Pulmonary Sequestration with Rare Blood Supply in an Asymptomatic Adult Female

Special Article - Surgical Case Reports

Austin J Surg. 2015; 2(7): 1076.

Pulmonary Sequestration with Rare Blood Supply in an Asymptomatic Adult Female

Nieh CC1,2*, Chua YC¹ and Agasthian T¹

¹Department of Cardiothoracic and Vascular Surgery, National University Hospital, Singapore

²Yong Loo Lin School of Medicine, National University of Singapore, Singapore

*Corresponding author: Nieh CC, Department of Cardiothoracic and Vascular Surgery, National University Hospital, Singapore

Received: September 28, 2015; Accepted: November 19, 2015; Published: November 26, 2015

Abstract

Intralobar pulmonary sequestration with two aberrant vessels connected to aorta is very rare. We describe the case of a 61-year-oldasymptomatic Chinese female with an incidental finding of abnormal chest radiograph showing a left lower lobe mass. Computed Tomography scan of the Thorax (CT Thorax) revealed a cystic mass in the left lower lobe, supplied by two branches from the thoracic aorta, likely to be intra lobar pulmonary sequestration. A dystrophic bronchial tree that did not communicate with the normal bronchial tree was identified. The sequestered lung was completely excised via Video-Assisted Thoracoscopic Surgery (VATS).

The patient had an uncomplicated clinical course after surgery and was discharged on postoperative-day three in satisfactory condition. Histology reviewed mild chronic inflammation along with overlying interstitial fibrosis and bronchial metaplasia. In addition, bronchial plugging along with an abundant arterial supply was identified to be consistent with intralobar pulmonary sequestration

CT Thorax is ideal to visualise the vascular anatomy which is essential for surgical management. Were commend that surgical segmental resection with VATS should continue to be the standard of care in asymptomatic adult patients with this rare condition.

Keywords: Pulmonary sequestration; Thoracoscopic; Case report; Asymptomatic; Adult

Introduction

Pulmonary sequestration is a rare condition contributing to 0.15-6.4%ofallcongenitalmalformations of the lower respiratory tract [1,2]. Typically, it comprises of systemic arterial supply, usually derived from an aberrant branch of the lower thoracic or upper abdominal aorta, to an associated anomalous lung segment with variations in its venous drainage, usually to the left atrium, sometimes right atrium, vena cava, or azygous systems. These tissues are not connected to the trachea bronchial tree. Sequestrations are typically classified an atomically into intralobar and extra lobar forms, with the former being defined as a lung segment contained within the native pleural lining while the latter is contained in its own pleural investment.

Amongst both forms of sequestrations, intralobar type is the commonest, approximately 70 to 95 percent of all sequestrations. Sixty percent of intrapulmonary sequestrations are diagnosed within the first decade of life and are more common in male’s by a 3:1 ratio1. Symptoms are variable, depending on the anatomical location of the lesion, but usually related to chronic respiratory infection although sequestrations can be incidental findings on radiological investigations.

The exact pathogenesis of pulmonary sequestration is unclear, for which several theories have been suggested to elucidate the embryologic mechanism. There is another hypothesis that describes a disruption during the processes of lung development which distorts its arterial blood supply which subsequently leads to retention and proliferation of the nascent systemic capillary network [3,4]. Till now, no one is certain about the embryological development of this condition. Usual age of presentation for intra pulmonary sequestration and extra pulmonary sequestration are adolescence with signs and symptoms of recurrent infection, and infancy with respiratory failure respectively.

We now demonstrate a recent case of intralobar pulmonary sequestration supplied by two aberrant vessels arising from the aorta at our institution who presented at an age of 61withoutsymptoms.

Case

The patient is a sixty-one year old Chinese female with no significant past medical history. Computed tomography of Thorax (Figures 1 & 2) revealed a cystic mass in the left lower lobe, supplied by two solitary branches from the thoracic aorta likely intra lobar pulmonary sequestration. There was no pleural effusion or consolidation. A dystrophic bronchial tree that did not communicate with the normal bronchial tree was identified. Chest radiography showed a left lower lobe mass.