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Identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an Iranian patient with myofibrillar myopathy

Myofibrillar myopathies (MFMs) are rare genetic and slowly progressive neuromuscular disorders. Several pathogenic mutations have been reported in MFM-related genes including DES, CRYAB, MYOT, LDB3 or ZASP, FLNC, BAG3, FHL1 and DNAJB6. Although MFMs is commonly inherited in an autosomal dominant man...

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Autores principales: Ebrahimzadeh-Vesal, Reza, Teymoori, Atieh, Dourandish, Ali Mohammad, Azimi-Nezhad, Mohsen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Chongqing Medical University 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6303478/
https://www.ncbi.nlm.nih.gov/pubmed/30591934
http://dx.doi.org/10.1016/j.gendis.2018.09.004
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author Ebrahimzadeh-Vesal, Reza
Teymoori, Atieh
Dourandish, Ali Mohammad
Azimi-Nezhad, Mohsen
author_facet Ebrahimzadeh-Vesal, Reza
Teymoori, Atieh
Dourandish, Ali Mohammad
Azimi-Nezhad, Mohsen
author_sort Ebrahimzadeh-Vesal, Reza
collection PubMed
description Myofibrillar myopathies (MFMs) are rare genetic and slowly progressive neuromuscular disorders. Several pathogenic mutations have been reported in MFM-related genes including DES, CRYAB, MYOT, LDB3 or ZASP, FLNC, BAG3, FHL1 and DNAJB6. Although MFMs is commonly inherited in an autosomal dominant manner, the inheritance pattern and novel mutated genes are not thoroughly elucidated in some cases. Here, we report discovery of a novel nonsense mutation in a 29-year-old Iranian male patient with motor disorders and deformity in his lower limbs. His parents are second cousins. Hereditary Motor Sensory Neuropathy as initial genetic diagnosis was ruled out. Whole exome sequencing using NGS on Illumina HiSeq4000 platform was performed to identify the disease and possible mutated gene(s). Our data analysis identified a homozygous nonsense unreported c.C415T (p.R139X) variant on kyphoscoliosis peptidase (KY) gene (NM_178554: exon4). Sanger sequencing of this mutation has been performed for his other related family members. Sequencing and segregation analysis was confirmed the NGS results and autosomal recessive inheritance pattern of the disease.
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spelling pubmed-63034782018-12-27 Identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an Iranian patient with myofibrillar myopathy Ebrahimzadeh-Vesal, Reza Teymoori, Atieh Dourandish, Ali Mohammad Azimi-Nezhad, Mohsen Genes Dis Article Myofibrillar myopathies (MFMs) are rare genetic and slowly progressive neuromuscular disorders. Several pathogenic mutations have been reported in MFM-related genes including DES, CRYAB, MYOT, LDB3 or ZASP, FLNC, BAG3, FHL1 and DNAJB6. Although MFMs is commonly inherited in an autosomal dominant manner, the inheritance pattern and novel mutated genes are not thoroughly elucidated in some cases. Here, we report discovery of a novel nonsense mutation in a 29-year-old Iranian male patient with motor disorders and deformity in his lower limbs. His parents are second cousins. Hereditary Motor Sensory Neuropathy as initial genetic diagnosis was ruled out. Whole exome sequencing using NGS on Illumina HiSeq4000 platform was performed to identify the disease and possible mutated gene(s). Our data analysis identified a homozygous nonsense unreported c.C415T (p.R139X) variant on kyphoscoliosis peptidase (KY) gene (NM_178554: exon4). Sanger sequencing of this mutation has been performed for his other related family members. Sequencing and segregation analysis was confirmed the NGS results and autosomal recessive inheritance pattern of the disease. Chongqing Medical University 2018-10-02 /pmc/articles/PMC6303478/ /pubmed/30591934 http://dx.doi.org/10.1016/j.gendis.2018.09.004 Text en © 2018 Chongqing Medical University. Production and hosting by Elsevier B.V. http://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Article
Ebrahimzadeh-Vesal, Reza
Teymoori, Atieh
Dourandish, Ali Mohammad
Azimi-Nezhad, Mohsen
Identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an Iranian patient with myofibrillar myopathy
title Identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an Iranian patient with myofibrillar myopathy
title_full Identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an Iranian patient with myofibrillar myopathy
title_fullStr Identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an Iranian patient with myofibrillar myopathy
title_full_unstemmed Identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an Iranian patient with myofibrillar myopathy
title_short Identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an Iranian patient with myofibrillar myopathy
title_sort identification of a novel nonsense mutation in kyphoscoliosis peptidase gene in an iranian patient with myofibrillar myopathy
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6303478/
https://www.ncbi.nlm.nih.gov/pubmed/30591934
http://dx.doi.org/10.1016/j.gendis.2018.09.004
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