Nakagawa Yoichiro, Hayashi Kenshi, Tada Takayasu, Asakawa Miwako, Yoshida Shohei, Nomura Akihiro, Miwa Kenji, Furusho Hiroshi, Takamura Masayuki, Yasuda Toshihiko
Department of Cardiology, Ishikawa Prefectural Central Hospital, Japan.
Department of Cardiovascular Medicine, Kanazawa University Hospital, Japan.
Intern Med. 2025 Mar 15;64(6):857-863. doi: 10.2169/internalmedicine.3953-24. Epub 2024 Aug 8.
Danon disease (DD) is a rare lysosomal storage disorder resulting from pathogenic variants of the lysosome-associated membrane protein type 2 (LAMP-2) gene. The disease is characterized by severe cardiomyopathy, which rapidly progresses to end-stage heart failure. This case, with DD caused by a missense variant, exhibited slow progressive cardiomyopathy and survived for an extended period despite being a male. A pathological analysis revealed that only a minority of the samples exhibited autophagic vacuoles with unique sarcolemmal features, which are typical of DD. Importantly, LAMP-2 expression was absent and the myocardial tissue contained a substantial amount of p62-positive aggregates.
丹农病(DD)是一种罕见的溶酶体贮积症,由溶酶体相关膜蛋白2(LAMP - 2)基因的致病变体引起。该疾病的特征是严重的心肌病,可迅速进展为终末期心力衰竭。本病例由错义变体导致丹农病,表现为缓慢进展的心肌病,尽管为男性,但存活了较长时间。病理分析显示,只有少数样本表现出具有独特肌膜特征的自噬空泡,这是丹农病的典型特征。重要的是,LAMP - 2表达缺失,心肌组织中含有大量p62阳性聚集体。