Literature DB >> 8841194

Autosomal recessive limb-girdle muscular dystrophy, LGMD2F, is caused by a mutation in the delta-sarcoglycan gene.

V Nigro1, E de Sá Moreira, G Piluso, M Vainzof, A Belsito, L Politano, A A Puca, M R Passos-Bueno, M Zatz.   

Abstract

Limb-girdle muscular dystrophies (LGMD) are a heterogeneous group of inherited neuromuscular disorders characterized by proximal muscular weakness of the pelvic and shoulder girdles and a variable progression with symptoms, ranging from very severe to mild. One autosomal dominant (LGMD1A, at chromosome 5q22.3-31.3) (ref. 3) and five autosomal recessive (AR) loci responsible for this phenotype have been identified: LGMD2A at 15q (ref. 4); LGMD2B at 2p (ref. 5), LGMD2C at 13q (ref. 6), LGMD2D at 17q (ref. 7) and LGMD2E at 4q (refs 8,9). In the muscle membrane, dystrophin associates with several proteins and glycoproteins organized in two main subcomplexes: the dystroglycan (DG) and sarcoglycan (SG) complexes. The genes for LGMD2C, LGMD2D and LGMD2E code for proteins of the SG complex. We recently mapped a sixth AR form of LGMD, LGMD2F, to chromosome 5q33-34 in two Brazilian families. In the same chromosomal interval we also mapped the delta SG gene, encoding a novel 35-kD component of the sarcoglycan (SG) complex. We now show that a homozygous mutation in the delta SG gene (a single nucleotide deletion that alters its reading frame) is the cause of LGMD2F.

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Year:  1996        PMID: 8841194     DOI: 10.1038/ng1096-195

Source DB:  PubMed          Journal:  Nat Genet        ISSN: 1061-4036            Impact factor:   38.330


  101 in total

1.  Worsening of cardiomyopathy using deflazacort in an animal model rescued by gene therapy.

Authors:  Ida Luisa Rotundo; Stefania Faraso; Elvira De Leonibus; Gerardo Nigro; Carmen Vitiello; Alessio Lancioni; Daniele Di Napoli; Sigismondo Castaldo; Vincenzo Russo; Fabio Russo; Giulio Piluso; Alberto Auricchio; Vincenzo Nigro
Journal:  PLoS One       Date:  2011-09-09       Impact factor: 3.240

Review 2.  Animal models of muscular dystrophy.

Authors:  Rainer Ng; Glen B Banks; John K Hall; Lindsey A Muir; Julian N Ramos; Jacqueline Wicki; Guy L Odom; Patryk Konieczny; Jane Seto; Joel R Chamberlain; Jeffrey S Chamberlain
Journal:  Prog Mol Biol Transl Sci       Date:  2012       Impact factor: 3.622

3.  A delta-sarcoglycan gene polymorphism as a risk factor for hypertrophic cardiomyopathy.

Authors:  Rosa M Ordoñez-Razo; Martín H Garrido-Garduño; Ramón A Pérez-Martínez; Victor M Ruiz; Esteban Herrera-Tepatlán; Maricela Rodríguez-Cruz; Ana L Jiménez-Vaca; Fernando Minauro-Sanmiguel; Fabio A Salamanca-Gómez
Journal:  Genet Test Mol Biomarkers       Date:  2012-04-23

4.  2004 William Allan Award address. Cloning of the DMD gene.

Authors:  Louis M Kunkel
Journal:  Am J Hum Genet       Date:  2005-02       Impact factor: 11.025

5.  Alpha7beta1 integrin does not alleviate disease in a mouse model of limb girdle muscular dystrophy type 2F.

Authors:  Derek J Milner; Stephen J Kaufman
Journal:  Am J Pathol       Date:  2007-02       Impact factor: 4.307

6.  Disease rescue and increased lifespan in a model of cardiomyopathy and muscular dystrophy by combined AAV treatments.

Authors:  Carmen Vitiello; Stefania Faraso; Nicolina Cristina Sorrentino; Giovanni Di Salvo; Edoardo Nusco; Gerardo Nigro; Luisa Cutillo; Raffaele Calabrò; Alberto Auricchio; Vincenzo Nigro
Journal:  PLoS One       Date:  2009-03-31       Impact factor: 3.240

7.  Dusp6 is a genetic modifier of growth through enhanced ERK activity.

Authors:  Andy H Vo; Kayleigh A Swaggart; Anna Woo; Quan Q Gao; Alexis R Demonbreun; Katherine S Fallon; Mattia Quattrocelli; Michele Hadhazy; Patrick G T Page; Zugen Chen; Ascia Eskin; Kevin Squire; Stanley F Nelson; Elizabeth M McNally
Journal:  Hum Mol Genet       Date:  2019-01-15       Impact factor: 6.150

8.  Thin, a Trim32 ortholog, is essential for myofibril stability and is required for the integrity of the costamere in Drosophila.

Authors:  Elisa M LaBeau-DiMenna; Kathleen A Clark; Kenneth D Bauman; Daniel S Parker; Richard M Cripps; Erika R Geisbrecht
Journal:  Proc Natl Acad Sci U S A       Date:  2012-10-15       Impact factor: 11.205

Review 9.  [Limb girdle muscular dystrophies].

Authors:  J Finsterer
Journal:  Nervenarzt       Date:  2004-12       Impact factor: 1.214

10.  Overexpression of Galgt2 reduces dystrophic pathology in the skeletal muscles of alpha sarcoglycan-deficient mice.

Authors:  Rui Xu; Sarah DeVries; Marybeth Camboni; Paul T Martin
Journal:  Am J Pathol       Date:  2009-06-04       Impact factor: 4.307

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