Omori Taku, Nakamori Shiro, Ohta-Ogo Keiko, Matsuda Akimasa, Ogihara Yoshito, Yamada Norikazu, Imanaka-Yoshida Kyoko, Ito Masaaki, Dohi Kaoru
Department of Cardiology and Nephrology, Mie University Hospital, Tsu, Japan.
Department of Pathology, National Cerebral and Cardiovascular Center, Suita, Japan.
Pulm Circ. 2020 Nov 23;10(4):2045894020960600. doi: 10.1177/2045894020960600. eCollection 2020 Oct-Dec.
Pulmonary capillary hemangiomatosis is a rare form of pulmonary artery hypertension; to date, only few descriptions of myocardial pathology in pulmonary capillary hemangiomatosis have been reported in the literature. We report the case of a Japanese female patient who was diagnosed with pulmonary capillary hemangiomatosis combined with acute myocardial inflammation on performing autopsy. She was admitted to our hospital because of acute pneumonia and subsequently suddenly developed severe hypoxemia with breathing difficulty and died 13 days after admission. At autopsy, the histology of the lung was consistent with pulmonary capillary hemangiomatosis. Additionally, a diffuse severe infiltration of inflammatory cells was associated with edema in the myocardium. Myocytolysis was limited and fibrosis was absent. To the best of our knowledge, pulmonary capillary hemangiomatosis with acute myocarditis-like histological findings has been described for the first time through our case.
肺毛细血管血管瘤病是一种罕见的肺动脉高压形式；迄今为止，文献中仅有少数关于肺毛细血管血管瘤病心肌病理学的描述。我们报告一例日本女性患者，尸检时被诊断为肺毛细血管血管瘤病合并急性心肌炎症。她因急性肺炎入院，随后突然出现严重低氧血症伴呼吸困难，入院13天后死亡。尸检时，肺组织学符合肺毛细血管血管瘤病。此外，心肌存在弥漫性严重炎性细胞浸润并伴有水肿。心肌溶解有限且无纤维化。据我们所知，通过我们的病例首次描述了具有急性心肌炎样组织学表现的肺毛细血管血管瘤病。
