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Manuscript Accepted Early View Article
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Early View Article: Online published version of an accepted article before publication in the
final form.
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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Manuscript Accepted Early View Article
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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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Manuscript Accepted Early View Article
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TITLE: Video-assisted thoracoscopic resection of intralobar pulmonary sequestration 32
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
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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
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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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Manuscript Accepted Early View Article
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TITLE: Video-assisted thoracoscopic resection of intralobar pulmonary sequestration 64
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
Manuscript Accepted Early View Article
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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
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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
Manuscript Accepted Early View Article
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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
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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
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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
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CONFLICT OF INTEREST 172
The authors declare no conflicts of interest. No funding for this study. 173
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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
Group 1 - Conception and design, Acquisition of data, Analysis and interpretation of 183
data 184
Group 2 - Critical revision of the article 185
Group 3 - Final approval of the version to be published 186
187
Edward Y Chan, MD 188
Group 1 - Conception and design, Acquisition of data, Analysis and interpretation of 189
data 190
Manuscript Accepted Early View Article
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Group 2 - Critical revision of the article 191
Group 3 - Final approval of the version to be published 192
193
Min P. Kim, MD 194
Group 1 - Conception and design 195
Group 2 - Critical revision of the article 196
Group 3 - Final approval of the version to be published 197
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
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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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Manuscript Accepted Early View Article
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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