead, scalp, face, neck, dorsa of the forearms

and hands (Figure 1). He had also vitiligo patches on his

face, neck and forearms surrounded by a reddish ul-

tralight-colored skin.

Laboratory tests revealed elevated eosinophil counts

(1400/mL) and a normal total leukocyte count

(8.600/mL), in addition to vitamin D deficiency

(10.55ng/dL, normal; 25–80ng/dL). Renal and hepatic

function tests were within normal limits. Serum ANA, ccp,

ANCA, ACE, anti-ds-DNA/jo1/scl70/sm/ssa/ssb were

also negative. Chest x-ray and pulmonary function tests

were stable when compared to previous values.

Figure 1: Severe erythema of the face (a), desquamative and erosive

erythema on the back of hands (b)

Histopathological findings of the biopsied forearm skin

revealed solar dermatitis with keratinocyte necrosis,

spongiosis with a lichenoid reaction, focal parakeratosis,

and irregular acanthosis in the epidermis, vacuolization,

perivascular lymphocytic cell infiltration and no specific

deposition of immunoglobulin or complements on the

basement membrane or vessel walls (Figure 2).

Pirfenidone was discontinued, and the patient was treated

with 40 mg/day (0.5 mg/kg/day) of intravenous methyl-

prednisolone for 10 days, with slight improvement. The

reaction was concluded to be a phototoxicity reaction in

the sun-exposed areas, in addition to solar dermatitis on

a skin biopsy. It was also considered to be a drug erup-

tion, having developed during pirfenidone treatment, in

accordance with serum hypereosinophilia, and after im-

provement was noted upon discontinuation. However, the

reaction time as of March was unexpected for photoreac-

tivity given the rarity of sunny days, and the solar dermati-

tis clinic was so severe with rapid progression. According-

ly, to confirm the diagnosis, an oral provocation test was

carried out 4 weeks after recovery in May, and the patient

was found to tolerate pirfenidone. The reaction was thus

concluded to be an ultraviolet recall reaction. The pres-

ence of necrotic keratinocytes in the skin biopsy was un-

likely for photodermatitis, but also supported this diagno-

sis. Even though the patient denied sun exposure, the

weather had been slightly sunny a few days before the

reaction, and this limited sun may have recalled the solar

dermatitis experienced in previous years to his extremely

white skin with vitiligo.

Pirfenidone was resumed in a tapered dose (1602

mg/day) following the complete regression of skin lesions

and the suggestion of ultraviolet recall dermatitis rather

than of drug eruption. The patient was successfully fol-

lowed up for 5 months for relapses of the skin reaction,

as well as disease activity of IPF.

DISCUSSION

Even though phototoxicity due to pirfenidone is a relative-

ly common reaction, this case report is important in its

rarity in terms of the tolerability of the drug after recovery.

The most common skin side-effects of pirfenidone have

been reported as 7.5% rash, 4.2% photosensitivity and

generalized pruritis in a single case, but no report of

ultraviolet recall reaction (3-6).

Photodermatitis following the Use of Pirfenidone in a Patient with Idiopathic Pulmonary Fibrosis: An Ultraviolet Recall Reaction | Baccioglu et al

81 www.respircase.com

Figure 2: Focal parakeratosis and irregular acanthosis in the superficial

layer, hydropic degeneration and necrotic keratinocytes in the epidermis

(H&E x40)

A diagnosis of ultraviolet recall reaction was made in the

presence of previous sunburn history accompanied by

systemic pirfenidone use, exaggerated clinic, skin biopsy

and tolerance in a rechallange test. Necrotic keratino-

cytes, epidermal spongiosis and vesiculation were sensi-

tive to ultraviolet recall reaction. Even though the history

of sun exposure was unremarkable, but had lived many

sunburn events in the past leaving behind photo aging

findings. The duration between sunburn and drug intake

varies, between days associated with chemotherapy med-

ications, and months in ampicillin (7,8). The mean dura-

tion between the initiation of pirfenidone and the devel-

opment of skin manifestations was 5.5 months in litera-

ture, and 4 months in the present case (3-6).

Photosensitivity reactions take two forms: phototoxicity,

occurring in sun exposed areas, whereas a generalized

rash is accepted as a photoallergic reaction. Photoaller-

gic reactions represent type IV hypersensitivity responses,

requiring a specific sensitization of the drug that makes

intolerance in rechallange (9). Phototoxicity has non-

immunological mechanism as direct cellular damage by

ultraviolet light (10). Ultraviolet recall refers to a photo-

toxicity reaction that initiates with irradiation and exacer-

bates with systemic medication (1). The reintroduction of

the drug or light exposure may not necessarily cause a

recurrence. Pirfenidone is known to become reactive

through the absorption of light, and the generation of

reactive oxygen radicals mediates to photo irritant results

(10). Another underlying mechanism is likely that

pirfenidone’s antifibrotic effect through suppressing some

inflammatory cytokines that are associated with viral reac-

tivation inducing cutaneous manifestation in some pa-

tients (4). Cofactors for photosensitivity are medications,

lupus and some vitamin deficiencies (9). The patient re-

fused systemic drugs other than pirfenidone before 4

weeks of the reaction time, and lupus was ruled out

based on clinical and negative collagen markers. The

patient’s low vitamin D level may be a result of long-time

sun avoidance, but it has been reported that photosensi-

tivity is not correlated with vitamin D levels (9). It may,

however, be possible to become more sensitive to sun-

light after winter.

Treatments of pirfenidone phototoxicity are topical emol-

lients and in mild-moderate events, while in severe cases,

systemic steroids may be required. A reduction in

pirfenidone dose may be a preventive action, since it has

been reported to be non-phototoxic in 30 mg/kg, but

phototoxic in 160 mg/kg (10). Even though ultraviolet

recall does not necessitate a withdrawal of therapy, many

adverse skin-related events related to pirfenidone may

indicate a discontinuation of the drug, and the appliance

of strict sun exposure may be helpful. In the event of a

reappearance of an adverse event or exacerbation

and/or progression of the disease, nintedanib, as an

alternative anti-fibrotic medication, would have been

started instead of pirfenidone. However, nintedanib was

not the first choice therapy in the present case as the

patient had been using an anticoagulation drug for car-

diac disease, and nintedanib may be associated with an

increased risk of bleeding.

Conclusion

This case report is interesting in that it reminds that a

photosensitive reaction experienced under pirfenidone

may not be due only to a drug allergy or solar dermatitis,

as it may also indicate an ultraviolet recall reaction,

which the drug can be tolerated in rechallange. Although

it has been reported to be a rare and moderate adverse

event, photosensitivity reaction can reduce quality of life

and to potentially increase the risk of the development of

skin cancer. Clinicians should pay particular attention to

risk factors for photosensitization, such as light skin color

and vitiligo, when making a decision for the treatment of

IPF.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - A.B., A.F.K., T.D.; Planning and Design - A.B.,

A.F.K., T.D.; Supervision - A.B., A.F.K., T.D.; Funding -;

Materials - A.B.; Data Collection and/or Processing -

.Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 82

A.B., T.D.; Analysis and/or Interpretation - A.B.; Literature

Review - A.B.; Writing - A.B.; Critical Review - A.B., A.F.K.

YAZAR KATKILARI

Fikir - A.B., A.F.K., T.D.; Tasarım ve Dizayn - A.B., A.F.K.,

T.D.; Denetleme - A.B., A.F.K., T.D.; Kaynaklar -; Mal-

zemeler - A.B.; Veri Toplama ve/veya İşleme - A.B., T.D.;

Analiz ve/veya Yorum - A.B.; Literatür Taraması - A.B.;

Yazıyı Yazan - A.B.; Eleştirel İnceleme - A.B., A.F.K.

REFERENCES

1. Shiohara T, Mizukawa Y. Recall phenomenon: some

skin-resident cells remember previous insults. Dermatolo-

gy 2003; 207:127-9. [CrossRef]

2. Noble PW, Albera C, Bradford WZ, Costabel U, Glass-

berg MK, Kardatzke D, et al. Pirfenidone in patients with

idiopathic pulmonary fibrosis (CAPACITY): two random-

ized trials. Lancet 2011; 377:1760-9. [CrossRef]

3. Costabel U, Bendstrup E, Cottin V, Dewint P, Egan JJ,

Ferguson J, et al. Pirfenidone in idiopathic pulmonary fi-

brosis: expert panel discussion on the management of

drug-related adverse events. Adv Ther 2014; 31:375-91.

[CrossRef]

4. Droitcourt C, Adamski H, Polat A, Polard E, Kerjouan M,

Arnouat B, et al. Pirfenidone photosensitization in pa-

tients with idiopathic pulmonary fibrosis a case series. Br

J Dermatol 2018; 178:e222-3. [CrossRef]

5. Papakonstantinou E, Prasse A, Schacht V, Kapp A, Raap

U. Pirfenidone-induced severe phototoxic reaction in a

patient with idiopathic lung fibrosis. J Eur Acad Dermatol

Venereol 2016; 30:1354-6. [CrossRef]

6. Tsuruta A, Washio K, Fukunaga A, Nishigori C.

Pirfenidone-induced photoleukomelanoderma in a pa-

tient with idiopathic pulmonary fibrosis. J Dermatology

2016; 43:207-9. [CrossRef]

7. Basile FG, Creamer S. Docataxel/cylophomide induced

ultraviolet recall dermatitis. J Clin Oncology 2011;

29:e840-1. [CrossRef]

8. Krishnan RS, Lewis AT, Kass JS, Hsu S. Ultraviolet recall-

like phenomenon occurring after piperacillin, tobramycin,

and ciprofloxacin therapy. J Am Acad Dermatol 2001;

44:1045-7. [CrossRef]

9. Mang R, Stege H, Krutmann J. Mechanisms of phototoxic

and photoallergic reactions. In: Frosch PJ, Menné T, Le-

poittevin JP. (eds) Contact Dermatitis Springer, Berlin,

Heidelberg. 2006: 97-104. [CrossRef]

10. Seto Y, Inoue R, Kato M, Yamada S, Onoue S. Pho-

tosafety assessments on pirfenidone: photochemical,

photobiological, and pharmacokinetic characterization. J

Photochem Photobiol B 2013; 120:44-51. [CrossRef]

Respir Case Rep 2020;9(2): 83-86 DOI: 10.5505/respircase.2020.04796

OLGU SUNUMU CASE REPORT

83

Fatma Tokgoz Akyil1, Ahmet Topbas

1, Mustafa Akyıl

2

Congenital bronchial atresia (CBA) is a rare congeni-

tal airway malformation that is caused by an interrup-

tion to a proximal lobar, the segmental or subseg-

mental bronchus, hyperinflation and mucoid impac-

tion distal to the atresic bronchus. Patients may be

asymptomatic, or a cough, shortness of breath or

recurrent infection may be encountered. We present

here the case of a 21-year-old male who presented

with exertional dyspnea and cough on exertion, and

who was diagnosed with congenital bronchial atresia.

Key words: Congenital bronchial atresia, dyspnea,

mucocele.

Konjenital bronş atrezisi lober, segmenter veya sub-

segmenter bronşların atrezik sonlanması ve bu ne-

denle distalde oluşan havalanma artışı ve mukus

birikimi ile karakterizedir. Hastalar asemptomatik

olabileceği gibi; nefes darlığı, öksürük ve rekürren

enfeksiyonlar ile başvurabilir. Bu olgu sunumunda,

ağır eforla ortaya çıkan nefes darlığı ve öksürük ne-

deniyle başvuran ve konjenital bronş atrezisi tanısı

konulan 21 yaşında bir erkek hasta nadir görülmesi

nedeniyle sunulmuştur.

Anahtar Sözcükler: Dispne, konjenital bronş atrezisi,

mucocele.

1Department of Chest Diseases, Çanakkale Mehmet Akif Ersoy

State Hospital, Çanakkale, Turkey

2Department of Thoracic Surgery, Çanakkale Mehmet Akif Ersoy

State Hospital, Çanakkale, Turkey

1Çanakkale Mehmet Akif Ersoy Devlet Hastanesi, Göğüs

Hastalıkları Kliniği, Çanakkale

2Çanakkale Mehmet Akif Ersoy Devlet Hastanesi, Göğüs

Cerrahisi Kliniği, Çanakkale

Submitted (Başvuru tarihi): 31.10.2019 Accepted (Kabul tarihi): 24.01.2020

Correspondence (İletişim): Fatma Tokgoz Akyil, Department of Chest Diseases, Çanakkale Mehmet Akif Ersoy State Hospital,

Çanakkale, Turkey

e-mail: fatmatokgoz86@gmail.com

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Congenital bronchial atresia (CBA) is a rare congenital

airway malformation caused by the interruption of a prox-

imal lobar, segmental or subsegmental bronchus and

mucoid impaction, distal to the atresic bronchus. It is

more commonly reported in males, with an estimated

prevalence of 1.2 cases per 100,000 (1-3).

The most frequently involved segments are the left

apicoposterior segment and the left lower lobe, with esti-

mated rates of 64% and 14%, respectively. Radiologically,

bronchocele is a rounded branching opacity showing a

mucus-filled bronchus, and adjacent airway trapping or

emphysematous change may suggest is a suspicion of

bronchial atresia. In bronchoscopy, a blinding-ending

bronchus with classic radiographic features is diagnosti-

cally indicative of bronchial atresia (4,5).

Patients are mostly diagnosed incidentally in their second

or third decades. If the case is symptomatic, cough,

shortness of breath and recurrent infections may present

(6). We report here on a symptomatic young male diag-

nosed with bronchial atresia during his military service.

CASE

A 21-year old male presented with shortness of breath,

cough and sputum on exertion. The patient had been

fulfilling his conscripted military service for the past three

months, and was found to experience dyspnea during

heavy exercise. The patient had no additional diseases or

previously diagnosed lung disease. Over the previous five

years, he had been prescribed antibiotics for bronchitis

on three occasions. A physical examination was normal.

Spirometer forced expiratory volume in one second

(FEV1)/forced vital capacity (FVC) was 83% and FEV1

was 4.66 liters (81% of predicted). Bronchodilator revers-

ibility was negative. On a chest roentgenogram, a finger

in glove sign originating in the right hilum was noted

(Figure 1). A thorax high-resolution computed tomogra-

phy revealed mucoid impaction and peripheral hyperlu-

cency (Figure 2).

In an analysis of a complete blood count, white blood

cells were 13.8 (4.0-10.5) (µl/mlK/ml), and C-reactive

protein was 15 (0-5 mg/dl). Routine laboratory values

were within normal limits.

Upon suspicion of a bronchial abnormality, a fiberoptic

bronchoscopy was performed, and the right intermediate

bronchus was divided into sole lower lobe segments. The

patient was diagnosed with bronchial atresia (Figure 3),

and his military service was terminated with a report of

congenital bronchial atresia. The patient was educated

for possible complications and close follow-up was

scheduled.

DISCUSSION

This case report presents a unique cause of shortness of

breath, and is highly demonstrative of bronchial atresia.

The first case of CBA was defined by Ramsay et al. in

1953, and around 100 cases is have been reported in

literature to date (4,7). The leading locations of involved

bronchi are the apicoposterior segmental bronchus of the

left upper lobe, the right upper lobe, the middle lobe and

the right lower lobe, respectively [6]. Bronchial atresia

may present along with other congenital lung malfor-

mations, such as congenital cystic adenomatoid malfor-

mation, bronchopulmonary sequestration, congenital

lobar emphysema and lesions of mixed pathology (1,2,6).

In the present case, the middle lobe bronchus was affect-

ed and no coincident malformation was detected.

Figure 1: Chest X-ray with finger in glove sign, originating in the right

hilum

Figure 2: High-resolution computed tomography of the chest showing

mucoid impaction and peripheral hyperlucency

An Extremely Rare Cause of Dyspnea on Exertion: Bronchial Atresia | Tokgoz Akyil et al.

85 www.respircase.com

Figure 3: Endobronchial view from distal end of the right intermediate

bronchus

The exact mechanism of atresia is not yet known, but the

most accepted hypothesis is that the proliferating cells

lose their connection with the developing respiratory bud

during normal lung maturation. Another hypothesis is that

a repetitive vascular insult to lung parenchyma during

early fetal development leads to the obliteration of an

already completed bronchus (1,2).

Atretic bronchi do not communicate with the bronchial

tree. Instead, the bronchoalveolar channels of Lambert,

the pores of Kohn and the interbronchiolar channels

permit the entry of air, but prevent air escape, acting like

a one-way check valve. This results in hyperlucency and

hyperinflation in the distal of the atretic segment. Gener-

ally, mucus amasses, accumulating distal to the atretic

bronchus and creating a mucocele, and this accumula-

tion may lead to recurrent infections (7,8).

On a chest roentgenogram, hilar opacity in a tubular,

round, ovoid or branching structure may be observed.

Thorax CT reveals branching tubular or nodular opacities

radiating from the hilum with a “finger-in-glove” appear-

ance from the formation of mucoid impaction of the dis-

tal bronchus, known as a mucocele (2). Cavitary lesions

and air-fluid levels may be seen distal to the atresic bron-

chus (8–10). In present case, a finger-in-glove sign and

hyperinflation were noted.

Only one-third of diagnosed patients present with symp-

toms, with the most common symptoms being cough,

dyspnea and recurrent infection, although wheezing,

hemoptysis, chest pain and pneumothorax may also be

encountered. Hyperinflation and obstructive pulmonary

defects may cause dyspnea, although dyspnea is mostly

reported as exertional (4,10,11,12). The subject of the

present case study also suffered from dyspnea on exertion.

Prior to starting his military service, he had no symptoms.

Only during heavy exercise did he experience shortness of

breath.

Pathologically, a diagnosis of bronchial atresia is made

based on macroscopic findings, being the mucus plug-

ging of the prominent alveoli. There are usually no acute

or chronic inflammatory changes associated with bron-

chial atresia unless concomitant infection occurs

(1,10,12).

Differential diagnoses are bronchogenic cyst, lung apla-

sia, congenital lobar emphysema, congenital cystic ade-

nomatiod malformation, anomalous pulmonary venous

return, pulmonary sequestration, cystic fibrosis, allergic

bronchopulmonary aspergillosis (ABPA), and other benign

and neoplastic processes, (2) while radiological findings

may suggest CBA. Bronchoscopy is not compulsory for

diagnosis, but it may be necessary for the exclusion of

other bronchial abnormalities (4). In the present case,

radiological findings were suggestive of bronchial atresia,

and were verified from the bronchoscopic findings of the

atretic bronchus.

Follow-up is adequate for asymptomatic patients, while

recurrent infections and complications may require sur-

gery (2). The reported complications are recurrent infec-

tions, spontaneous pneumothorax and degradation of the

pulmonary parenchyma in the long term (1). The patient

reported no symptoms or infections prior to military ser-

vice, and military service was terminated after the diagno-

sis. At 6-months follow-up, the patient is asymptomatic,

and follow-up is continuing.

In conclusion, CBA is a rare airway malformation with

specific exceptional radiologic findings. Other than inci-

dental findings, patients with recurrent infections and

exertional dyspnea, the clinician should consider CBA as

a differential diagnosis.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - F.T.A., A.T., M.A.; Planning and Design -

F.T.A., A.T., M.A.; Supervision - F.T.A., A.T., M.A.; Fund-

ing -; Materials - F.T.A., A.T., M.A.; Data Collection

and/or Processing - F.T.A., A.T., M.A.; Analysis and/or

Interpretation - F.T.A.; Literature Review - F.T.A.; Writing -

F.T.A.; Critical Review - F.T.A., A.T., M.A.

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 86

YAZAR KATKILARI

Fikir - F.T.A., A.T., M.A.; Tasarım ve Dizayn - F.T.A., A.T.,

M.A.; Denetleme - F.T.A., A.T., M.A.; Kaynaklar -; Mal-

zemeler - F.T.A., A.T., M.A.; Veri Toplama ve/veya İşleme

- F.T.A., A.T., M.A.; Analiz ve/veya Yorum - F.T.A.; Lite-

ratür Taraması - F.T.A.; Yazıyı Yazan - F.T.A.; Eleştirel

İnceleme - F.T.A., A.T., M.A.

REFERENCES

1. Gipson MG, Cummings KW, Hurth KM. Bronchial atresia.

Radiographics 2009; 29:1531-5. [CrossRef]

2. Berrocal T, Madrid C, Novo S, Gutiérrez J, Arjonilla A,

Gómez-León N. Congenital anomalies of the tracheo-

bronchial tree, lung, and mediastinum: embryology, ra-

diology, and pathology. Radiographics 2004; 24:e17.

[CrossRef]

3. Schittny JC. Development of the lung. Cell Tissue Res

2017; 367:427-44. [CrossRef]

4. Mahajan AK, Rahimi R, Vanderlaan P, Folch E, Gan-

gadharan S, Majid A. Unique approach to diagnosing

and treating congenital bronchial atresia: a case series. J

Pulm Respir Med 2017; 7:2. [CrossRef]

5. Murat A, Ozdemir H, Yıldırım H, Kursad Poyraz A, Artas

H. Bronchial Atresia of Right Lower Lobe. Acta Radiol

2005; 46:480-3. [CrossRef]

6. Hutchison MJ, Winkler L. Bronchial Atresia. [Updated

2019 Jan 14]. In: StatPearls [Internet]. Treasure Island

(FL): StatPearls Publishing; 2019 Jan-.Available from:

https://www.ncbi.nlm.nih.gov/books/NBK537142/

7. Zylak CJ, Eyler WR, Spizarny DL, Stone CH. Develop-

mental lung anomalies in the adult: radiologic-

pathologic correlation. Radiographics 2002; 22:S25-43.

[CrossRef]

8. Psathakis K, Eleftheriou D, Boulas P, Mermigkis C,

Tsintiris K. Congenital bronchial atresia presenting as a

cavitary lesion on chest radiography: a case report. Cas-

es J 2009; 2:17. [CrossRef]

9. Karaman S, Deveci R, Bahçeci Erdem S, Karkiner A,

Alper H, Can D. Unusual radiological sign in bronchial

atresia. Turk Thorac J 2016; 17:79-81. [CrossRef]

10. Batchelor TJP, Rasburn NJ, Abdelnour-Berchtold E, Bru-

nelli A, Cerfolio RJ, Gonzalez M, et al. Guidelines for

enhanced recovery after lung surgery: recommendations

of the Enhanced Recovery After Surgery (ERAS®) Society

and the European Society of Thoracic Surgeons (ESTS).

Eur J Cardiothorac Surg 2019; 55:91-115. [CrossRef]

11. Traibi A, Seguin-Givelet A, Grigoroiu M, Brian E, Gossot

D. Congenital bronchial atresia in adults: thoracoscopic

resection. J Vis Surg 2017; 3:174. [CrossRef]

12. Wang Y, Dai W, Sun Y, Chu X, Yang B, Zhao M. Con-

genital bronchial atresia: diagnosis and treatment. Int J

Med Sci 2012; 9:207-12. [CrossRef]

Respir Case Rep 2020;9(2): 87-90 DOI: 10.5505/respircase.2020.62687

OLGU SUNUMU CASE REPORT

87

Ahmet Emre Hatır1, Büşra Özyalvaç

2, Sevgi Pekcan

2, Necdet Poyraz

3

Partial anomalous pulmonary venous return is a rare

congenital cardiac defect characterized by a flow of

blood from a few of the pulmonary veins returning to

the right atrium rather than the left atrium. Diagnosis

of this anomaly can be quite difficult. The condition is

usually diagnosed with echocardiography, and mag-

netic resonance imaging may also be useful. The

clinical manifestation and progression of the disease

varies depending on the amount of intra-cardiac

shunt. Signs or symptoms may include dyspnea,

coughing and fatigue. This defect usually causes no

negative symptoms and the child can develop nor-

mally. This research presents the case on an 8-year-

old female patient who complained of coughing and

whose abnormal findings on a chest X-ray led to a

further investigation. The patient was diagnosed with

partial anomalous pulmonary venous return. Diagno-

sis of this anomaly at an early stage can lead to the

patient gaining an awareness of the disease and

being able to deal with any complications that arise

in the adolescent period.

Key words: Partial anomalous pulmonary venous

connection, Atrial septal defects, Magnetic Resonance

Imaging.

Parsiyel anormal pulmoner venöz dönüş, pulmoner

venlerin bir kaçının sol atriyum yerine sağ atriyuma

geri dönmesi ile karakterize nadir görülen konjenital

bir kardiyak defekttir. Bu anomalinin tanısını koymak

oldukça zordur. Bu durum genellikle ekokardiyografi

ile teşhis edilir, ayrıca manyetik rezonans görüntüsü

de yararlı olabilir. Hastalığın klinik belirtisi ve seyri,

intrakardiyak şantın miktarına bağlı olarak değişir.

Belirti veya semptomlar; dispne, halsizlik ve öksürüğü

içerebilir. Bu anomali genellikle ciddi semptomlara

neden olmaz ve çocuk normal olarak büyür ve gelişir.

Bu çalışmada akciğer grafisinde anormal bulguları

olan ve öksürük şikayeti olan sekiz yaşında bir kız

hasta sunulmuş ve ileri araştırma sonucunda parsiyel

anormal pulmoner venöz dönüş tanısı konulmuştur.

Bu hastalığın tanısının erken konulması, hastanın bu

anomaliye olan farkındalığını artırarak ergenlik dö-

neminde ortaya çıkabilecek komplikasyonlar ile baş

etmesine olanak tanır.

Anahtar Sözcükler: Parsiyel anormal pulmoner venöz

dönüş anomalisi, Atriyal septal defektler, Manyetik

Rezonans Görüntüleme.

1Department of Family Medicine, Necmettin Erbakan University,

Konya, Turkey

2Department of Pediatrics, Necmettin Erbakan University, Konya,

Turkey

3Department of Radiology, Necmettin Erbakan University, Konya,

Turkey

1Necmettin Erbakan Üniversitesi Meram Tıp Fakültesi, Aile

Hekimliği Anabilim Dalı, Konya

2Necmettin Erbakan Üniversitesi Meram Tıp Fakültesi,

Çocuk Hastalıkları Anabilim Dalı, Konya

3Necmettin Erbakan Üniversitesi Meram Tıp Fakültesi,

Radyoloji Anabilim Dalı, Konya

Submitted (Başvuru tarihi): 27.12.2019 Accepted (Kabul tarihi): 26.02.2020

Correspondence (İletişim): Ahmet Emre Hatır, Department of Family Medicine, Necmettin Erbakan University, Konya, Turkey

e-mail: hatiremre@gmail.com

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Partial anomalous pulmonary venous return (PAPVR) is a

developmental disorder in which one or more pulmonary

vein cannot connect to the left atrium. PAPVR is usually an

acyanotic lesion (1), and while patients with this anomaly

usually have an asymptomatic course during childhood,

they may begin to show signs at an advanced age. This is

due to volume overload, and leads to complaints such as

fatigue, shortness of breath and coughing (2).

In this study, we present the case of an 8-year-old girl

who was admitted to our hospital with fever and vomiting,

and who had a previous history of hospitalization for

pneumonia. The patient underwent further investigations

after a suspicious lesion was identified on chest X-ray. As

a result, partial anomalous pulmonary venous return was

detected. We present this case report due to the sponta-

neous detection of PAPVR.

CASE

An 8-year-old female patient was brought to our hospital

who, it was learned, complained of coughing, especially

during the winter months, but that did not suffer severely

and did not have frequent illnesses. There was no pathol-

ogy related to birth, although there was a history of hos-

pitalization due to a urinary tract infection at 6 months

and pneumonia at 1 year of age. Her vaccines were

completed. She had no known disease and had no regu-

lar medication. She had 2 healthy siblings and there was

no kinship between her parents.

The height and weight percentiles were in the appropriate

range. An examination of the oropharynx was normal,

respiratory sounds were bilaterally equal and respiratory

sounds were normal. No murmur was heard in the cardi-

ovascular system.

There were no pathological features in laboratory tests. A

computed tomography (CT) was carried out as a suspi-

cious area was identified form the chest X-ray (Figure 1).

The pulmonary veins located in the upper lobe of the left

lung and the flow into the left brachiocephalic vein fol-

lowing the anterior aortic arch was reported (Figure 2).

Left lateral upper partial abnormal pulmonary venous

return was considered.

The patient consulted a pediatric cardiologist. An echo-

cardiography (Echo) was performed and a small atrial

septal defect (ASD) was detected, in addition to a partial

pulmonary venous return anomaly. A 4 mm left-to-right

shunt between the atria was observed. It was found that

the vertical vein collecting the flow originating from the

upper zone of the left lung was entering the innominate

vein. There was also mild insufficiency in the tricuspid

valve (4th degree). As a result of these findings and clini-

cal picture, the patient was not planned to be operated

on by the pediatric cardiologist, and medical follow-up

was recommended.

DISCUSSION

PAPVR was first described by Winslow in 1739 (3). When

an ASD is detected on an Echo, it is always necessary to

look for an associated PAPVR. It is difficult to differentiate

the symptoms, signs, electrocardiographic and radiologi-

cal findings of isolated ostium secundum ASD findings.

An Echo usually confirms the diagnosis. PAPVR is rare,

and has a good prognosis, similar to ostium secundum

ASD (1). PAPVR is examined in five different types, with

the type that connects to the superior vena cava (SVC)

being the most common. Abnormal pulmonary veins

connect to the lower side of the SVC or SVC-right atrial

junction in the most common type. Anomalies are fre-

quently seen with sinus venosus-atrial septal defects (4).

Our case had the most common type of PAPVR, and a

small ASD was detected. In a retrospective multi-slice

computer tomography (MSCT) study performed by Ho et

al. (5), PAPVR anomalies were encountered at a rate of

0.1% in 45,538 live cases.

Figure 1: The suspicious area indicated in blue is remarkable on the

Posterior-Anterior Chest X-ray

Figure 2: Axial MSCT images show that the left superior pulmonary vein,

indicated by the blue arrow in A, opens into the left brachiocephalic vein

at the point indicated by the blue arrow in B

Incidental Diagnosis of Partial Anomalous Pulmonary Venous Return: A Case Report | Hatır et al.

89 www.respircase.com

linical findings may vary in the left-to-right shunt ratio, in

the number and localization of abnormal veins, in the

presence of possible complications such as infection, in

the accompaniment of ASD, and in the size of such ASDs.

If the volume of the shunt is high, secondary pulmonary

hypertension may develop. Cases in which less than half

of the lung volume join the systemic circulation are

asymptomatic (6,7). Common signs and symptoms may

include dyspnea, coughing, fatigue, chest pain, palpita-

tions and tachycardia (5). Heart murmurs and arrhythmi-

as are also likely. In contrast to PAPVR, patients with scim-

itar syndrome are more likely to develop symptoms at an

early age and are more likely to have cyanosis (8).

The radiographical diagnosis of PAPVR is very difficult. If

PAPVR is accompanied by hypogenetic lung syndrome,

radiography plays an important role in the diagnosis (9).

Chest radiography is usually the first imaging method.

Radiographic findings depend on abnormal drainage, the

affected lobe and the left to right shunt. A chest X-ray may

be normal, or dilated SVC, cardiomegaly, right ventricu-

lar dilatation, right atrial dilatation, pulmonary edema or

right ventricular dilatation may be seen (10).

If a significant left-to-right shunt is identified in Echo,

there is a paradoxical interventricular septal movement,

and a right ventricular volume overload may develop.

Abnormal venous drainage to the right SVC may be more

difficult to detect unless a systematic approach is per-

formed. Surgical interventions should be considered in

cases with a volume-loaded right ventricle. Surgery is not

necessary when a single abnormally drained vein does

not load the right ventricle volume. Surgery is typically

performed at a similar time to ASD repair, when the pa-

tient is 3–5 years of age. The type of surgery depends on

the location of the drainage, but generally involves re-

connecting the abnormal vessels directly to the left. Pa-

tients with a repaired PAPVR have good prognosis (11).

In our case, minimal ASDs were seen in Echo. The patient

had no common symptom or infection, and so no cardio-

logic operation was planned, with follow-up recommend-

ed instead. In this study, we conclude that a chest X-ray

taken as the first approach, together with the patient's

clinic and history, provided us with important clues. Our

patient had a history of hospitalization due to pneumonia,

coughing especially in winter, and a suspicious area on a

chest radiography, leading to further investigations.

CONCLUSION

Patients can cope with the complications that may arise in

the adolescent period and at a more advanced age be-

cause of the diagnosis of partial anomalous pulmonary

venous return and patients also may be under control.

We can also learn that knowing the cause of the symp-

toms of the disease can prevent the patient from applying

to the hospital continuously and unnecessary hospitaliza-

tion can be prevented.

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - A.E.H., B.Ö., S.P., N.P.; Planning and Design

- A.E.H., B.Ö., S.P., N.P.; Supervision - A.E.H., B.Ö., S.P.,

N.P.; Funding - S.P., N.P.; Materials - S.P.; Data Collec-

tion and/or Processing - S.P., A.E.H.; Analysis and/or

Interpretation - S.P., N.P., A.E.H., B.Ö.; Literature Review

- A.E.H., S.P., B.Ö.; Writing - A.E.H., S.P., B.Ö.; Critical

Review - S.P.

YAZAR KATKILARI

Fikir - A.E.H., B.Ö., S.P., N.P.; Tasarım ve Dizayn -

A.E.H., B.Ö., S.P., N.P.; Denetleme - A.E.H., B.Ö., S.P.,

N.P.; Kaynaklar - S.P., N.P.; Malzemeler - S.P.; Veri Top-

lama ve/veya İşleme - S.P., A.E.H.; Analiz ve/veya Yorum

- S.P., N.P., A.E.H., B.Ö.; Literatür Taraması - A.E.H.,

S.P., B.Ö.; Yazıyı Yazan - A.E.H., S.P., B.Ö.; Eleştirel

İnceleme - S.P.

REFERENCES

1. Bernstein D. Partial anomalous pulmonary venous return.

In: Kliegman RM, St. Geme JW, Blum NJ et al, eds. Nel-

son textbook of pediatrics. 21th edition. Elsevier;

2019:2376.

2. Yüksekkaya R, Çelikyay F, Yılmaz A, Deniz Ç, Gökçe E.

Parsiyel anormal pulmoner venöz dönüş anomalisinin çok

kesitli bilgisayarlı tomografi anjiografi bulguları: İki ol-

gunun sunumu. Dicle Med J 2013; 40:128-30.

[CrossRef]

3. Winslow J. Mem Acad Roy Sci 1739:113.

4. Kirklin JW, Barrett-Boyes BG. Atrial septal defect and par-

tial anomalous pulmonary venous connection. Cardiac

surgery. 2nd edition, Vol 1. New York, NY: Churchill Liv-

ingstone 1993; 1:627-30.

5. Ho LM, Bhalla S, Bierhals A, Gutierrez F. MDCT of par-

tial anomalous pulmonary venous return (PAPVR) in

adults. J Thoracic Imaging 2009; 24:89-95. [CrossRef]

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 90

6. Miller S, Waltman A. The pulmonary circulation. In:

Tavares JM, Ferrucci JT eds. Radiology: Diagnosis-

Imaging Intervention. Philedelphia: JB Lippincott;

1995;1-19.

7. Miller S, Waltman A. The pulmonary circulation. In:

Tavares JM, Ferrucci JT eds. Radiology: Diagnosis-

Imaging Intervention. Philedelphia: JB Lippincott;

1995;1-19. [CrossRef]

8. Vyas HV, Greenberg SB, Krishnamurthy R. MR imaging

and CT evaluation of congenital pulmonary vein abnor-

malities in neonates and infants. Radiographics 2011;

32:87-98. [CrossRef]

9. Neill CA, Ferencz C, Sabiston DC, Sheldon H. The famil-

ial occurrence of hypoplastic right hung with systemic ar-

terial supply and venous drainage "Scimitar syndrome".

Bull Johns Hopkins Hosp 1960; 107:1-21.

10. Katre R, Burns SK, Murillo H, Lane MJ, Restrepo CS.

Anomalous pulmonary venous connections. Semin Ultra-

sound CT MR 2012; 33:485-99. [CrossRef]

11. Webb GD, Smallhorn JF, Therrien J, Redington AN.

Congenital heart diseases. In: Bonow RO, Mann DL,

Zipes DP, Libby P eds. Braunwald's heart disease: a text-

book of cardiovascular medicine. 9th edition. Philadelph-

ia: Elsevier Saunders; 2011:1464-5.

Respir Case Rep 2020;9(2): 91-95 DOI: 10.5505/respircase.2020.04557

OLGU SUNUMU CASE REPORT

91

Esra Aktiz Bıçak, Zeki Korhan, Mustafa Bıçak, Osman Uzundere, Cem Kıvılcım Kaçar, Abdulkadir Yektaş

Negatif basınçlı pulmoner ödem üst solunum yolunda

meydana gelen akut tıkanıklık sonrası veya kronik

tıkanıklığın kalkmasına sekonder gelişebilen bir du-

rumdur. Biz bu olgu sunumunda apendektomi sonrası

gelişen negatif basınçlı pulmoner ödem sonrası geli-

şen şiddetli hipoksi için APRV modunun başarılı yöne-

timini bildiriyoruz. Bilinen sistemik bir hastalık öyküsü

olmayan, 33 yaşında, 85 kilo ağırlığında, erkek has-

taya akut apandisit ön tanısı nedeniyle genel cerrahi

kliniğince apendektomi operasyonu planlandı. Apen-

dektomi ameliyatına alındı ve sonrasında ciddi larin-

gospazm ve şiddetli inspiratuvar efor gelişti. Negatif

basınçlı akciğer ödemi gelişen hasta APRV moduyla

tedavi edilerek operasyon sonrası 5. günde servise

verildi. İnvaziv mekanik ventilasyonda hipoksinin

düzeltilemediği olgularda APRV modu kullanımının

hipoksiyi dramatik şekilde düzelttiğini düşünmekteyiz.

Anahtar Sözcükler: Negatif basınçlı akciğer ödemi,

airway pressure release ventilation, aspirasyon pnö-

monisi, apandektomi.

Negative pressure pulmonary edema is a condition

that may develop after an acute obstruction in the

upper respiratory tract, or may occur secondary to the

elimination of a chronic obstruction. In this case

study, we report on the successful management of the

APRV mode for severe hypoxia developing after a

negative pressure pulmonary edema that developed

after an appendectomy. An appendectomy operation

was planned by the general surgery clinic for a 33-

year-old male patient weighing 85 kg with no known

systemic disease history, due to a preliminary

diagnosis of acute appendicitis. The patient

underwent an appendectomy, after which severe

laryngospasm and severe inspiratory effort

developed. The patient, who developed a negative

pressure pulmonary edema, was treated with the

APRV mode and taken to the service on day 5

following the operation. We believe that the use of

the APRV mode can dramatically improve hypoxia in

cases where hypoxia cannot be corrected in invasive

mechanical ventilation.

Key words: Negative pressure pulmonary edema,

airway pressure release ventilation, aspiration pneu-

monia, appendectomy.

TC. SBÜ. Gazi Yaşargil Eğitim ve Araştorma Hastanesi, Diyarbakır,

Anesteziyoloji ve Reanimasyon Kliniği, Diyarbakır

Department of Anesthesiology and Reanimation, TR. HSU.

Gazi Yaşargil Training and Research Hospital, Diyarbakır,

Turkey

Başvuru tarihi (Submitted): 23.01.2020 Kabul tarihi (Accepted): 16.03.2020

İletişim (Correspondence): Abdulkadir Yektaş, TC. SBÜ. Gazi Yaşargil Eğitim ve Araştorma Hastanesi, Diyarbakır,

Anesteziyoloji ve Reanimasyon Kliniği, Diyarbakır

e-mail: akyektas@hotmail.com

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Negatif basınçlı pulmoner ödem (NBAÖ) üst solunum

yolunda meydana gelen akut tıkanıklık sonrası veya kro-

nik tıkanıklığın kalkmasına sekonder gelişebilen bir du-

rumdur. Ender olmasına karşın ciddi bir komplikasyondur

(1). NBAÖ’nün patofizyolojisinde üst hava yolu tıkanıklığı

sonucu zorlu-kuvvetli inspiryum çabasının, intratorasik

basıç artışına neden olduğu ve bunun da pulmoner in-

terstisyuma non-kardiyojenik sıvı geçişine yol açtığı bildi-

rilmektedir (2-4).

Airway pressure releasing ventilation (APRV), akut solu-

num sıkıntısı sendromu olan hastalarda düşük tidal ha-

cimli, yardımcı kontrol ventilasyonuna göre çeşitli avantaj-

lara sahip yeni bir pozitif basınçlı ventilasyon modudur

(5,6).

Biz bu olgu sunumunda, apendektomi sonrası, negatif

basınçlı pulmoner ödeme ikincil gelişen şiddetli hipoksi

için APRV modunun başarılı yönetimini bildiriyoruz.

OLGU

Bilinen sistemik bir hastalık öyküsü olmayan, 33 yaşında,

85 kilo ağırlığında, erkek hastaya akut apandisit ön tanısı

nedeni ile genel cerrahi kliniğince apendektomi operas-

yonu planlandı. Preoperatif değerlendirmede ASA 1 sını-

fında, sigara anamnezi bulunmayan, kronik bir hastalığı

ve geçirilmiş cerrahi öyküsü bulunmayan hastaya genel

anestezi altında operasyon planlandı. Hastanın preop

laboratuvar değerlerinde, lökosit: 15,7 103/UL, hemog-

lobin: 14,1 g/dL, trombosit: 260 103/UL, PT: 13,0 sn,

INR: 1,21 üre: 31 mg/dl, kreatinin: 0,93 mg/dl, AST: 32

U/L ALT: 21 U/L CRP: 4,6 mg/dl olarak saptandı. Hasta-

nın 8 saat açlık süresi sağlandıktan sonra operasyon

masasına alındı. Hasta premedikasyon uygulanmadan,

elektrokardiyogram, non invazif kan basıncı, periferik

oksijen satürasyonu ölçümü yapılacak şekilde monitörize

edildi. Hastanın kalp atım sayısı:84 /dk, arteryel tansiyon:

130/85 mmHg, SpO2: %98 olarak kaydedildi. Hastaya

18 G intraket ile antekubital venden intravenöz yol sağla-

nılarak 15 ml/kg’den serum fizyolojik verildi. Daha sonra

2 mg midazolam, 2 mg/kg propofol, 1mcg/kg fentanil ve

0,6 mg/kg rokuronyum yapılarak 8,0 F çaplı trakeal tüple

orotrakeal entübasyon gerçekleştirildi. Anestezi idame-

si %50 N2O/O2 ve %1-2 sevofluran ile sağlandı. Kırk beş

dakika süren operasyon sürecinde herhangi bir hemodi-

namik ve solunumsal komplikasyon yaşanmadı. Operas-

yon süresince hastaya toplam 1000 ml kristalloid sıvı

verildi. Cerrahinin tamamlanmasını takiben, spontan

solunum aktivitesi görülen hasta 1 mg neostigmin ve

0,5mg atropinle reverse edildi. Ekstübasyon aşamasında

herhangi bir sorunla karşılaşılmayan hastada birkaç daki-

ka içerisinde ciddi laringospazm ve şiddetli inspiratuvar

efor gelişti. SpO2 %85 altına düşmesi üzerine hasta %100

FİO2 ve non invazif CPAP ile ventile edildi. Hastaya 1

mg/kg prednol ve 1 mg/kg lidokain yapıldı. CPAP ile

SpO2 %85’lerde seyreden hasta yoğun bakım ünitesine

alındı. Hasta 8F entübasyon tüpü ile tekrar 0,9 mg/kg

rokuronyum, 2 mg/kg propofol ve 100 mcg fentanil ile

entübe edilerek 0,1 mcg/kg/dk remifentanil infüzyonu ile

sedasyon sağlandı. Sağ internal juguler venden 7F santral

venöz kateter ve sağ radial arterden 20 G intraket ile

arterial kateter takıldı. Hastanın endotrakeal tüpünde hafif

kanlı köpüklü balgam mevcuttu. Hastanın fizik muayene-

sinde dinleme bulgusu olarak her iki hemitoraksta yaygın

krepitan raller bulunması üzerine arteryal kan gazı alındı.

Hastaya AP akciğer filmi çekildi (Şekil 1). FiO2 %100 iken

ve SIMV modunda PEEP 12 cmH2O, Solunum sayısı

16/dk ve PEEP üstü basınç 20 cmH2O iken arter kan

gazında pH: 7,15, PCO2: 58 mmHg PO2: 57mmHg

SpO2: %82 olması üzerine toraks BT çekildi (Şekil 2).

Bunun üzerine hasta PRVC moduna alındı solunum sayısı

16/dk, TV 390 mL, PEEP 12 cmH2O, PEEP üstü basınç

20 cmH2O ve Ptepe alarm üst limiti 35 cmH2O olarak

ayarlandı. İki saat bu şekilde takip edilen hastanın SpO2

si FiO2 %100 iken %85 in üzerine çıkmadı. Hastadan

kardiyoloji konsültasyonu istendi ve yapılan yatak başı

EKO da hastada kalp yetmezliği olmadığı teyit edildi.

Toraks BT’de bilateral konsolidasyon-buzlu cam görüntü-

leri mevcut olan hastada solunum sıkıntısının laringos-

pazm sonrası gelişmesi, fizik muayene, klinik süreç ve

laboratuvar incelemeleri ile tanı, negatif basınçlı akciğer

ödemi ve aspirasyon pnömonisi olarak düşünüldü. İnha-

ler bronkodilatatör, metilprednizolon 250 mg, aminofilin

240 mg ve 40 mg furosemid yapıldı. Ampirik antibiyote-

rapi (meropenem 3X1gr ve vankomisin 2X1gr) başlandı.

İki saat sonra çalışılan kan gazında oksijenasyonu düzel-

meyen hasta APRV modunda takip edilmeye başlandı.

Aynı zamanda sedasyon olarak midazolam 5

mg/kg/h’den ve ketamin 0,5 mg/kg/h’den başlandı.

APRV başlangıç ayarları Phigh: 30 cmH2O, Plow: 0

cmH2O Thigh: 5 sn Tlow: 1sn olarak ayarlandı. Hastanın

takip süreçlerinde APRV moduna alındıktan 2 saat sonra

çalışılan arter kan gazında FiO2: %70 iken pH: 7,33,

PCO2: 40 mmHg, PO2: 105 mmHg SpO2: %95’e yük-

seldi. Hastanın 2. gün mekanik ventilatör ayarları

FiO2: %50 iken Phigh: 25 cmH2O, Plow: 8 cmH2O,

Thigh: 1sn Tlow: 1sn’e düşüldü. Yatışının 2. gününde

çekilen AP akciğer grafisinde minimal bir düzelme vardı

(Şekil 3). Yatışının 3. gününde arter kan gazında pH:

7,43, PCO2: 37 mmHg, PO2: 158 mmHg olan hasta

Negatif Basınçlı Akciğer Ödemi ve İnvazif Mekanik Ventilasyonda APRV Modu: Olgu Sunumu | Yektaş et al.

93 www.respircase.com

CPAP’a alındı. FiO2: %50, PEEP: 8 cmH2O ve PEEP üstü

basıncı: 20 cmH2O olarak ayarlandı. CPAP değerleri

kademeli olarak düşürülen hasta 4. gününde extübe

edildi. Ekstübasyon sonrası AP-akciğer filmi tamama ya-

kın düzeldi (Şekil 4). Hasta 5. gününde genel cerrahi

servisine transfer edildi.

TARTIŞMA

NBAÖ’ye özellikle sağlıklı genç erişkinlerde, göğüs içi

yüksek negatif basınç oluşturabildikleri için daha sık rast-

lanır. Erkeklerde daha sıktır (%80) ve ASA fiziksel durum

P1 ve P2 de %73’dür. Ayrıca obez, üst hava yolu darlığı

olan, kısa boyun, üst hava yolları ile ilişkili operasyon,

uyku apne sendromu ve mediyastinal kitlesi bulunan has-

talarda diğer risk grubunu oluşturmaktadır (7).

Şekil 1: PA akciğer grafisinde bilateral parakardial heterojen parankimal

infiltrasyonlar.

Şekil 2: Toraks tomografisinde bilateral konsolidasyon-buzlu cam görün-

tüleri.

Şekil 3: İkinci gün PA akciğer grafisinde minimal regresyon.

Şekil 4: Ekstübasyon sonrası PA akciğer grafisinde tama yakın regresyon.

Yapılan bir çalışmada hastalar iki gruba ayrılmış ve has-

taları ekstübe etmek için sugammadex kullanan grupta

negatif basınçlı pulmoner ödem genel anestezi pratiğin-

de % 0,009, sugammadex kullanmayan grupta % 0 ora-

nında görülmüştür. Hastaların gruplara ayrılmadığı başka

bir çalışmada ise NBAÖ sıklığı % 0,1-% 11 aralığında

bulunmuş ve laringospazm sonrası ise NBAÖ sıklığının %

3’e ulaştığı bildirilmiştir (7,8).

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 94 94

NBAÖ, tekrarlanan obstrüksiyonlarla zorlu inspiriyum

yapılması sonucu hızlı negatif intraplevral basınç artışıyla

ilişkilidir. NBAÖ ile ilişkili akut hava yolu obstrüksiyonu-

nun nedeni laringospazmdır ve orofaringeal, baş ve bo-

yun cerrahisi bu riski arttırır. İntratorasik negatif basıncın

artışı venöz dönüşü arttırır ve bu da pulmoner venöz ba-

sıncı yükseltir. Artan vasküler permeabilite de sıvının pul-

moner kapillerlerden alveoler boşluğa geçişine katkıda

bulunur. Solunum yetmezliğine bağlı hipoksi ve asidozis

alveoler memran kapiller yaralanmayla ilişkili olarak

pulmoner vasküler rezistansı arttırır, oluşan dispne, asido-

zis ve hiperadrenerjik cevabı arttırarak diffüz alveoler

hemoraji ve kardiyak sorunlara da neden olabilir (3,4).

İnspiratuvar plevral basınç normalde (-2) – (-5) cmH2O

aralığında olup, tıkanıklığa karşı zorlu inspiryum eforu

sırasında intraplevral negatif basınç -100 cmH2O civarı-

na kadar çıkabilmektedir (3,4). NBAÖ’de ödemin daha

çok postoperatif dönemde gelişen laringospazm veya

preoperatif üst hava yolu patolojilerine bağlı gelişen akut

üst hava yolu obstrüksiyonundan sonra hızlı bir şekilde

ortaya çıktığı görülmüştür (9).

NBAÖ’nün klinik seyri tipiktir. Ayırıcı tanıda aspirasyon,

kardiyojenik pulmoner ödem ve sıvı yüklenmesi düşünül-

melidir (10). Bizim olgumuzda intraoperatif olarak hasta-

ya verilen sıvının normal değerlerde olması, ek bir kardi-

yak sorunun olmaması, dinlemekle yeni oluşan rallerin

bulunması ve EKO’sunun normal olması bizi nonkardiyo-

jenik akciğer ödemine yöneltti. Ayrıca ekstübasyon sonrası

laringospazm yaşamamız ve takiben tablonun oluşması,

akciğer grafisi ve akciğer tomografi bulgularının tanımızı

desteklemesi NBAÖ tanısını güçlendirdi.

Olgumuzda, pulmoner ödemin medikal tedaviye cevap

vermemesi, noninvazif solunum desteğinin yetersiz kalma-

sı reentübasyon ihtiyacının olması, konvansiyonel invazif

modlara yanıt vermemesi ve invazif mekanik ventilasyon

modu olarak kullanılan APRV modunun hipoksiyi drama-

tik olarak düzeltmesi dikkat çekicidir.

NBAÖ’de tanı konulması, yeterli oksijenasyonun sağlan-

ması ve pozitif hava yolu basıncı uygulanması tedavide

esastır. Ciddi NBAÖ tedavisinde agresif hemodinamik

monitörizasyon, reentübasyon ve invazif mekanik ventilas-

yon düşünülmelidir (11). Bu olguda ciddi bir NBAÖ tab-

losu ile karşılaşıldı ve tedavi protokolü ve yapılması gere-

ken agresif monitorizasyon yapıldı. Burada özellikle dikkat

çekmek istediğimiz nokta, bizim olgumuzda da olduğu

gibi non-invazif solunum desteğinin yetersiz kaldığı hasta-

larda tekrar entübasyon kararı vermekte gecikilmemelidir.

Yeterli sedasyon altında invazif solunum desteği sağlanan

hastalar, kısa sürede tedaviye cevap vermektedirler.

APRV modu, gaz dağılım volümü ile akciğer volümünün

uyumunu optimalleştirerek alveoler aşırı gerilimini engel-

ler, ek olarak spontan ventilasyon eforlarında intermittant

release periodda Plow esnasında alveoler ventilasyonu

arttırır ve CO2’i uzaklaştırır. APRV konvansiyonel modlara

göre verilen tidal volümle Pplato ve Ppeak basıncını daha

fazla düşürür. APRV daha az sedasyon ve paralizise izin

verir. Bu mod zorunlu spontan ventilasyon modu oldu-

ğundan farmakolojik paraliziden kaçınılmış olur. Diafram

hareketlerine izin verdiği için de ventilasyon/perfüzyon

oranı iyileşmektedir (5).

Postoperatif NBAÖ nedenli APRV uygulaması hakkında

çok fazla sayıda literatür yoktur. APRV, transalveolar ba-

sınç gradyendini sınırlandırırken alveoler rekruitment en

üst düzeye çıkararak pulmoner disfonksiyon yönetimine

yardımcı olur ve böylece barotravma riskini de azaltır

(5,9,10,11).

Sonuç olarak; NBAÖ nadir görülen fakat yüksek morbidi-

te ve mortalite ile sonuçlanabilen bir durumdur. Riskli

olguların belirlenmesi, özellikle ekstübasyon işlemi esna-

sında laringospazm gelişen genç-erişkin ve erkek hasta-

larda dikkatli olunması ve akciğer ödemi bulguları oluşur-

sa ayırıcı tanıda NBAÖ tanısının mutlaka göz önünde

bulundurulması gerekmektedir. NBAÖ tedavisinde üst

hava yolu obstrüksiyonunun erken tanınması, hipoksinin

önlenmesi, gerekirse reentübasyon ve invazif mekanik

ventilasyonun sağlanması unutulmamalıdır. İnvazif meka-

nik ventilasyonda hipoksinin düzeltilemediği olgularda

APRV modu kullanımının hipoksiyi dramatik şekilde düzel-

teceğini düşünmekteyiz.

ÇIKAR ÇATIŞMASI

Bu makalede herhangi bir çıkar çatışması bildirilmemiştir.

YAZAR KATKILARI

Fikir - E.A.B., Z.K., M.B., O.U., C.K.K., A.Y.; Tasarım ve

Dizayn - E.A.B., Z.K., M.B., O.U., C.K.K., A.Y.; Denetle-

me - E.A.B., Z.K., M.B., O.U., C.K.K., A.Y.; Kaynaklar -;

Malzemeler -; Veri Toplama ve/veya İşleme - A.Y.; Analiz

ve/veya Yorum - A.Y.; Literatür Taraması - A.Y.; Yazıyı

Yazan - A.Y.; Eleştirel İnceleme - A.Y.

KAYNAKLAR

1. Oswalt CE, Gates GA, Holmstrom MG. Pulmonary ede-

ma as a complication of acute airway obstruction. JAMA

1977; 238:1833-5. [CrossRef]

2. Schwartz DR, Maroo A, Malhotra A, Kesselman H. Nega-

tive pressure pulmonary hemorrhage. Chest 1999;

115:1194-7. [CrossRef]

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95 www.respircase.com

3. Liu R, Wang J, Zhao G, Su Z. Negative pressure pulmo-

nary edema after general anesthesia: A case report and

literature review. Medicine (Baltimore) 2019; 98:e15389.

[CrossRef]

4. Xiong J, Sun Y. Negative pressure pulmonary edema: a

case report. BMC Anesthesiol. 2019; 19:63. [CrossRef]

5. Fredericks AS, Bunker MP, Gliga LA, Ebeling CG,

Ringqvist JR, Heravi H, et al. Airway pressure release ven-

tilation: a review of the evidence, theoretical benefits,

and alternative titration strategies. Clin Med Insights Circ

Respir Pulm Med 2020; 14:1179548420903297.

[CrossRef]

6. Morimoto Y, Sugimoto T, Arase H, Haba F. Successful

management using airway pressure release ventilation for

severe postoperative pulmonary edema. Int J Surg Case

Rep 2016; 27:93-5. [CrossRef]

7. Silva LAR, Guedes AA, Salgado Filho MF, Chaves LFM,

Araújo FP. Negative pressure pulmonary edema: report

of case series and review of the literature. Rev Bras

Anestesiol 2019; 69:222-6. [CrossRef]

8. Kao CL, Kuo CY, Su YK, Hung KC. Incidence of nega-

tive-pressure pulmonary edema following sugammadex

administration during anesthesia emergence: A pilot au-

dit of 27,498 general anesthesia patients and literature

review. J Clin Anesth 2020; 62:109728. [CrossRef]

9. Choi E, Yi J, Jeon Y. Negative pressure pulmonary ede-

ma after nasal fracture reduction in an obese female pa-

tient: a case report. Int Med Case Rep J 2015; 8:169-71.

[CrossRef]

10. Harmon E, Estrada S, Koene RJ, Mazimba S, Kwon Y.

Concurrent Negative-Pressure Pulmonary Edema (NPPE)

and Takotsubo Syndrome (TTS) after Upper Airway Ob-

struction. Case Rep Cardiol 2019; 2019:5746068.

[CrossRef]

11. Tsai PH, Wang JH, Huang SC, Lin YK, Lam CF. Charac-

terizing post-extubation negative pressure pulmonary

edema in the operating room-a retrospective matched

case-control study. Perioper Med (Lond) 2018; 7:28.

[CrossRef]

Respir Case Rep 2020;9(2): 96-98 DOI: 10.5505/respircase.2020.78300

LETTER TO EDITOR EDİTÖRE MEKTUP

96

To the Editor,

A 22-year old woman presented to the chest dis-

eases clinic with complaints of non-productive

cough and left side chest pain, ongoing for two

days. She had been admitted to the hospital with a

sudden abdominal pain three days earlier. She was

diagnosed with an abdominopelvic abscess with a

ruptured dermoid cyst, and a salpingo-

oopherectomy was performed. The left side chest

pain initiated three days later.

Upon physical examination, vital signs were normal

and room air oxygen saturation was 98%. Respira-

tory sounds in the bilateral lower chest were de-

creased. An instant chest radiograph revealed

blunt costophrenic angles suggestive of a bilateral

pleural effusion (Figure 1).

Upon ED admission, biochemistry values were

within normal limits, complete blood count param-

eters revealed a mild leukocytosis of 12.6 K/L,

hemoglobin was decreased to 8.9 g/dl, and C-

reactive protein (CRP) was 154 mg/L. On the day

of the consultation, a complete blood count re-

vealed leukocytosis of 9.6 K/L, hemoglobin of

10.1 g/dl and CRP of 58 mg/L. She had neither

fever nor sputum. D-dimer was 812 ng/mL.

Upon ED admission, no pleural effusion was seen

at the proximal sections in an abdominal computer

tomography (CT), although ascites and a minimal

pericardial effusion were detected (Figure 2). The

pleural fluid was exudative according to Light’s

criteria (1); the pleural fluid to serum ratio for pro-

tein was 0.62 (3.6 vs. 5.8 g/dl, respectively); and

the pleural fluid to serum ratio for lactate dehydro-

genase was 0.89 (197 vs. 219 U/L, respectively). A

cytological examination identified the usual pleural

fluid cells, and no proliferation of microbiologic

agents.

Figure 1: Chest radiograph at the time of admission

Figure 2: Preoperative abdominal computer tomography

Figure 3: Two months after admission

RES

PIR

ATO

RY

CA

SE R

EPO

RTS

Respiratory Case Reports

Cilt - Vol. 9 Sayı - No. 2 97

A cardiac examination revealed also a 2-centimeter

pericardial effusion. No ascites were found in a

novel abdominal ultrasonography.

The rapid occurrence of a post-operative pleural

effusion led us to consider a reactional pleuro-

pericardial effusion. A chest radiograph two days

later showed a decreased left side pleural effusion.

The patient was informed that the fluid would regress

in a few weeks.

The patient was discharged the following day and

prescribed antibiotics and anti-inflammatory drugs.

One week later, she was re-admitted to the hospital

with continuous side pain. A cardiac evaluation

detected a regressed pericardial effusion with no

tamponade finding. This time, to exclude a pulmo-

nary embolism, she underwent a thorax CT angi-

ography, revealing no thrombus. Excluding the other

possible diagnoses, the patient was pre-diagnosed

with non-classic Meigs-like syndrome. All symptoms

were resolved two months later, and a chest radio-

graph showed complete regression (Figure 3).

Meigs’ syndrome is a rare finding in coexistence with

a benign ovarian tumor of fibroma, or a fibroma-

like tumor, ascites, pleural effusion and a curative

resection of the tumor (2). A relationship with other

benign ovarian tumors and other symptoms is de-

fined as non-classic Meigs’ syndrome (3).

Only a few studies examining the coexistence of

pericardial effusion alongside Meigs’ syndrome have

been published to date, in which the condition is

referred to as “Meigs-like syndrome”. A postopera-

tive increase in pleural effusion has been reported in

only a single patient with an 8-year history of fibro-

ma (4). In line with this case report, our case had

pericardial effusion and ascites, and developed

pleural effusion along with pericardial effusion after

surgery.

A pleural effusion may occur just immediately after

the operation, in which regressed ascites and peri-

cardial effusion may have overflowed into the pleu-

ral cavity. We believe that our patient differs from

those analyzed in literature in her acute presentation

of a ruptured dermoid cyst. An accurate diagnosis of

non-classic Meigs-like syndrome was accomplished

2 months after the operation, when the effusion had

completely regressed.

This case report is important in revealing that the

development of a pleural effusion following opera-

tions on benign ruptured ovarian tumors is an en-

countered phenomenon. While pericardial effusion

regresses rather early, the complete regression of a

pleural effusion may take longer. Taking this syn-

drome into account, informing the patient and a

close follow-up may allow needless investigations

and examinations to be avoided.

Fatma Tokgoz Akyil1

, Mustafa Akyıl2

1Department of Chest Diseases, Çanakkale Mehmet Akif

Ersoy State Hospital, Çanakkale, Turkey

2Department of Thoracic Surgery, Çanakkale Mehmet Akif

Ersoy State Hospital, Çanakkale, Turkey

Correspondence (İletişim): Fatma Tokgoz Akyil, Department

of Chest Diseases, Çanakkale Mehmet Akif Ersoy State

Hospital, Çanakkale, Turkey

e-mail: fatmatokgoz86@gmail.com

CONFLICTS OF INTEREST

None declared.

AUTHOR CONTRIBUTIONS

Concept - F.T.A., M.A.; Planning and Design -

F.T.A., M.A.; Supervision - F.T.A., M.A.; Funding -;

Materials - F.T.A, M.A.; Data Collection and/or

Processing - F.T.A., M.A.; Analysis and/or Interpre-

tation - F.T.A.; Literature Review - F.T.A.; Writing -

F.T.A.; Critical Review - F.T.A., M.A.

YAZAR KATKILARI

Fikir - F.T.A., M.A.; Tasarım ve Dizayn - F.T.A., M.A.;

Denetleme - F.T.A., M.A.; Kaynaklar -; Malzemeler -

F.T.A., M.A.; Veri Toplama ve/veya İşleme - F.T.A.,

M.A.; Analiz ve/veya Yorum - F.T.A.; Literatür Tara-

ması - F.T.A.; Yazıyı Yazan - F.T.A.; Eleştirel İncele-

me - F.T.A., M.A.

REFERENCES

1. Light RW, Macgregor MI, Luchsinger PC, Ball WC Jr.

Pleural effusions: the diagnostic separation of transu-

dates and exudates. Ann Intern Med 1972; 77:507-

13. [CrossRef]

2. Meigs JV. Fibroma of the ovary with ascites and

hydrothorax‐ Meigs syndrome. Am J Obstet Gyne-

col 1954; 67:962-85. [CrossRef]

Respiratory Case Reports

98 www.respircase.com

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