表现为缓慢进展性心肌病并携带溶酶体相关膜蛋白 2 型 (LAMP-2) 基因新型错义变异的达农病:病例报告。

IF 1 4区 医学 Q3 MEDICINE, GENERAL & INTERNAL
Internal Medicine Pub Date : 2025-03-15 Epub Date: 2024-08-08 DOI:10.2169/internalmedicine.3953-24
Yoichiro Nakagawa, Kenshi Hayashi, Takayasu Tada, Miwako Asakawa, Shohei Yoshida, Akihiro Nomura, Kenji Miwa, Hiroshi Furusho, Masayuki Takamura, Toshihiko Yasuda
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引用次数: 0

摘要

达农病(DD)是一种罕见的溶酶体储积症,由溶酶体相关膜蛋白2型(LAMP-2)基因的致病变体引起。该病的特征是严重的心肌病,并迅速发展为终末期心力衰竭。该病例的DD是由错义变异引起的,表现为缓慢进展的心肌病,尽管是男性,但存活时间较长。病理分析表明,只有少数样本表现出具有独特肌浆特征的自噬空泡(AVSFs),这是DD的典型特征。重要的是,LAMP-2没有表达,心肌组织中含有大量p62阳性聚集体。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Danon Disease Presenting with Slowly Progressive Cardiomyopathy and Harboring a Novel Missense Variant in the Lysosome-associated Membrane Protein Type 2 (LAMP-2) Gene.

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.

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来源期刊
Internal Medicine
Internal Medicine 医学-医学:内科
CiteScore
1.90
自引率
8.30%
发文量
0
审稿时长
2.2 months
期刊介绍: Internal Medicine is an open-access online only journal published monthly by the Japanese Society of Internal Medicine. Articles must be prepared in accordance with "The Uniform Requirements for Manuscripts Submitted to Biomedical Journals (see Annals of Internal Medicine 108: 258-265, 1988), must be contributed solely to the Internal Medicine, and become the property of the Japanese Society of Internal Medicine. Statements contained therein are the responsibility of the author(s). The Society reserves copyright and renewal on all published material and such material may not be reproduced in any form without the written permission of the Society.
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