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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...
Autores principales: | , , , , , |
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Formato: | Texto |
Lenguaje: | English |
Publicado: |
Wiley Subscription Services, Inc., A Wiley Company
2009
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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. |
format | Text |
id | pubmed-2729699 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2009 |
publisher | Wiley Subscription Services, Inc., A Wiley Company |
record_format | MEDLINE/PubMed |
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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