Literature DB >> 7143387

Clinical evidence for heterogeneity in myotonic dystrophy.

S Bundey.   

Abstract

In a study of 35 index patients who developed myotonic dystrophy between birth and 30 years (neonatal cases aware excluded), 30 could be categorised into two clinical types. The 13 type 1 patients had a more severe limb weakness, of patchy distribution, associated with proportional facial weakness. The 17 type 2 patients had a milder and more diffuse limb weakness; their facial weakness, however, was very pronounced and preceded the limb weakness by several years. All but one of the 25 affected relatives who were examined belonged to the same category as their index relative, providing evidence that the cause of the clinical heterogeneity was genetic. Subsequent observations showed that mental retardation, male infertility, and neonatally affected offspring were commoner in type 2 patients. Congenital myotonic dystrophy could occur among the offspring of either affected males or affected females, but neonatal symptoms were confined to the offspring of affected women. The overall risk for having neonatally affected offspring for this prospective study of young adult patients was 7 in 38, and for the offspring of affected females 7 in 27. The risk for having a surviving child whose mental or physical handicap or both required special schooling was 1 in 12 for males and 4 in 27 for females.

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Year:  1982        PMID: 7143387      PMCID: PMC1048918          DOI: 10.1136/jmg.19.5.341

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  8 in total

1.  Congenital myotonic dystrophy in Britain. II. Genetic basis.

Authors:  P S Harper
Journal:  Arch Dis Child       Date:  1975-07       Impact factor: 3.791

2.  Early-onset dystrophia myotonica. Evidence supporting a maternal environmental factor.

Authors:  P S Harper; P R Dyken
Journal:  Lancet       Date:  1972-07-08       Impact factor: 79.321

3.  Congenital dystrophia myotonica.

Authors:  P R Dyken; P S Harper
Journal:  Neurology       Date:  1973-05       Impact factor: 9.910

4.  Genetic heterogeneity for dystrophia myotonica.

Authors:  S Bundey; C O Carter
Journal:  J Med Genet       Date:  1972-09       Impact factor: 6.318

5.  Early recognition of heterozygotes for the gene for dystrophia myotonica.

Authors:  S Bundey; C O Carter; J F Soothill
Journal:  J Neurol Neurosurg Psychiatry       Date:  1970-06       Impact factor: 10.154

6.  Congenital myotonic dystrophy in Britain. I. Clinical aspects.

Authors:  P S Harper
Journal:  Arch Dis Child       Date:  1975-07       Impact factor: 3.791

7.  Neonatal form of dystrophia myotonica. Five cases in preterm babies and a review of earlier reports.

Authors:  R G Pearse; C J Höweler
Journal:  Arch Dis Child       Date:  1979-05       Impact factor: 3.791

8.  Myotonia dystrophica: unusual features in a Labrador family.

Authors:  D Webb; A Mathews; M Harris; I Muir; J Hostetter; W Marshall; L Salimonu; J Gray; J Faulkner; G Johnson
Journal:  Can Med Assoc J       Date:  1978-03-04       Impact factor: 8.262

  8 in total
  4 in total

1.  Genetic risks for children of women with myotonic dystrophy.

Authors:  M C Koch; T Grimm; H G Harley; P S Harper
Journal:  Am J Hum Genet       Date:  1991-06       Impact factor: 11.025

2.  Neuropsychological profile in myotonic dystrophy.

Authors:  B Censori; M Danni; M Del Pesce; L Provinciali
Journal:  J Neurol       Date:  1990-07       Impact factor: 4.849

3.  Risk estimates for neonatal myotonic dystrophy.

Authors:  A Glånz; F C Fråser
Journal:  J Med Genet       Date:  1984-06       Impact factor: 6.318

4.  Cognitive and personality function in myotonic muscular dystrophy.

Authors:  T D Bird; C Follett; E Griep
Journal:  J Neurol Neurosurg Psychiatry       Date:  1983-11       Impact factor: 10.154

  4 in total

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