Literature DB >> 2821406

Preferential deletion of exons in Duchenne and Becker muscular dystrophies.

S M Forrest1, G S Cross, A Speer, D Gardner-Medwin, J Burn, K E Davies.   

Abstract

Duchenne and Becker muscular dystrophy (DMD and BMD) genes are located in Xp21 on the short arm of the X chromosome. DMD patients display a much more severe clinical course than BMD patients, and yet about 10% of cases of each have been reported to have deletions for parts of the gene. Using a complementary DNA subclone of the DMD gene we have screened 66 DMD and BMD patients who had not previously shown deletions with the probes then available. Fifteen patients have a deletion of this part of the gene, indicating a higher deletion frequency in this region (22%). Exons were deleted in five severely affected DMD patients and in ten BMD patients. Significantly, most of these deletions begin in the same region of the cDNA, which implies that there is a common mechanism for the generation of many of these mutations. An apparently identical deletion in one family gave classical BMD in two brothers (presenting in their teens) and only very mild muscle weakness in their 86-year-old great-great-uncle. Taking these data together with data using the probes previously published, we are able to detect deletions directly in 40% of our families requiring antenatal diagnosis or carrier detection.

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Year:  1987        PMID: 2821406     DOI: 10.1038/329638a0

Source DB:  PubMed          Journal:  Nature        ISSN: 0028-0836            Impact factor:   49.962


  60 in total

1.  Quantitative Southern blot analysis in the dystrophin gene of Japanese patients with Duchenne or Becker muscular dystrophy: a high frequency of duplications.

Authors:  Y Hiraishi; S Kato; T Ishihara; T Takano
Journal:  J Med Genet       Date:  1992-12       Impact factor: 6.318

2.  Gene deletions causing human genetic disease: mechanisms of mutagenesis and the role of the local DNA sequence environment.

Authors:  M Krawczak; D N Cooper
Journal:  Hum Genet       Date:  1991-03       Impact factor: 4.132

3.  Germinal mosaicism increases the recurrence risk for 'new' Duchenne muscular dystrophy mutations.

Authors:  E Bakker; H Veenema; J T Den Dunnen; C van Broeckhoven; P M Grootscholten; E J Bonten; G J van Ommen; P L Pearson
Journal:  J Med Genet       Date:  1989-09       Impact factor: 6.318

4.  Duchenne muscular dystrophy in Wales: impact of DNA linkage analysis and cDNA deletion screening.

Authors:  A M Norman; M Upadhyaya; N S Thomas; K Roberts; P S Harper
Journal:  J Med Genet       Date:  1989-09       Impact factor: 6.318

5.  A normal male with an inherited deletion of one exon within the DMD gene.

Authors:  M Nordenskjöld; L Nicholson; L Edström; M Anvret; M Eiserman; C Slater; L Stolpe
Journal:  Hum Genet       Date:  1990-01       Impact factor: 4.132

Review 6.  Improved diagnosis of Duchenne/Becker muscular dystrophy.

Authors:  A H Beggs; L M Kunkel
Journal:  J Clin Invest       Date:  1990-03       Impact factor: 14.808

7.  Normal human genomic restriction-fragment patterns and polymorphisms revealed by hybridization with the entire dystrophin cDNA.

Authors:  B T Darras; U Francke
Journal:  Am J Hum Genet       Date:  1988-11       Impact factor: 11.025

8.  Use of epitope libraries to identify exon-specific monoclonal antibodies for characterization of altered dystrophins in muscular dystrophy.

Authors:  T M Nguyen; G E Morris
Journal:  Am J Hum Genet       Date:  1993-06       Impact factor: 11.025

9.  Non-isotopic analysis of single strand conformation polymorphism (SSCP) in the exon 13 region of the human dystrophin gene.

Authors:  U Lenk; R Hanke; U Kräft; K Grade; I Grunewald; A Speer
Journal:  J Med Genet       Date:  1993-11       Impact factor: 6.318

10.  Multiple mutation in an extended Duchenne muscular dystrophy family.

Authors:  A Miciak; A Keen; D Jadayel; S Bundey
Journal:  J Med Genet       Date:  1992-02       Impact factor: 6.318

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