An incidental and uncommon pulmonary sequestration with an ...

CASE REPORT – OPEN ACCESS International Journal of Surgery Case Reports 4 (2013) 861–862

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An incidental and uncommon pulmonary sequestration with an uncommon feeding artery夽 Masatsugu Hamaji a,∗ , Bryan M. Burt a , Syed Osman Ali a , Jelena Mirkovic b a b

The Brigham & Women’s Hospital, Division of Thoracic Surgery, USA Department of Pathology, The Brigham & Women’s Hospital, Division of Thoracic Surgery, USA

a r t i c l e

i n f o

Article history: Received 12 May 2013 Received in revised form 9 July 2013 Accepted 22 July 2013 Available online 3 August 2013 Keywords: Infection Thoracoscopy Surgery

a b s t r a c t INTRODUCTION: Pulmonary sequestration is a rare congenital anomaly and most intralobar sequestrations were located in lower lobes. There is little information on middle lobe intralobar sequestration. PRESENTATION OF CASE: A 44 year-old man with right middle lobe syndrome was referred for surgical management. He underwent video-assisted thoracoscopic right middle lobectomy and his postoperative course was uneventful. Pathology is notable for an incidental intralobar sequestration. DISCUSSION: Our case was unique in that the location of incidental pulmonary sequestration is uncommon and in that its feeding artery was uncommonly located in the fissure. CONCLUSION: We report an extremely rare case of right middle lobe intralobar sequestration with a feeding artery in the fissure. © 2013 The Authors. Published by Elsevier Ltd on behalf of Surgical Associates Ltd. Open access under CC BY license.

1. Introduction Pulmonary sequestration is a rare congenital anomaly and most of intralobar sequestrations were located in lower lobes [1,2]. Upper lobe intralobar sequestrations were very rare and a middle lobe intralobar sequestration is even rarer. There is very little information on right middle lobe sequestration or its feeding artery because of no detailed description or no case report to date on an adult patient’s right middle lobe sequestration.

lobe pulmonary vein branches were divided with a linear stapler. Subsequently, the minor and major fissures were completed with linear staplers. Between pulmonary artery branch and the middle lobe bronchus, a presumably systemic artery was identified (Fig. 1) and it was divided en bloc with the pulmonary artery branch after dissecting off the bronchus. No venous return to a systemic vein was noted. His postoperative course was uneventful and the patient was discharged on postoperative day two. Pathology showed intralobar pulmonary sequestration (Fig. 2).

2. Presentation of a case A 44 year-old man with multiple hospitalizations due to pneumonia presented to us for a surgical consultation. His family history was remarkable for two brothers with congenital right lower lobe atelectasis, both requiring lobectomies. Chest computed tomography is notable for atelectasis of a portion of the middle lobe. Prior to surgery, the anatomy of the airway was normal on bronchoscopy and bronchoalveolar lavage was positive for Hemophilus influenzae on culture and nagetive on cytology. Video-assisted thoracoscopic right middle lobectomy was performed with four ports. The middle lobe was noted to adhere moderately to the pericardium and the adhesion was taken down easily. Middle

∗ Corresponding author at: The Brigham & Women’s Hospital, 75 Francis Street, Boston, MA 02115, USA. Tel.: +1 617 732 6824; fax: +1 617 566 3441. E-mail address: [email protected] (M. Hamaji).

Fig. 1. The intraoperative photo showed a feeding artery (arrow) between the pulmonary artery branch and the bronchus.

2210-2612 © 2013 The Authors. Published by Elsevier Ltd on behalf of Surgical Associates Ltd. Open access under CC BY license. http://dx.doi.org/10.1016/j.ijscr.2013.07.022

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M. Hamaji et al. / International Journal of Surgery Case Reports 4 (2013) 861–862

Fig. 2. Gross photograph of the specimen demonstrates a segment of lung with effacement of normal architecture with diffuse fibrosis, scaring, and bronchiectasis. An enlarged vessel is indicated by a white arrow. Microscopic hematoxylin and eosin stained tissue section demonstrates obliteration of the normal architecture with chronic inflammation, interstitial fibrosis, bronchiectasis and a dominant enlarged elastic vessel.

3. Discussion In 373 cases (93%) out of 400 cases in Savic’s report, the origin of a feeding artery was documented. There is no information on the feeding artery for right middle lobe sequestration, although only one report on a pediatric patient described multiple systemic feeding arteries from the internal mammary artery and the diaphragmatic artery [3]. Sequestrations can be fed by a pulmonary artery branch, but in our case, there was an artery in the fissure which is unlikely a pulmonary artery branch macroscopically and microscopically. Video-assisted thoracoscopic surgery (VATS) has been reported on pulmonary sequestration recently. Potential barriers to VATS include intrapleural adhesion due to an inflammatory process and dissecting and dividing of a feeding artery, while VATS case series reported low rates of conversion to a thoracotomy [4,5]. Our case was unique in that the location of incidental pulmonary sequestration is uncommon and in that its feeding artery was uncommonly located in the fissure. Our case suggests that in a case of a middle lobe syndrome, an intralobar sequestration is a differential diagnosis, thus, a feeding artery can appear in the fissure. 4. Conclusion We reported an extremely rare case of right middle lobe intralobar sequestration with a feeding artery in the fissure. Confict of interest None declared.

Funding None. Ethical approval Written informed consent was obtained from the patient for publication of this case report and case series and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request. Authors’ contributions Masatsugu Hamaji, writing the manuscript; Bryan Burt, writing the manuscript; Syed Osman Ali, writing the manuscript; Jelena Mirkovic, writing the manuscript. References [1].Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung sequestration: report of seven cases and review of 540 published cases. Thorax 1979;34(February (1)):96–101. [2].Wei Y, Li F. Pulmonary sequestration: a retrospective analysis of 2625 cases in China. European Journal Cardio-Thoracic Surgery 2011;40(July (1)):e39–42 [Epub 2011 April 2]. [3].Lau YL, Radhi JM, Blesovsky A, Hunter AS. A case of middle lobe pulmonary sequestration. Thorax 1986;41(October (10)):810–1. [4].Kestenholz PB, Schneiter D, Hillinger S, Lardinois D, Weder W. Thoracoscopic treatment of pulmonary sequestration. European Journal Cardio-Thoracic Surgery 2006;29(May (5)):815–8. [5].Tsang FH, Chung SS, Sihoe AD. Video-assisted thoracic surgery for bronchopulmonary sequestration. Interactive CardioVasc Thoracic Surgery 2006;5(August (4)):424–6.