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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...

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Autores principales: Rodolico, Carmelo, Politano, Luisa, Portaro, Simona, Murru, Stefania, Boccone, Loredana, Sera, Francesco, Passamano, Luigia, Brizzi, Teresa, Tupler, Rossella
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Saunders 2020
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.
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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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