Isowa Masahide, Tanaka Satona, Nakanobo Ryo, Yamada Yoshito, Date Hiroshi
Department of Thoracic Surgery, Kyoto University Hospital, Shogoin-kawahara-cho 54, Sakyo-ku, Kyoto, 606-8507, Japan.
Surg Case Rep. 2020 Oct 22;6(1):272. doi: 10.1186/s40792-020-01001-w.
Pulmonary metastasis of scalp angiosarcoma (SA) is a rare, but life-threatening disease, challenging to diagnose and manage. We report two cases of pneumothorax and hemothorax with pathologically proven metastasis of SA in the parietal pleura, which was not predictable from images and difficult to manage.
A 73-year-old man with SA underwent chemoradiotherapy and surgical resection for primary skin lesion, was sent to our department to treat right empyema, which was developed during chest tube drainage for pneumothorax. Computed tomography (CT) showed multiple bullous lesions. We performed repetitive video-assisted thoracoscopic surgery (VATS) for the debridement and hemostasis; however, hemothorax was uncontrollable. The repeated cytology of pleural effusion showed no malignancy. We eventually performed fenestration and metastatic SA was pathologically diagnosed by the biopsy of parietal pleura. The patient developed respiratory failure and uncontrolled anemia, which were fatal.
A 71-year-old man with SA previously treated with chemoradiotherapy was referred to our department for left pneumothorax. CT showed multiple bullous lesions at apex without any changes at parietal pleura. VATS was performed and the apex bullous lesion with air leakage was resected. The parietal pleura showed several dark-red spots and the biopsy was undertaken. The pathological diagnosis was a metastasis of SA along with visceral pleura and parietal pleura. The patient then developed right pneumothorax and left hemopneumothorax. Bilateral pleurodesis was ineffective and the patient died due to deteriorating general condition.
In patients with a history of SA who develop pneumothorax and hemothorax, metastatic SA to visceral and parietal pleura should be always considered. Surgical biopsy, not cytology, is needed for pathological diagnosis. Lesions in the parietal pleura prior to hemothorax were thoracoscopically observed in one case. Surgeons must recognize that conventional surgical intervention or pleurodesis will have unsatisfactory results.
头皮血管肉瘤（SA）的肺转移是一种罕见但危及生命的疾病，诊断和治疗具有挑战性。我们报告两例气胸和血胸病例，经病理证实SA转移至壁层胸膜，影像学无法预测，治疗困难。
患者A：一名73岁患有SA的男性因原发性皮肤病变接受了放化疗和手术切除，因气胸胸腔闭式引流期间并发右侧脓胸被送至我科治疗。计算机断层扫描（CT）显示多发大疱性病变。我们多次进行电视辅助胸腔镜手术（VATS）清创止血，但血胸无法控制。反复的胸腔积液细胞学检查未发现恶性肿瘤。我们最终进行了开窗术，通过壁层胸膜活检病理诊断为转移性SA。患者出现呼吸衰竭和难以控制的贫血，最终死亡。
患者B：一名71岁曾接受放化疗的SA男性因左侧气胸转诊至我科。CT显示肺尖多发大疱性病变，壁层胸膜无变化。行VATS手术，切除漏气的肺尖大疱性病变。壁层胸膜可见数个暗红色斑点并进行了活检。病理诊断为SA转移至脏层和壁层胸膜。随后患者出现右侧气胸和左侧血气胸。双侧胸膜固定术无效，患者因全身状况恶化死亡。
有SA病史且出现气胸和血胸的患者，应始终考虑SA转移至脏层和壁层胸膜的可能。病理诊断需要手术活检而非细胞学检查。1例患者在血胸发生前通过胸腔镜观察到壁层胸膜病变。外科医生必须认识到传统的手术干预或胸膜固定术效果不佳。
