Suppr超能文献

以缓慢进展性心肌病为表现且溶酶体相关膜蛋白2(LAMP-2)基因存在新型错义变异的Danon病

Danon Disease Presenting with Slowly Progressive Cardiomyopathy and Harboring a Novel Missense Variant in the Lysosome-associated Membrane Protein Type 2 (LAMP-2) Gene.

作者信息

Nakagawa Yoichiro, Hayashi Kenshi, Tada Takayasu, Asakawa Miwako, Yoshida Shohei, Nomura Akihiro, Miwa Kenji, Furusho Hiroshi, Takamura Masayuki, Yasuda Toshihiko

机构信息

Department of Cardiology, Ishikawa Prefectural Central Hospital, Japan.

Department of Cardiovascular Medicine, Kanazawa University Hospital, Japan.

出版信息

Intern Med. 2025 Mar 15;64(6):857-863. doi: 10.2169/internalmedicine.3953-24. Epub 2024 Aug 8.

Abstract

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.

摘要

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。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/19c8/11986313/ccbf69db5ff8/1349-7235-64-0857-g001.jpg

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验