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Deletion of the Williams Beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy
Among 1339 unrelated cases accrued by the Italian National Registry for facioscapulohumeral muscular dystrophy (FSHD), we found three unrelated cases who presented signs of Williams-Beuren Syndrome (WBS) in early childhood and later developed FSHD. All three cases carry the molecular defects associa...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Saunders
2020
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7427329/ https://www.ncbi.nlm.nih.gov/pubmed/32553920 http://dx.doi.org/10.1016/j.ejpn.2020.05.006 |
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author | Rodolico, Carmelo Politano, Luisa Portaro, Simona Murru, Stefania Boccone, Loredana Sera, Francesco Passamano, Luigia Brizzi, Teresa Tupler, Rossella |
author_facet | Rodolico, Carmelo Politano, Luisa Portaro, Simona Murru, Stefania Boccone, Loredana Sera, Francesco Passamano, Luigia Brizzi, Teresa Tupler, Rossella |
author_sort | Rodolico, Carmelo |
collection | PubMed |
description | Among 1339 unrelated cases accrued by the Italian National Registry for facioscapulohumeral muscular dystrophy (FSHD), we found three unrelated cases who presented signs of Williams-Beuren Syndrome (WBS) in early childhood and later developed FSHD. All three cases carry the molecular defects associated with the two disorders. The rarity of WBS and FSHD, 1 in 7500 and 1 in 20,000 respectively, makes a random association of the two diseases unlikely. These cases open novel and unexpected interpretation of genetic findings. The nonrandom association of both FSHD and WBS points at a gene co-expression network providing hints for the identification of modules and functionally enriched pathways in the two conditions. |
format | Online Article Text |
id | pubmed-7427329 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | Saunders |
record_format | MEDLINE/PubMed |
spelling | pubmed-74273292020-08-16 Deletion of the Williams Beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy Rodolico, Carmelo Politano, Luisa Portaro, Simona Murru, Stefania Boccone, Loredana Sera, Francesco Passamano, Luigia Brizzi, Teresa Tupler, Rossella Eur J Paediatr Neurol Article Among 1339 unrelated cases accrued by the Italian National Registry for facioscapulohumeral muscular dystrophy (FSHD), we found three unrelated cases who presented signs of Williams-Beuren Syndrome (WBS) in early childhood and later developed FSHD. All three cases carry the molecular defects associated with the two disorders. The rarity of WBS and FSHD, 1 in 7500 and 1 in 20,000 respectively, makes a random association of the two diseases unlikely. These cases open novel and unexpected interpretation of genetic findings. The nonrandom association of both FSHD and WBS points at a gene co-expression network providing hints for the identification of modules and functionally enriched pathways in the two conditions. Saunders 2020-07 /pmc/articles/PMC7427329/ /pubmed/32553920 http://dx.doi.org/10.1016/j.ejpn.2020.05.006 Text en © 2020 The Authors 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 Rodolico, Carmelo Politano, Luisa Portaro, Simona Murru, Stefania Boccone, Loredana Sera, Francesco Passamano, Luigia Brizzi, Teresa Tupler, Rossella Deletion of the Williams Beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy |
title | Deletion of the Williams Beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy |
title_full | Deletion of the Williams Beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy |
title_fullStr | Deletion of the Williams Beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy |
title_full_unstemmed | Deletion of the Williams Beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy |
title_short | Deletion of the Williams Beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy |
title_sort | deletion of the williams beuren syndrome critical region unmasks facioscapulohumeral muscular dystrophy |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7427329/ https://www.ncbi.nlm.nih.gov/pubmed/32553920 http://dx.doi.org/10.1016/j.ejpn.2020.05.006 |
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