Eosinophilic cholecystitis: An infrequent cause of acute...

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CASE REPORTS Eosinophilic cholecystitis: An infrequent cause of acute cholecystitis María del-Moral-Martínez 1 , Andrés Barrientos-Delgado 1 , Vicente Crespo-Lora 2 , María Eloísa Cervilla-Sáez-de-Tejada 1 and Javier Salmerón-Escobar 1 1 Digestive Diseases Clinical Management Unit. 2 Pathology Clinical Management Unit. Hospital Universitario San Cecilio. Granada, Spain 1130-0108/2015/107/1/45-47 REVISTA ESPAÑOLA DE ENFERMEDADES DIGESTIVAS COPYRIGHT © 2015 ARÁN EDICIONES, S. L. REV ESP ENFERM DIG (Madrid Vol. 107, N.º 1, pp. 45-47, 2015 CASE REPORTS ABSTRACT Eosinophilic cholecystitis (EC) is a rare disease that is characterised by eosinophilic infiltration of the gallbladder. Its pathogenesis is unknown, although many hypotheses have been made. Clinical and laboratory manifestations do not differ from those of other causes of cholecystitis. Diagnosis is histological and usually performed after analysis of the surgical specimen. We report the case of a woman aged 24 years, with symptoms of fever, vomiting and pain in the right upper quadrant. When imaging tests revealed acalculous cholecystitis, an urgent cholecystectomy was performed. Histological examination of the surgical specimen revealed eosinophilic cholecystitis. No cause of the symptoms was found. Key words: Eosinophilic cholecystitis. Acalculous cholecystitis. INTRODUCTION Eosinophilic cholecystitis is an uncommon condition of the gallbladder. It is characterised by an inflammatory infiltrate constituted mainly of eosinophils. Its aetiology is often unknown, although cases have been associated with hyper-eosinophilic syndrome, parasitosis, infec- tions, drugs and medicinal herbs. Clinically, it is indistin- guishable from common cholecystitis, although periph- eral eosinophilia is sometimes observed, as is the case in hyper-eosinophilic syndrome and parasitic disease. When the effect is limited to the bladder, the treatment of choice is cholecystectomy, and the prognosis is usually favourable. CASE REPORT A 24-year-old woman presented to the emergency de- partment complaining of abdominal pain, located in the epigastrium and radiating to the right upper quadrant, to- gether with nausea, vomiting and fever of 39 °C for the past two days. The presence of choluria was also reported. The patient had no personal or family history of interest. She smoked about five cigarettes per day and was a habit- ual consumer of oral contraceptives. Physical examination revealed good general condition, with cutaneous-mucous jaundice and tenderness in the right upper quadrant, and a positive Murphy sign. Other results of the examination were normal. Laboratory analysis revealed the follow- ing alterations: Total bilirubin 3.76 mg/dL (range 0-1.2 mg/dL); direct bilirubin 3.5 mg/dL (range 0-0.5 mg/dL); indirect bilirubin 0.26 mg/dL (range 0-0.75 mg/dL); ala- nine aminotransferase 174 U/l (range 0-35 U/l); amylase 49 U/L (range 28-100 U/L); C-reactive protein 149.12 mg/L (range 0-5 mg/L); leukocytes 15,500 µl (range 4.8- 10.8 µl); neutrophils 86.3 % (range 40-74 %); lympho- cytes 4.2 % (range 19-48 %); monocytes 6.9 % (range 1-9 %); eosinophils 2.2 % (range 0-7 %); prothrombin activity 60.8 % (range 70-120 %), INR 1.34 (range 0.85- 1.2); APTT 28.2 sec (range 23.5-33.2 sec). Abdominal ultrasound findings: Thin-walled acalculous gallbladder; non-dilated bile duct; no evidence of pancreatic abnor- malities. In view of the clinical and laboratory findings, the patient was admitted to monitor the evolution of the condition and for further study. Received: 05-03-2014 Accepted: 23-06-2014 Correspondence: María del-Moral-Martínez. Digestive Diseases Clinical Management Unit. Hospital Universitario San Cecilio. Avda. Doctor Oloriz, 16. 18012 Granada, Spain e-mail: [email protected] Del-Moral-Martínez M, Barrientos-Delgado A, Crespo-Lora V, Cervilla-Sáez-de-Tejada ME, Salmerón-Escobar J. Eosinophilic cholecystitis: An infrequent cause of acute cholecystitis. Rev Esp Enferm Dig 2015;107:45-47.

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CASE REPORTS

Eosinophilic cholecystitis: An infrequent cause of acute cholecystitis

María del-Moral-Martínez1, Andrés Barrientos-Delgado1, Vicente Crespo-Lora2, María Eloísa Cervilla-Sáez-de-Tejada1 and Javier Salmerón-Escobar1

1Digestive Diseases Clinical Management Unit. 2Pathology Clinical Management Unit. Hospital Universitario San Cecilio. Granada, Spain

1130-0108/2015/107/1/45-47Revista española de enfeRmedades digestivasCopyRight © 2015 aRán ediCiones, s. l.

Rev esp enfeRm dig (MadridVol. 107, N.º 1, pp. 45-47, 2015

CASE REPORTS

ABSTRACT

Eosinophilic cholecystitis (EC) is a rare disease that is characterised by eosinophilic infiltration of the gallbladder. Its pathogenesis is unknown, although many hypotheses have been made. Clinical and laboratory manifestations do not differ from those of other causes of cholecystitis. Diagnosis is histological and usually performed after analysis of the surgical specimen. We report the case of a woman aged 24 years, with symptoms of fever, vomiting and pain in the right upper quadrant. When imaging tests revealed acalculous cholecystitis, an urgent cholecystectomy was performed. Histological examination of the surgical specimen revealed eosinophilic cholecystitis. No cause of the symptoms was found.

Key words: Eosinophilic cholecystitis. Acalculous cholecystitis.

INTRODUCTION

Eosinophilic cholecystitis is an uncommon condition of the gallbladder. It is characterised by an inflammatory infiltrate constituted mainly of eosinophils. Its aetiology is often unknown, although cases have been associated with hyper-eosinophilic syndrome, parasitosis, infec-tions, drugs and medicinal herbs. Clinically, it is indistin-guishable from common cholecystitis, although periph-eral eosinophilia is sometimes observed, as is the case in hyper-eosinophilic syndrome and parasitic disease. When the effect is limited to the bladder, the treatment of choice is cholecystectomy, and the prognosis is usually favourable.

CASE REPORT

A 24-year-old woman presented to the emergency de-partment complaining of abdominal pain, located in the epigastrium and radiating to the right upper quadrant, to-gether with nausea, vomiting and fever of 39 °C for the past two days. The presence of choluria was also reported. The patient had no personal or family history of interest. She smoked about five cigarettes per day and was a habit-ual consumer of oral contraceptives. Physical examination revealed good general condition, with cutaneous-mucous jaundice and tenderness in the right upper quadrant, and a positive Murphy sign. Other results of the examination were normal. Laboratory analysis revealed the follow-ing alterations: Total bilirubin 3.76 mg/dL (range 0-1.2 mg/dL); direct bilirubin 3.5 mg/dL (range 0-0.5 mg/dL); indirect bilirubin 0.26 mg/dL (range 0-0.75 mg/dL); ala-nine aminotransferase 174 U/l (range 0-35 U/l); amylase 49 U/L (range 28-100 U/L); C-reactive protein 149.12 mg/L (range 0-5 mg/L); leukocytes 15,500 µl (range 4.8-10.8 µl); neutrophils 86.3 % (range 40-74 %); lympho-cytes 4.2 % (range 19-48 %); monocytes 6.9 % (range 1-9 %); eosinophils 2.2 % (range 0-7 %); prothrombin activity 60.8 % (range 70-120 %), INR 1.34 (range 0.85-1.2); APTT 28.2 sec (range 23.5-33.2 sec). Abdominal ultrasound findings: Thin-walled acalculous gallbladder; non-dilated bile duct; no evidence of pancreatic abnor-malities. In view of the clinical and laboratory findings, the patient was admitted to monitor the evolution of the condition and for further study.

Received: 05-03-2014Accepted: 23-06-2014

Correspondence: María del-Moral-Martínez. Digestive Diseases Clinical Management Unit. Hospital Universitario San Cecilio. Avda. Doctor Oloriz, 16. 18012 Granada, Spaine-mail: [email protected]

Del-Moral-Martínez M, Barrientos-Delgado A, Crespo-Lora V, Cervilla-Sáez-de-Tejada ME, Salmerón-Escobar J. Eosinophilic cholecystitis: An infrequent cause of acute cholecystitis. Rev Esp Enferm Dig 2015;107:45-47.

46 M. DEL-MORAL-MARTÍNEZ ET AL. Rev esp enfeRm Dig (maDRiD)

Rev esp enfeRm Dig 2015; 107 (1): 45-47

During admission, abdominal and cholangio MRI were performed to assess the bile duct, obtaining the following results: no dilatation; no evidence of bile duct filling de-fects; space-occupying lesion of 9 mm in segment 8 with uptake and density suggesting hemangioma; no evidence of alterations in pancreas or liver. Further analyses were performed, which revealed increased total bilirubin, de-creased direct bilirubin, increased leukocytosis, increased C-reactive protein, and normal levels of amylase, trans-aminases and cholestatic enzymes. The patient had mal-aise, with increased pain despite analgesia, and painful abdominal tenderness, with a tightening in the epigastric right upper quadrant. In view of this situation, an urgent abdominal CT scan with IV contrast was performed (Fig. 1), which showed perivesicular oedema without cal-culi or biliary dilatation, with a small amount of free fluid in the Douglas pouch. There were no images suggestive of perforation or pancreatitis.

The patient’s clinical condition was worsening and presence of cholecystitis was suspected, and so an urgent cholecystectomy was performed, which revealed a thick-ened gallbladder wall with oedema on the rear surface. The pathology examination revealed the presence of a transmural infiltration, and of a more intense infiltration in the muscular layer, by eosinophilic polynuclear leuko-cytes (Fig. 2).

After surgery, the patient was asymptomatic and was discharged a few days later.

DISCUSSION

Eosinophilic cholecystitis (EC) is a rare and poorly understood disease of the gallbladder, which was first

described in 1949. It can be considered an inflammatory condition of the gallbladder, in which the inflammatory infiltrate consists primarily of eosinophils (1).

The aetiology of EC is unknown. In patients with eo-sinophilic infiltrate affecting other organs and tissues, it has been suggested that these lesions could be due to a local allergic reaction to substances released at sites of inflammation within the target organ or tissue. It has also been hypothesised that EC may be caused by hypersensi-tivity to bile acids (2,3).

Cases have also been reported secondary to infections, parasitosis, allergies, hyper-eosinophilic syndrome, eo-sinophilia-myalgia syndrome, eosinophilic gastroenteri-tis, drugs and herbal medicines (4,5). In the absence of evident causes, we consider the present case to be an id-iopathic EC (6).

EC is three times more common in patients with acal-culous cholecystitis than in patients with cholelithiasis (6).

EC does not present any clinical or laboratory manifes-tation to distinguish it from common cholecystitis, and so it is difficult to detect prior to cholecystectomy and histo-logical examination of the surgical specimen. Peripheral eosinophilia may or may not be present; when it is, it has been associated with hyper-eosinophilic syndrome, eosin-ophilic gastroenteritis and parasitosis. In addition, symp-toms secondary to the eosinophilic infiltration of other organs have been described (8).

Diagnosis is histological; EC is said to be present when the cellular infiltrate in the gallbladder wall is composed of more than 90 % eosinophils, and the cholecystitis is chronic with an eosinophilic (lympho-eosinophilic) com-ponent if the infiltrating inflammatory leukocyte popula-tion contains 50-75 % eosinophils (9). In imaging tests,

Fig. 1. Abdominal CT with IV contrast, showing perivesicular oedema with no image of calculi or biliary dilatation.

Fig. 2. Optical microscopy image (haematoxylin). Inflammatory transmu-ral infiltrate, predominantly eosinophilic, in the gallbladder wall.

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ultrasound results may be normal or show signs sugges-tive of cholecystitis (gallbladder distension, wall thicken-ing, perivesicular liquid or sonographic Murphy sign). A CT scan may reveal similar features, with perivesicular oedema or decreased attenuation in the adjacent liver, in-dicative of perihepatitis (10).

EC prognosis is favourable. When the disease is con-fined to the bladder, the treatment of choice is cholecys-tectomy, preferably performed laparoscopically. Treat-ment with corticosteroids can be effective when the bile ducts are affected, or when the condition is associated with eosinophilic gastroenteritis.

It is generally accepted that EC should not be con-sidered a separate entity, because the clinical and lab-oratory manifestations are indistinguishable from those of common cholecystitis, and therefore it is considered more a histological finding than a pathology in itself. The importance of EC lies in the fact that it can be as-sociated with other diseases, and therefore, when it is observed, possible associated syndromes should be in-vestigated.

REFERENCES

1. Albot G, Poilleux H, Oliver C. Les cholécystites a éosinophils. Presse Med 1949;57:558-9.

2. Pardo-Mindan FJ, Joly MA, Santamaria M, Munoz Navas M. Eosino-phil inflammatory reaction in isolated organs. Allergol Immunopathol (Madr.) 1980;8:23-30.

3. Alfaro J, Fernández L, Hörndler C, Ruiz JM, Sanz JM, López M, et al. Eosinophilic cholecystitis associated with rupture of hepatic hydatid cyst of the bile ducts. Rev Esp Enferm Dig 1995;87:889-90.

4. Adusumilli PS, Lee B, Parekh K, Farrelly PA. Acalculous eosinophilic cholecystitis from herbal medicine: a review of adverse effects of herb-al medicine in surgical patients. Surgery 2002;131:352-6.

5. Hepburn A, Coady A, Livingstone J, Pandit N. Eosinophilic chol-ecystitis as a possible late manifestation of the eosinophilia-myalgia syndrome. Clin Rheumatol 2000;19:470-2.

6. Sánchez-Pobre P, López-Ríos Moreno F, Colina F, Yela C, Manzano M, et al. Eosinophilic cholecystitis: An infrequent cause of cholecys-tectomy. Gastroenterol Hepatol 1997;20:21-3.

7. Singh DK, Shankar R, Gondal R, Malhotra V, Mishra P. Idiopathic eosinophilic cholecystitis with cholelithiasis: A case report and review of literature. The Internet Journal of Surgery. 2008;16.

8. Shakov R, Simoni G, Villacin A, Baddoura W. Eosinophilic cholecys-titis, with a review of the literature. Ann Clin Lab Sc 2007;37:182-5.

9. Punia RP, Arya S, Jain P, Bal A, Mohan H. Eosinophilic and lympho-eosinophilic cholecystitis. Indian J Gastroenterol 2003;22:153-4.

10. Patel NB, Oto A, Thomas S. Multidetector CT of emergent biliary pathologic conditions. Radiographics 2013;33:1867-88.