Literature DB >> 3285207

Characterization of dystrophin in muscle-biopsy specimens from patients with Duchenne's or Becker's muscular dystrophy.

E P Hoffman1, K H Fischbeck, R H Brown, M Johnson, R Medori, J D Loike, J B Harris, R Waterston, M Brooke, L Specht.   

Abstract

A deficiency of the protein dystrophin has recently been shown to be the probable cause of Duchenne's muscular dystrophy. We sought to determine the relation between the clinical phenotype and the status of dystrophin in muscle-biopsy specimens from 103 patients with various neuromuscular disorders. We found very low levels (less than 3 percent of normal levels) or no dystrophin in the severe Duchenne phenotype (35 of 38 patients), low concentrations of dystrophin in the intermediate (outlier) phenotype (4 of 7), and dystrophin of abnormal molecular weight in the mild Becker phenotype (12 of 18). Normal levels of dystrophin of normal molecular weight were found in nearly all the patients (38 of 40) with 20 other neuromuscular disorders we studied. These data show the clinical consequences of both quantitative alterations (in Duchenne's and intermediate dystrophy) in a single protein. The biochemical assay for dystrophin should prove helpful in delineating myopathies that overlap clinically with Duchenne's and Becker's dystrophies, and it shows promise as an accurate diagnostic tool.

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Year:  1988        PMID: 3285207     DOI: 10.1056/NEJM198805263182104

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  207 in total

1.  "Quadriceps myopathy": a clinical variant form of Becker muscular dystrophy.

Authors:  Y Wada; Y Itoh; T Furukawa; H Tsukagoshi; K Arahata
Journal:  J Neurol       Date:  1990-08       Impact factor: 4.849

2.  Monoclonal antibodies for dystrophin analysis. Epitope mapping and improved binding to SDS-treated muscle sections.

Authors:  T M Nguyen; I B Ginjaar; G J van Ommen; G E Morris
Journal:  Biochem J       Date:  1992-12-01       Impact factor: 3.857

Review 3.  Recent advances in understanding muscular dystrophy.

Authors:  K M Bushby
Journal:  Arch Dis Child       Date:  1992-10       Impact factor: 3.791

Review 4.  The muscular dystrophies.

Authors:  V Dubowitz
Journal:  Postgrad Med J       Date:  1992-07       Impact factor: 2.401

Review 5.  Muscular dystrophies.

Authors:  L Specht
Journal:  Indian J Pediatr       Date:  1990 May-Jun       Impact factor: 1.967

6.  Characterization of patients with glycerol kinase deficiency utilizing cDNA probes for the Duchenne muscular dystrophy locus.

Authors:  J A Towbin; D R Wu; J Chamberlain; P D Larsen; W K Seltzer; E R McCabe
Journal:  Hum Genet       Date:  1989-09       Impact factor: 4.132

7.  Mutation analysis in a population-based cohort of boys with Duchenne or Becker muscular dystrophy.

Authors:  Christopher Cunniff; Jennifer Andrews; F John Meaney; Katherine D Mathews; Dennis Matthews; Emma Ciafaloni; Timothy M Miller; John B Bodensteiner; Lisa A Miller; Katherine A James; Charlotte M Druschel; Paul A Romitti; Shree Pandya
Journal:  J Child Neurol       Date:  2008-12-12       Impact factor: 1.987

8.  Antisense-induced multiexon skipping for Duchenne muscular dystrophy makes more sense.

Authors:  Annemieke Aartsma-Rus; Anneke A M Janson; Wendy E Kaman; Mattie Bremmer-Bout; Gert-Jan B van Ommen; Johan T den Dunnen; Judith C T van Deutekom
Journal:  Am J Hum Genet       Date:  2003-12-16       Impact factor: 11.025

Review 9.  Diagnosis and cell-based therapy for Duchenne muscular dystrophy in humans, mice, and zebrafish.

Authors:  Louis M Kunkel; Estanislao Bachrach; Richard R Bennett; Jeffrey Guyon; Leta Steffen
Journal:  J Hum Genet       Date:  2006-04-01       Impact factor: 3.172

10.  Dystrophin and utrophin "double knockout" dystrophic mice exhibit a spectrum of degenerative musculoskeletal abnormalities.

Authors:  Christian Isaac; Adam Wright; Arvydas Usas; Hongshuai Li; Ying Tang; Xiaodong Mu; Nicholas Greco; Qing Dong; Nam Vo; James Kang; Bing Wang; Johnny Huard
Journal:  J Orthop Res       Date:  2012-10-23       Impact factor: 3.494

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