Journal Name: Journal of Case Reports and Images in ... · thoracoscopic resection of intralobar...

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Early View Article: Online published version of an accepted article before publication in the

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Journal Name: Journal of Case Reports and Images in Surgery

Type of Article: Case Series

Title: Video-assisted thoracoscopic resection of intralobar pulmonary sequestration in

adults

Authors: Kristi Pence, Puja Gaur, Edward Chan, Min P. Kim

doi: To be assigned

Early view version published: February 16, 2016

How to cite the article: Pence K, Gaur P, Chan E, Kim M P. Video-assisted

thoracoscopic resection of intralobar pulmonary sequestration in adults. Journal of Case

Reports and Images in Surgery. Forthcoming 2016.

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TYPE OF ARTICLE: Case Series 1

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TITLE: Video-assisted thoracoscopic resection of intralobar pulmonary sequestration 3

in adults 4

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AUTHORS: 6

Kristi Pence, MD1, Puja Gaur, MD1,2, Edward Chan, MD1,2, Min P. Kim, MD1,2 7

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AFFILIATIONS: 9

1Division of Thoracic Surgery, Department of Surgery, Houston Methodist Hospital, 10

Houston, Texas, 11

2Department of Surgery, Weill Cornell Medical College, Houston Methodist Hospital, 12

Houston, Texas 13

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CORRESPONDING AUTHOR DETAILS 15

Min P. Kim, MD, FACS 16

6550 Fannin Street, Suite 1661 17

Houston, TX 77030 18

Phone number: 713-441-5177 19

Email:[email protected] 20

Fax: 713-790-5030 21

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Short Running Title: VATS for Adult Pulmonary Sequestration 23

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Guarantor of Submission : The corresponding author is the guarantor of 25

submission. 26

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in adults 33

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ABSTRACT 35

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Introduction 37

Intralobar pulmonary sequestration is a rare congenital abnormality that is usually 38

diagnosed in childhood. When diagnosed in adults, patients are usually symptomatic 39

and undergo open thoracotomy and lobectomy. However, video-assisted 40

thoracoscopic surgery (VATS) has become a viable alternative. 41

42

Case Series 43

We present three cases in which adults underwent VATS lobectomy. The resection 44

of the aberrant vessels was aided by the superior visualization of VATS without any 45

additional risk for the patients. All of the procedures were successfully completed 46

without any major morbidity. 47

48

Conclusion 49

VATS allows for superior visualization, decreased length of stay, decreased pain 50

medication, and less morbidity than thoracotomy, and should be considered for 51

management of adult patients with pulmonary sequestration. 52

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Keywords: Pulmonary sequestration, adult, VATS, lobectomy 54

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in adults 65

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INTRODUCTION 67

Pulmonary sequestration is a rare congenital abnormality that consists of a non-68

functioning segment of lung tissue in which there is no communication between the 69

sequestrated lung and the bronchial tree or pulmonary arteries. It is estimated to 70

comprise 0.15-6.4% of congenital pulmonary malformations and approximately 1% 71

of pulmonary resections [1]. In intralobar sequestration, the lung tissue lies within 72

same visceral pleura of the lobe and typically has pulmonary venous outflow, while 73

extralobar sequestration has its own visceral pleura and has systemic venous 74

outflow. The standard treatment for symptomatic intralobar sequestrations is a 75

lobectomy, which is typically performed through an open thoracotomy. Recently, 76

VATS lobectomy has grown in popularity. We present a series of three adults 77

diagnosed with intralobar pulmonary sequestrations safely treated with VATS 78

lobectomy. 79

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CASE SERIES 81

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Case 1 83

A 29-year old male presented to the clinic with a 10-year history of hemoptysis that 84

had worsened over the previous three months. Computed tomography (CT) showed 85

a left lower lobe pulmonary sequestration with a 1.5 cm anomalous vessel arising 86

from the thoracic aorta (Figure 1A). Left VATS was performed using a 4 cm utility 87

incision placed in the 4th intercostal space in the mid-axillary line, a 1 cm camera port 88

was placed in the 7th intercostal space posterior to posterior axillary line and a 1 cm 89

utility incision was placed in the 7th intercostal space in the mid-axillary line. The 90

aberrant vessel coming off the aorta was isolated (Figure 1B) using electrothermal 91

bipolar tissue sealing system and divided by vascular stapler. Three small branches 92

from pulmonary artery to left lower lobe were identified and divided. The patient’s 93

pain was well controlled and he was discharged on post-operative day 3 with 94


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resolution of his hemoptysis. At follow-up one month later, he had resolution of 95

hemoptysis. 96

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Case 2 98

A 17-year old male with an autism spectrum disorder and a history of recurrent left 99

lower lobe pneumonia for five years underwent a CT scan that showed a complex 100

cavitary lesion (9.3 x 7.1 cm) in the left lower lobe with an air-fluid collection and a 4 101

mm vessel arising from the descending thoracic aorta extending to the left lower lobe 102

consistent with a left lower lobe intralobar pulmonary sequestration. After a course of 103

Zosyn for four days, the abscess decreased in size. Patient then underwent left 104

VATS lower lobe lobectomy, as discussed in detail in Case 1. There were two small 105

anomalous vessels were noted to arise from the aorta going to left lower lobe, in 106

addition to a small branch from pulmonary artery to superior segment of left lower 107

lobe. The patient’s pain was controlled without narcotics. Repeated chest x-rays 108

showed increasing left apical pneumothorax despite chest tube remaining in place, 109

so he had an IR-placed chest tube placement. On post-operative day 5, chest x-ray 110

showed no pneumothorax so both chest tubes were discontinued. He was 111

discharged home on post-operative day 6 with an additional four weeks of 112

Augmentin and Doxycycline. He was discharged on post-operative day 6. Upon his 113

follow up visit one month later, the patient’s pneumonia had resolved. 114

115

Case 3 116

A 37-year old female presented to her primary care provider with complaints of 117

dyspnea and coughing that had started during a pregnancy the previous year. She 118

had suffered from severe GERD during pregnancy, which resolved after her child 119

was born, but continued to have a persistent cough and intermittent low fevers. A CT 120

of her chest without IV contrast showed that her right lower lobe had bronchiectasis. 121

A bronchoscopy showed irritation of mucosa and a small pocket of pus in the right 122

lower lobe and the washings were negative for malignancy. After course of Levaquin 123

for seven days, patient had a repeat CT with IV contrast showed right lower lobe 124

intralobar pulmonary sequestration with associated abscess (Figure 1C) supplied by 125

6mm branch from descending aorta just superior to the diaphragm. The patient 126


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underwent right VATS lower lobectomy. Due to the low location of the anomalous 127

branch from the descending aorta, the posterior axillary line incision was placed at 128

the 8th, instead of 7th, intercostal space. The 6 mm anomalous branch from the 129

descending aorta just superior to the diaphragm was isolated and divided (Figure 130

1D). The culture grew Staphylococcus aureus and patient was treated with Levaquin 131

for five days. Her pain was well controlled and she was discharged on post-operative 132

day 2. At follow-up one month after resection, she had resolution of her cough and 133

low-grade fevers. 134

135

DISCUSSION 136

Pulmonary sequestration is a rare congenital anomaly that usually manifests early in 137

life, but can be diagnosed as an adult. Symptoms are present in 84% of adults 138

diagnosed with pulmonary sequestration and 71-79% of sequestrations are in the left 139

lower lobe [2,3]. A retrospective analysis of 2625 patients showed that the most 140

common symptoms of pulmonary sequestration in adults are cough (69%), fever 141

(39%), hemoptysis (28%), and chest pain (11%), with 13% of patients being 142

asymptomatic [3]. Chest x-rays often simply show bronchiectasis or a hazy opacity. 143

Chest CT scans show mass lesions (49%), cystic lesions (29%), cavitary lesions 144

(12%), and pneumonic lesions (8%) [3,4]. The lack of specific symptoms and 145

radiographic findings can mimic a variety of clinical conditions. 146

The majority of lobectomies are still performed via thoracotomy, but the prevalence 147

of VATS resection has increased in recent years. Interestingly, VATS resection is 148

utilized more often in children than adults; 75% of lobectomies for sequestration in 149

children are performed via VATS compared 16% in adults [5]. In our series, two 150

patients had a long-standing history of recurrent symptoms, resulting in significant 151

scar tissue, abscess cavities and less than ideal tissue planes. In our experience, 152

VATS allows for better visualization of the planes and aberrant vessels, making them 153

less susceptible to injury to the vasculature or lung parenchyma. 154

A thorough review of the blood supply to the symptomatic portion of lung can aid in 155

diagnosis, as anomalous arteries arise from the thoracic aorta (81-86%), abdominal 156

aorta (7-19%), and phrenic artery (5.6%) [3,4]. The majority of sequestrations are 157

supplied by a single artery, but 21% are supplied by two or more arteries [3]. The 158


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size, number, and origin of the supplying artery are highly varied. The correct 159

identification of these branches is of vital importance to avoid vascular injury. VATS 160

is able to provide excellent visualization of the vasculature, thus providing a safe way 161

to identify and control the vessels. 162

163

CONCLUSION 164

All of our patients successfully underwent VATS lobectomy, despite dense 165

adhesions and abscess cavities, multiple aberrant vessels, and difficult to access 166

anatomy, with no post-operative complications. Additionally, our patients’ pain was 167

adequately controlled with oral pain medications. VATS lobectomy is a safe and 168

viable option for pulmonary sequestration resection and should be considered for 169

treatment of pulmonary sequestration. 170

171

CONFLICT OF INTEREST 172

The authors declare no conflicts of interest. No funding for this study. 173

174

AUTHOR’S CONTRIBUTIONS 175

Kristi Pence, MD 176

Group 1 - Conception and design, Acquisition of data, Analysis and interpretation of 177

data 178

Group 2 - Drafting the article, critical revision of the article 179

Group 3 - Final approval of the version to be published 180

181

Puja Gaur, MD 182


data 184

Group 2 - Critical revision of the article 185


187

Edward Y Chan, MD 188


data 190


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193

Min P. Kim, MD 194

Group 1 - Conception and design 195



198

REFERENCES 199

1. Van Raemdonck D, De Boeck K, Devlieger H, Demedts M, Moerman P, 200

Coosemans W, et al. Pulmonary sequestration: a comparison between 201

pediatric and adult patients. Eur J Cardiothorac Surg. 2001;19(4):388-395. 202

2. Wei Y, Li F. Pulmonary sequestration: a retrospective analysis of 2625 cases 203

in China. Eur J Cardiothorac Surg. 2011;40(1):39-42. 204

3. Sun X, Xiao Y. Pulmonary sequestration in adult patients: a retrospective 205

study. Eur J Cardiothorac Surg. 2015;48(2):279-282. 206

4. Liu C, Pu Q, Ma L, Mei J, Xiao Z, Liao H, et al. Video-assisted thoracic 207

surgery for pulmonary sequestration compared with posterolateral 208

thoracotomy. J Thorac Cardiovasc Surg. 2013;146(3):557-561. 209

5. Fievet L, Natale C, D'Journo XB, Coze S, Dubus JC, Guys JM, et al. 210

Congenital pulmonary airway malformation and sequestration: two 211

standpoints for a single condition. J Minim Access Surg. 2015;11(2):129-133. 212

213

FIGURE LEGEND 214

Figure 1: Aberrant vessel to pulmonary sequestration (A) – A computed tomography 215

image shows a 1.5 cm vessel arising from the descending aorta going into the left 216

lower lobe intralobar pulmonary sequestration. (B) – A thoracoscopic image of an 217

aberrant blood vessel arising from the aorta with vascular stapler going around the 218

isolated vessel. (C) – A computed tomography image shows a 6 mm vessel arising 219

from descending aorta. (D) – A thoracoscopic image of an aberrant blood vessel in 220

the inferior pulmonary ligament isolated by thoracoscopic instrument. 221

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FIGURE 223

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Figure 1: Aberrant vessel to pulmonary sequestration (A) – A computed tomography 227

image shows a 1.5 cm vessel arising from the descending aorta going into the left 228

lower lobe intralobar pulmonary sequestration. (B) – A thoracoscopic image of an 229

aberrant blood vessel arising from the aorta with vascular stapler going around the 230

isolated vessel. (C) – A computed tomography image shows a 6 mm vessel arising 231

from descending aorta. (D) – A thoracoscopic image of an aberrant blood vessel in 232

the inferior pulmonary ligament isolated by thoracoscopic instrument. 233

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