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The TREAT-NMD DMD Global Database: analysis of more than 7,000 Duchenne muscular dystrophy mutations

Authors :
Guergueltcheva, Velina
Chamova, Teodora
Bellgard, Matthew I
Vojinovic, Dina
Straub, Volker
Bloetzer, Clemens
Rasic, Vedrana Milic
Kostera-Pruszczyk, Anna
Dai, Yi
Koeks, Zaïda
El Sherif, Rasha
Neagu, Elena
Dawkins, Hugh
Lamont, Leanne
Campbell, Craig
Walter, Maggie C
Zimowski, Janusz
Santos, Rosário
Korngut, Lawrence
Artemieva, Svetlana
Barišić, Nina
Karaduman, A Ayşe
Kirschner, Jan
Jeannet, Pierre-Yves
Karcagi, Veronika
Roy, Anna J
Peay, Holly L
Lahdetie, Jaana
Van Den Bergen, Janneke C
Díaz-Manera, Jordi
Bayat, Farhad
Kekou, Kyriaki
Chan, Sophelia
Kimura, En
Topaloğlu, Haluk
Joncourt, Franziska
Gallardo, Eduard
Salgado, David
Monges, Soledad
Kosma, Konstantina
Posada, Manuel
Rodrigues, Miriam
Stringer, Angela
Foncuberta, Maria E
Béroud, Christophe
Martin, Ann S
Bushby, Kate
Buccella, Filippo
Aartsma-Rus, Annemieke
Wang, Jen
Schreiber-Katz, Olivia
Roxburgh, Richard
Verschuuren, Jan
Lusakowska, Anna
Garami, Marta
Bladen, Catherine L
Flanigan, Kevin M
Brabec, Petr
Viswanathan, Venkatarman
Shatillo, Andriy V
Lochmüller, Hanns
Publisher :
Wiley-Blackwell

Abstract

Analyzing the type and frequency of patient-specific mutations that give rise to Duchenne muscular dystrophy (DMD) is an invaluable tool for diagnostics, basic scientific research, trial planning, and improved clinical care. Locus-specific databases allow for the collection, organization, storage, and analysis of genetic variants of disease. Here, we describe the development and analysis of the TREAT-NMD DMD Global database (http://umd.be/TREAT_DMD/). We analyzed genetic data for 7,149 DMD mutations held within the database. A total of 5,682 large mutations were observed (80% of total mutations), of which 4,894 (86%) were deletions (1 exon or larger) and 784 (14%) were duplications (1 exon or larger). There were 1,445 small mutations (smaller than 1 exon, 20% of all mutations), of which 358 (25%) were small deletions and 132 (9%) small insertions and 199 (14%) affected the splice sites. Point mutations totalled 756 (52% of small mutations) with 726 (50%) nonsense mutations and 30 (2%) missense mutations. Finally, 22 (0.3%) mid-intronic mutations were observed. In addition, mutations were identified within the database that would potentially benefit from novel genetic therapies for DMD including stop codon read-through therapies (10% of total mutations) and exon skipping therapy (80% of deletions and 55% of total mutations).

Details

Language :
English
Database :
OpenAIRE
Accession number :
edsair.doi...........4e07f74b617c9c10d83be7b9fdfe1f0e