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Somatic mosaicism for Duchenne dystrophy: Evidence for genetic normalization mitigating muscle symptoms

We describe a young adult male presenting with cardiac failure necessitating cardiac transplantation 7 months after presentation. Skeletal muscle biopsy showed mosaic immunostaining for dystrophin. DNA studies showed somatic mosaicism for a nonsense mutation in the dystrophin gene (Arg2905X). The fr...

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Autores principales: Kesari, Akanchha, Neel, Robert, Wagoner, Lynne, Harmon, Brennan, Spurney, Christopher, Hoffman, Eric P
Formato: Texto
Lenguaje:English
Publicado: Wiley Subscription Services, Inc., A Wiley Company 2009
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2729699/
https://www.ncbi.nlm.nih.gov/pubmed/19530190
http://dx.doi.org/10.1002/ajmg.a.32891
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author Kesari, Akanchha
Neel, Robert
Wagoner, Lynne
Harmon, Brennan
Spurney, Christopher
Hoffman, Eric P
author_facet Kesari, Akanchha
Neel, Robert
Wagoner, Lynne
Harmon, Brennan
Spurney, Christopher
Hoffman, Eric P
author_sort Kesari, Akanchha
collection PubMed
description We describe a young adult male presenting with cardiac failure necessitating cardiac transplantation 7 months after presentation. Skeletal muscle biopsy showed mosaic immunostaining for dystrophin. DNA studies showed somatic mosaicism for a nonsense mutation in the dystrophin gene (Arg2905X). The frequency of normal versus mutant genes were determined in blood/DNA (50:50), muscle/DNA (80:20) and muscle/mRNA (90:10). These data are consistent with genetic normalization processes that may biochemically rescue skeletal muscle in male somatic mosaic patients mitigating muscle symptoms (gradual loss of dystrophin-negative skeletal muscle tissue replaced by dystrophin-positive stem cells). To our knowledge, this is only the second reported case of a clinically ascertained patient showing somatic mosaicism for Duchenne muscular dystrophy (DMD). We hypothesize that many somatic mosaic males for DMD exist, yet they are not detected clinically due to genetic normalization. Somatic mosaicism for DMD should be considered in acute heart failure with dilated cardiomyopathy, as genetic normalization in heart is unlikely to occur.
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spelling pubmed-27296992009-08-27 Somatic mosaicism for Duchenne dystrophy: Evidence for genetic normalization mitigating muscle symptoms Kesari, Akanchha Neel, Robert Wagoner, Lynne Harmon, Brennan Spurney, Christopher Hoffman, Eric P Am J Med Genet A Clinical Report We describe a young adult male presenting with cardiac failure necessitating cardiac transplantation 7 months after presentation. Skeletal muscle biopsy showed mosaic immunostaining for dystrophin. DNA studies showed somatic mosaicism for a nonsense mutation in the dystrophin gene (Arg2905X). The frequency of normal versus mutant genes were determined in blood/DNA (50:50), muscle/DNA (80:20) and muscle/mRNA (90:10). These data are consistent with genetic normalization processes that may biochemically rescue skeletal muscle in male somatic mosaic patients mitigating muscle symptoms (gradual loss of dystrophin-negative skeletal muscle tissue replaced by dystrophin-positive stem cells). To our knowledge, this is only the second reported case of a clinically ascertained patient showing somatic mosaicism for Duchenne muscular dystrophy (DMD). We hypothesize that many somatic mosaic males for DMD exist, yet they are not detected clinically due to genetic normalization. Somatic mosaicism for DMD should be considered in acute heart failure with dilated cardiomyopathy, as genetic normalization in heart is unlikely to occur. Wiley Subscription Services, Inc., A Wiley Company 2009-07 2009-06-15 /pmc/articles/PMC2729699/ /pubmed/19530190 http://dx.doi.org/10.1002/ajmg.a.32891 Text en Copyright © 2009 Wiley-Liss, Inc., A Wiley Company http://creativecommons.org/licenses/by/2.5/ Re-use of this article is permitted in accordance with the Creative Commons Deed, Attribution 2.5, which does not permit commercial exploitation.
spellingShingle Clinical Report
Kesari, Akanchha
Neel, Robert
Wagoner, Lynne
Harmon, Brennan
Spurney, Christopher
Hoffman, Eric P
Somatic mosaicism for Duchenne dystrophy: Evidence for genetic normalization mitigating muscle symptoms
title Somatic mosaicism for Duchenne dystrophy: Evidence for genetic normalization mitigating muscle symptoms
title_full Somatic mosaicism for Duchenne dystrophy: Evidence for genetic normalization mitigating muscle symptoms
title_fullStr Somatic mosaicism for Duchenne dystrophy: Evidence for genetic normalization mitigating muscle symptoms
title_full_unstemmed Somatic mosaicism for Duchenne dystrophy: Evidence for genetic normalization mitigating muscle symptoms
title_short Somatic mosaicism for Duchenne dystrophy: Evidence for genetic normalization mitigating muscle symptoms
title_sort somatic mosaicism for duchenne dystrophy: evidence for genetic normalization mitigating muscle symptoms
topic Clinical Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2729699/
https://www.ncbi.nlm.nih.gov/pubmed/19530190
http://dx.doi.org/10.1002/ajmg.a.32891
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