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Case Report: Identification of Mutations in LAMP2 in Two Chinese Infants With Danon Disease

Danon disease (DD) is a monogenic lysosomal storage disorder characterized by cardiomyopathy, skeletal myopathy, and variable degrees of intellectual disability. It is caused by a deficiency of lysosomal-associated membrane protein 2 (LAMP2). Two unrelated boys who presented with severe hypertrophic...

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Autores principales: Zhang, Luyan, Yang, Fan, Chen, Mei, Zhou, Ming, Qian, Tianwei, Mujtaba, Mohammed Omer, Mohammed, Abdul Haseeb, Yin, Jie, Cheng, Xueying, Chen, Jinlong, Qin, Yuming, Yang, Shiwei
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7831386/
https://www.ncbi.nlm.nih.gov/pubmed/33505424
http://dx.doi.org/10.3389/fgene.2020.589838
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author Zhang, Luyan
Yang, Fan
Chen, Mei
Zhou, Ming
Qian, Tianwei
Mujtaba, Mohammed Omer
Mohammed, Abdul Haseeb
Yin, Jie
Cheng, Xueying
Chen, Jinlong
Qin, Yuming
Yang, Shiwei
author_facet Zhang, Luyan
Yang, Fan
Chen, Mei
Zhou, Ming
Qian, Tianwei
Mujtaba, Mohammed Omer
Mohammed, Abdul Haseeb
Yin, Jie
Cheng, Xueying
Chen, Jinlong
Qin, Yuming
Yang, Shiwei
author_sort Zhang, Luyan
collection PubMed
description Danon disease (DD) is a monogenic lysosomal storage disorder characterized by cardiomyopathy, skeletal myopathy, and variable degrees of intellectual disability. It is caused by a deficiency of lysosomal-associated membrane protein 2 (LAMP2). Two unrelated boys who presented with severe hypertrophic cardiomyopathy and elevated levels of liver enzymes, and were diagnosed with Danon disease at a very young age, were investigated. One boy was diagnosed at 4 months old and died soon after; his mother also died of hypertrophic cardiomyopathy shortly after his birth. Another developed hypertrophic cardiomyopathy at 3 months old but reported no significant cardiovascular symptoms during more than 5 years follow-up. Genetic screening found compound variants of LAMP2 and MYH7 in both of them. This report highlights the clinical heterogeneity in DD. The timely identification of LAMP2 mutation plays a critical role in their treatment and family counseling.
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spelling pubmed-78313862021-01-26 Case Report: Identification of Mutations in LAMP2 in Two Chinese Infants With Danon Disease Zhang, Luyan Yang, Fan Chen, Mei Zhou, Ming Qian, Tianwei Mujtaba, Mohammed Omer Mohammed, Abdul Haseeb Yin, Jie Cheng, Xueying Chen, Jinlong Qin, Yuming Yang, Shiwei Front Genet Genetics Danon disease (DD) is a monogenic lysosomal storage disorder characterized by cardiomyopathy, skeletal myopathy, and variable degrees of intellectual disability. It is caused by a deficiency of lysosomal-associated membrane protein 2 (LAMP2). Two unrelated boys who presented with severe hypertrophic cardiomyopathy and elevated levels of liver enzymes, and were diagnosed with Danon disease at a very young age, were investigated. One boy was diagnosed at 4 months old and died soon after; his mother also died of hypertrophic cardiomyopathy shortly after his birth. Another developed hypertrophic cardiomyopathy at 3 months old but reported no significant cardiovascular symptoms during more than 5 years follow-up. Genetic screening found compound variants of LAMP2 and MYH7 in both of them. This report highlights the clinical heterogeneity in DD. The timely identification of LAMP2 mutation plays a critical role in their treatment and family counseling. Frontiers Media S.A. 2021-01-11 /pmc/articles/PMC7831386/ /pubmed/33505424 http://dx.doi.org/10.3389/fgene.2020.589838 Text en Copyright © 2021 Zhang, Yang, Chen, Zhou, Qian, Mujtaba, Mohammed, Yin, Cheng, Chen, Qin and Yang. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Genetics
Zhang, Luyan
Yang, Fan
Chen, Mei
Zhou, Ming
Qian, Tianwei
Mujtaba, Mohammed Omer
Mohammed, Abdul Haseeb
Yin, Jie
Cheng, Xueying
Chen, Jinlong
Qin, Yuming
Yang, Shiwei
Case Report: Identification of Mutations in LAMP2 in Two Chinese Infants With Danon Disease
title Case Report: Identification of Mutations in LAMP2 in Two Chinese Infants With Danon Disease
title_full Case Report: Identification of Mutations in LAMP2 in Two Chinese Infants With Danon Disease
title_fullStr Case Report: Identification of Mutations in LAMP2 in Two Chinese Infants With Danon Disease
title_full_unstemmed Case Report: Identification of Mutations in LAMP2 in Two Chinese Infants With Danon Disease
title_short Case Report: Identification of Mutations in LAMP2 in Two Chinese Infants With Danon Disease
title_sort case report: identification of mutations in lamp2 in two chinese infants with danon disease
topic Genetics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7831386/
https://www.ncbi.nlm.nih.gov/pubmed/33505424
http://dx.doi.org/10.3389/fgene.2020.589838
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