Cargando…
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...
Autores principales: | , , , , , , , , , , , |
---|---|
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 |
_version_ | 1783641618970050560 |
---|---|
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. |
format | Online Article Text |
id | pubmed-7831386 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT zhangluyan casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT yangfan casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT chenmei casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT zhouming casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT qiantianwei casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT mujtabamohammedomer casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT mohammedabdulhaseeb casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT yinjie casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT chengxueying casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT chenjinlong casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT qinyuming casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease AT yangshiwei casereportidentificationofmutationsinlamp2intwochineseinfantswithdanondisease |