Literature DB >> 2783976

21-hydroxylase deficiency families with HLA identical affected and unaffected sibs.

P J Sinnott1, P A Dyer, D A Price, R Harris, T Strachan.   

Abstract

During our investigations of polymorphisms at, and in the immediate chromosomal vicinity of, the 21-hydroxylase locus in families with 21-hydroxylase deficiency, three families were found to show marked discordance in clinical features of HLA identical subjects. In one family, there is discordance between a boy with the simple virilising form of 21-hydroxylase deficiency and his two younger sisters, who are both HLA identical to their brother, but who have additional salt wasting features. In the other two families, one subject is severely affected and has very high 17-hydroxyprogesterone levels, but has an HLA identical sib who is asymptomatic and shows only slightly raised 17-hydroxyprogesterone levels. In all cases, HLA identity, as indicated by protein polymorphism studies (HLA-A, B, DR, C4A, C4B, and Bf typing), has been verified at the gene organisation level using 21-hydroxylase and complement C4 DNA probes. An HLA-Bw47 bearing haplotype in one of the latter families has not been transmitted to the affected child and appears to carry a normal 21-OHB allele and two genes which specify C4A allotypes.

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Year:  1989        PMID: 2783976      PMCID: PMC1015530          DOI: 10.1136/jmg.26.1.10

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


  31 in total

1.  Detection of specific sequences among DNA fragments separated by gel electrophoresis.

Authors:  E M Southern
Journal:  J Mol Biol       Date:  1975-11-05       Impact factor: 5.469

2.  Analysis of human Y-chromosome-specific reiterated DNA in chromosome variants.

Authors:  L M Kunkel; K D Smith; S H Boyer; D S Borgaonkar; S S Wachtel; O J Miller; W R Breg; H W Jones; J M Rary
Journal:  Proc Natl Acad Sci U S A       Date:  1977-03       Impact factor: 11.205

3.  The study of a French family with two duplicated C4A haplotypes.

Authors:  C M Giles; B Uring-Lambert; W Boksch; M Braun; J Goetz; R Neumann; G Mauff; G Hauptmann
Journal:  Hum Genet       Date:  1987-12       Impact factor: 4.132

4.  "A technique for radiolabeling DNA restriction endonuclease fragments to high specific activity". Addendum.

Authors:  A P Feinberg; B Vogelstein
Journal:  Anal Biochem       Date:  1984-02       Impact factor: 3.365

5.  HLA and congenital adrenal hyperplasia linkage confirmed.

Authors: 
Journal:  Lancet       Date:  1978-04-29       Impact factor: 79.321

6.  Linkage and association between HLA and 21-hydroxylase deficiency.

Authors:  P T Klouda; R Harris; D A Price
Journal:  J Med Genet       Date:  1980-10       Impact factor: 6.318

7.  Varying expression for salt losing in related patients with congenital adrenal hyperplasia.

Authors:  A L Rosenbloom; D W Smith
Journal:  Pediatrics       Date:  1966-08       Impact factor: 7.124

8.  The 21-hydroxylase activity in the glomerulosa and fasciculata of the adrenal cortex in congenital adrenal hyperplasia.

Authors:  U Kuhnle; D Chow; R Rapaport; S Pang; L S Levine; M I New
Journal:  J Clin Endocrinol Metab       Date:  1981-03       Impact factor: 5.958

9.  The structural basis of the multiple forms of human complement component C4.

Authors:  K T Belt; M C Carroll; R R Porter
Journal:  Cell       Date:  1984-04       Impact factor: 41.582

10.  Close genetic linkage between HLA and congenital adrenal hyperplasia (21-hydroxylase deficiency).

Authors:  B Dupont; S E Oberfield; E M Smithwick; T D Lee; L S Levine
Journal:  Lancet       Date:  1977 Dec 24-31       Impact factor: 79.321

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  9 in total

1.  Molecular genetics in the National Health Service in Britain.

Authors:  R Harris; R Elles; D Craufurd; A Dodge; A Ivinson; K Hodgkinson; R Mountford; M Schwartz; T Strachan; A Read
Journal:  J Med Genet       Date:  1989-04       Impact factor: 6.318

2.  Molecular characterisation of C4 null alleles found in Felty's syndrome.

Authors:  M C Hillarby; T Strachan; D M Grennan
Journal:  Ann Rheum Dis       Date:  1990-10       Impact factor: 19.103

3.  Genesis by meiotic unequal crossover of a de novo deletion that contributes to steroid 21-hydroxylase deficiency.

Authors:  P Sinnott; S Collier; C Costigan; P A Dyer; R Harris; T Strachan
Journal:  Proc Natl Acad Sci U S A       Date:  1990-03       Impact factor: 11.205

Review 4.  The molecular biology, biochemistry, and physiology of human steroidogenesis and its disorders.

Authors:  Walter L Miller; Richard J Auchus
Journal:  Endocr Rev       Date:  2010-11-04       Impact factor: 19.871

5.  CYP21/C4 gene organisation in Italian 21-hydroxylase deficiency families.

Authors:  P J Sinnott; C Livieri; M Sampietro; M Marconi; R Harris; F Severi; T Strachan
Journal:  Hum Genet       Date:  1992-03       Impact factor: 4.132

6.  Mutation screening in British 21-hydroxylase deficiency families and development of novel microsatellite based approaches to prenatal diagnosis.

Authors:  M Lako; S Ramsden; R D Campbell; T Strachan
Journal:  J Med Genet       Date:  1999-02       Impact factor: 6.318

7.  Extended MHC haplotypes and CYP21/C4 gene organisation in Irish 21-hydroxylase deficiency families.

Authors:  P J Sinnott; C Costigan; P A Dyer; R Harris; T Strachan
Journal:  Hum Genet       Date:  1991-07       Impact factor: 4.132

8.  Molecular analysis of patient and carrier genes with congenital steroid 21-hydroxylase deficiency by using polymerase chain reaction and single strand conformation polymorphism.

Authors:  T Tajima; K Fujieda; K Nakayama; Y Fujii-Kuriyama
Journal:  J Clin Invest       Date:  1993-11       Impact factor: 14.808

9.  Pulsed field gel electrophoresis identifies a high degree of variability in the number of tandem 21-hydroxylase and complement C4 gene repeats in 21-hydroxylase deficiency haplotypes.

Authors:  S Collier; P J Sinnott; P A Dyer; D A Price; R Harris; T Strachan
Journal:  EMBO J       Date:  1989-05       Impact factor: 11.598

  9 in total

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