Chen Tian, Yu Jie, Zhang Na, Chen Chenghao, Fu Libing, Zeng Qi
Department of Thoracic Surgery, Beijing Children's Hospital, Capital Medical University, National Center for Children's Health, Beijing, China.
Department of Pathology, Beijing Children's Hospital, Capital Medical University, National Center for Children's Health, Beijing, China.
Front Pediatr. 2022 Jun 30;10:937563. doi: 10.3389/fped.2022.937563. eCollection 2022.
Patients with pulmonary sequestration (PS), a rare congenital lung malformation, are mostly asymptomatic. Recurrent localized infection is a major complication, while sudden hemothorax is extremely rare. We present a case of intralobar PS presenting as hemothorax secondary to spontaneous pneumothorax and comprehensively review the relevant literature.
A 16-year-old male presented with chest pain after strenuous exercise. Chest X-ray showed a moderate pneumothorax. After admission and conservative treatment, he developed dizziness, amaurosis, and urinary incontinence. Bedside chest X-ray suggested a massive pleural effusion, and hemothorax was further identified via catheter drainage. Contrast-enhanced computed tomography was performed, and no abnormal blood vessels or leakage of contrast agent were observed. As the hemoglobin level continued to drop, exploratory thoracoscopic surgery was performed immediately. The abnormal systemic artery supplying the lung tissue was found to be ruptured; therefore, ligation of the abnormal artery with resection of the diseased lung tissue was performed. Pathological examination revealed non-specific manifestations of PS. He was followed up for 1 year without related complications.
Our case suggests that the abnormal supply vessels of PS are unstable, which may cause sudden hemothorax. Therefore, patients with PS should undergo surgery promptly after diagnosis. In patients with hemothorax, we should consider the diagnosis of PS; however, contrast-enhanced computed tomography or angiography cannot confirm the diagnosis in all cases. Surgical intervention is recommended in emergency settings.
肺隔离症（PS）是一种罕见的先天性肺畸形，患者大多无症状。反复局部感染是主要并发症，而突发血胸极为罕见。我们报告一例叶内型PS表现为自发性气胸继发血胸的病例，并对相关文献进行全面回顾。
一名16岁男性在剧烈运动后出现胸痛。胸部X线显示中度气胸。入院保守治疗后，他出现头晕、黑矇和尿失禁。床边胸部X线提示大量胸腔积液，通过导管引流进一步确诊为血胸。进行了增强计算机断层扫描，未观察到异常血管或造影剂渗漏。由于血红蛋白水平持续下降，立即进行了 exploratory thoracoscopic surgery（此处原文有误，推测应为“胸腔镜探查手术”）。发现供应肺组织的异常体动脉破裂；因此，结扎异常动脉并切除病变肺组织。病理检查显示PS的非特异性表现。随访1年无相关并发症。
我们的病例表明，PS的异常供血血管不稳定，可能导致突发血胸。因此，PS患者确诊后应及时手术。对于血胸患者，应考虑PS的诊断；然而，增强计算机断层扫描或血管造影不能在所有病例中确诊。在紧急情况下建议进行手术干预。
