Literature DB >> 8490621

Is the maintainance of the C-terminus domain of dystrophin enough to ensure a milder Becker muscular dystrophy phenotype?

M Vainzof1, R I Takata, M R Passos-Bueno, R C Pavanello, M Zatz.   

Abstract

The severe Duchenne muscular dystrophy (DMD) and the more benign Becker type (BMD) are allelic conditions, controlled by a defective gene at Xp21, caused by the absence (DMD) or a defect in quantity or quality (BMD) of the protein dystrophin. It has been suggested that the C-terminus domain of dystrophin is fundamental to ensure the proper protein sub-cellular localization and function. We wish to report our dystrophin findings in 4 among 142 DMD patients studied for DNA deletions and dystrophin analysis. Although they have a severe clinical course, a positive dystrophin immunofluorescence pattern was seen using C-terminal antibody, and a dystrophin band of reduced molecular weight (corresponding to their DNA deletions), but which maintained the C-terminus was seen through Western blot (WB). Based on these findings, we suggest that in order to partially maintain its function, resulting in a milder phenotype, dystrophin may carry large internal deletions but in addition to the C-terminus, the region encompassing both the N-terminus and the proximal region of the rod domain cannot be absent. Therefore, the prognosis of a Becker phenotype in a young patient should be done with caution if based only on the presence or not of dystrophin.

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Year:  1993        PMID: 8490621     DOI: 10.1093/hmg/2.1.39

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  11 in total

Review 1.  The Dystrophin Complex: Structure, Function, and Implications for Therapy.

Authors:  Quan Q Gao; Elizabeth M McNally
Journal:  Compr Physiol       Date:  2015-07-01       Impact factor: 9.090

2.  Dystrophin genotype-cardiac phenotype correlations in Duchenne and Becker muscular dystrophies using cardiac magnetic resonance imaging.

Authors:  Animesh Tandon; John L Jefferies; Chet R Villa; Kan N Hor; Brenda L Wong; Stephanie M Ware; Zhiqian Gao; Jeffrey A Towbin; Wojciech Mazur; Robert J Fleck; Joshua J Sticka; D Woodrow Benson; Michael D Taylor
Journal:  Am J Cardiol       Date:  2015-01-15       Impact factor: 2.778

3.  Mild deficiency of dystrophin-associated proteins in Becker muscular dystrophy patients having in-frame deletions in the rod domain of dystrophin.

Authors:  K Matsumura; I Nonaka; F M Tomé; K Arahata; H Collin; F Leturcq; D Récan; J C Kaplan; M Fardeau; K P Campbell
Journal:  Am J Hum Genet       Date:  1993-08       Impact factor: 11.025

4.  A novel point mutation (G-1 to T) in a 5' splice donor site of intron 13 of the dystrophin gene results in exon skipping and is responsible for Becker muscular dystrophy.

Authors:  Y Hagiwara; H Nishio; Y Kitoh; Y Takeshima; N Narita; H Wada; M Yokoyama; H Nakamura; M Matsuo
Journal:  Am J Hum Genet       Date:  1994-01       Impact factor: 11.025

5.  Deletions in the 5' region of dystrophin and resulting phenotypes.

Authors:  F Muntoni; P Gobbi; C Sewry; T Sherratt; J Taylor; S K Sandhu; S Abbs; R Roberts; S V Hodgson; M Bobrow
Journal:  J Med Genet       Date:  1994-11       Impact factor: 6.318

6.  Myotubes from transgenic mdx mice expressing full-length dystrophin show normal calcium regulation.

Authors:  W F Denetclaw; F W Hopf; G A Cox; J S Chamberlain; R A Steinhardt
Journal:  Mol Biol Cell       Date:  1994-10       Impact factor: 4.138

7.  Assessing pathogenicity for novel mutation/sequence variants: the value of healthy older individuals.

Authors:  Mayana Zatz; Rita de Cassia M Pavanello; Naila Cristina V Lourenço; Antonia Cerqueira; Monize Lazar; Mariz Vainzof
Journal:  Neuromolecular Med       Date:  2012-06-16       Impact factor: 3.843

Review 8.  Dystrophic Cardiomyopathy: Complex Pathobiological Processes to Generate Clinical Phenotype.

Authors:  Takeshi Tsuda; Kristi K Fitzgerald
Journal:  J Cardiovasc Dev Dis       Date:  2017-09-08

9.  Immunohistochemical analysis of dystrophin-associated proteins in Becker/Duchenne muscular dystrophy with huge in-frame deletions in the NH2-terminal and rod domains of dystrophin.

Authors:  K Matsumura; A H Burghes; M Mora; F M Tomé; L Morandi; F Cornello; F Leturcq; M Jeanpierre; J C Kaplan; P Reinert
Journal:  J Clin Invest       Date:  1994-01       Impact factor: 14.808

10.  Neuromuscular disorders: genes, genetic counseling and therapeutic trials.

Authors:  Mayana Zatz; Maria Rita Passos-Bueno; Mariz Vainzof
Journal:  Genet Mol Biol       Date:  2016 Jul-Sep       Impact factor: 1.771

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