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Limb-girdle muscular dystrophy due to GMPPB mutations: A case report and comprehensive literature review
Mutations in the guanosine diphosphate mannose (GDP-mannose) pyrophosphorylase B (GMPPB) gene are rare. To date, 72 cases with GMPPB gene mutations have been reported. Herein, we reported a case of a 29-year-old Chinese male presenting with limb-girdle muscular dystrophy (LGMD) who was found to have...
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Association of Basic Medical Sciences of Federation of Bosnia and Herzegovina
2020
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7202191/ https://www.ncbi.nlm.nih.gov/pubmed/30684953 http://dx.doi.org/10.17305/bjbms.2019.3992 |
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author | Sun, LiuQing Shen, DingGuo Xiong, Ting Zhou, Zhibin Lu, Xianghui Cui, Fang |
author_facet | Sun, LiuQing Shen, DingGuo Xiong, Ting Zhou, Zhibin Lu, Xianghui Cui, Fang |
author_sort | Sun, LiuQing |
collection | PubMed |
description | Mutations in the guanosine diphosphate mannose (GDP-mannose) pyrophosphorylase B (GMPPB) gene are rare. To date, 72 cases with GMPPB gene mutations have been reported. Herein, we reported a case of a 29-year-old Chinese male presenting with limb-girdle muscular dystrophy (LGMD) who was found to have two heterozygous GMPPB mutations. The patient had a progressive limb weakness for 19 years. His parents and elder brother were healthy. On examination he had a waddling gait and absent tendon reflexes in all four limbs. Electromyography showed myogenic damage. Muscle magnetic resonance imaging (MRI) showed fatty degeneration in the bilateral medial thigh muscles. High-throughput gene panel sequencing revealed that the patient carried compound heterozygous mutations in the GMPPB gene, c.553C>T (p.R185C, maternal inheritance) and c.346C>T (p.P116S, paternal inheritance). This case provides additional information regarding the phenotypic spectrum of GMPPB mutations in the Chinese population. |
format | Online Article Text |
id | pubmed-7202191 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | Association of Basic Medical Sciences of Federation of Bosnia and Herzegovina |
record_format | MEDLINE/PubMed |
spelling | pubmed-72021912020-05-06 Limb-girdle muscular dystrophy due to GMPPB mutations: A case report and comprehensive literature review Sun, LiuQing Shen, DingGuo Xiong, Ting Zhou, Zhibin Lu, Xianghui Cui, Fang Bosn J Basic Med Sci Case Report Mutations in the guanosine diphosphate mannose (GDP-mannose) pyrophosphorylase B (GMPPB) gene are rare. To date, 72 cases with GMPPB gene mutations have been reported. Herein, we reported a case of a 29-year-old Chinese male presenting with limb-girdle muscular dystrophy (LGMD) who was found to have two heterozygous GMPPB mutations. The patient had a progressive limb weakness for 19 years. His parents and elder brother were healthy. On examination he had a waddling gait and absent tendon reflexes in all four limbs. Electromyography showed myogenic damage. Muscle magnetic resonance imaging (MRI) showed fatty degeneration in the bilateral medial thigh muscles. High-throughput gene panel sequencing revealed that the patient carried compound heterozygous mutations in the GMPPB gene, c.553C>T (p.R185C, maternal inheritance) and c.346C>T (p.P116S, paternal inheritance). This case provides additional information regarding the phenotypic spectrum of GMPPB mutations in the Chinese population. Association of Basic Medical Sciences of Federation of Bosnia and Herzegovina 2020-05 /pmc/articles/PMC7202191/ /pubmed/30684953 http://dx.doi.org/10.17305/bjbms.2019.3992 Text en Copyright: © The Author(s) (2020) http://creativecommons.org/licenses/by-nc-sa/4.0 This work is licensed under a Creative Commons Attribution 4.0 International License |
spellingShingle | Case Report Sun, LiuQing Shen, DingGuo Xiong, Ting Zhou, Zhibin Lu, Xianghui Cui, Fang Limb-girdle muscular dystrophy due to GMPPB mutations: A case report and comprehensive literature review |
title | Limb-girdle muscular dystrophy due to GMPPB mutations: A case report and comprehensive literature review |
title_full | Limb-girdle muscular dystrophy due to GMPPB mutations: A case report and comprehensive literature review |
title_fullStr | Limb-girdle muscular dystrophy due to GMPPB mutations: A case report and comprehensive literature review |
title_full_unstemmed | Limb-girdle muscular dystrophy due to GMPPB mutations: A case report and comprehensive literature review |
title_short | Limb-girdle muscular dystrophy due to GMPPB mutations: A case report and comprehensive literature review |
title_sort | limb-girdle muscular dystrophy due to gmppb mutations: a case report and comprehensive literature review |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7202191/ https://www.ncbi.nlm.nih.gov/pubmed/30684953 http://dx.doi.org/10.17305/bjbms.2019.3992 |
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