Kinjo M, Tanaka K, Ishimaru S, Shigematsu N, Sugiyama K
Acta Pathol Jpn. 1978 Mar;28(2):335-44.
A 37-year-old female with multiple pulmonary aneurysms, thrombotic obstruction of the inferior vena cava and mural thrombosis in the right heart was reported. Arterial blood culture repeatedly examined had been negative. The patient died of massive hemoptysis. This case was equivalent to Hughes-Stovin syndrome and was the first female case of typical Hughes-Stovin synrome. Review of the literature revealed 9 typical cases of this syndrome and 5 atypical cases who had solitary intrapulmonary aneurysm. It was obscure whether the typical cases were essentially different from the atypical ones or not, but the cases with solitary pulmonary aneurysm might proceed to the cases with multiple ones, as shown in this case. Pathogenesis of the syndrome has been controversial. In the present case, development of pulmonary aneurysms seemed to be closely related to thromboembolization derived from venous thrombosis due to artificial abortion.
报道了一名37岁女性，患有多发性肺动脉瘤、下腔静脉血栓形成阻塞以及右心壁血栓。多次进行的动脉血培养结果均为阴性。该患者死于大量咯血。此病例符合休斯 - 斯托文综合征，是典型休斯 - 斯托文综合征的首例女性病例。文献回顾显示，该综合征有9例典型病例和5例伴有孤立性肺内动脉瘤的非典型病例。典型病例与非典型病例是否本质上存在差异尚不清楚，但如本病例所示，伴有孤立性肺动脉瘤的病例可能会发展为多发性肺动脉瘤病例。该综合征的发病机制一直存在争议。在本病例中，肺动脉瘤的形成似乎与人工流产导致静脉血栓形成引起的血栓栓塞密切相关。
