Bilateral intralobar bronchopulmonary sequestration presenting as spontaneous hemopneumothorax: Case report

Introduction

Bilateral intralobar bronchopulmonary sequestration (BPS) is an exceptionally rare congenital anomaly. While BPS can present with recurrent infections or chronic respiratory symptoms, hemothorax as an initial manifestation is uncommon and potentially life-threatening.

Case presentation

A 17-year-old male presented with acute left-sided chest pain and tachypnea. The initial chest X-ray revealed a left-sided pneumothorax. After transferring to a children's hospital for management, a follow-up radiograph was obtained, showing a new fluid collection in addition to the pneumothorax. A chest tube was placed, yielding 500 mL of bright red blood, confirming hemothorax. A chest CT angiogram identified active arterial bleeding from the left lung apex. Video-assisted thoracoscopic surgery (VATS) was performed, revealing 1.5 L of hemothorax and ongoing bleeding from a chest wall artery and a severed arterial stump at the lung apex, adjacent to apical blebs. The bleeding was controlled with cauterization and clipping, and a wedge resection of the left lung apex was performed. The chest CT scan demonstrated additional cysts in the apex of the right lung. A month later, elective right-sided VATS, apical wedge resection, and pleurodesis were performed. Intraoperative findings included a systemic feeding artery along with associated cysts at the right lung apex, similar to the left side. The procedures were well-tolerated, and the patient recovered without complications. Final pathology made the diagnosis of bilateral intralobar BPS with complete resection of the lesions.

Conclusion

When spontaneous pneumothorax is complicated by hemothorax, underlying congenital anomalies such as intralobar BPS should be considered.