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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...

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Autores principales: Sun, LiuQing, Shen, DingGuo, Xiong, Ting, Zhou, Zhibin, Lu, Xianghui, Cui, Fang
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Association of Basic Medical Sciences of Federation of Bosnia and Herzegovina 2020
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.
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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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