Literature DB >> 176660

Androgen insensitivity in man: evidence for genetic heterogeneity.

J A Amrhein, W J Meyer, H W Jones, C J Migeon.   

Abstract

We have studied ten phenotypically similar patients with complete androgen insensitivity. All of the patients tested had significantly elevated serum luteinizing hormone and plasma androgens within or above the normal adult male range. On the basis of specific dihydrotestosterone binding by skin fibroblasts, we identified two subgroups. Six patients from five different families had undetectable dihydrotestosterone binding, while four patients from two families had normal binding activity. Our results indicate that within the clinical syndrome of androgen insensitivity there are at least two distinct genetic variants. These variants may result from allelic mutations of the same X-linked gene specifying the dihydrotestosterone receptor or, alternatively, from mutations of separate genes both being essential for androgen action in responsive cells.

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Year:  1976        PMID: 176660      PMCID: PMC336025          DOI: 10.1073/pnas.73.3.891

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  18 in total

1.  FURTHER OBSERVATIONS ON THE SYNDROME, "TESTICULAR FEMINIZATION".

Authors:  J M MORRIS; V B MAHESH
Journal:  Am J Obstet Gynecol       Date:  1963-11-15       Impact factor: 8.661

2.  A study of the conditions and mechanism of the diphenylamine reaction for the colorimetric estimation of deoxyribonucleic acid.

Authors:  K BURTON
Journal:  Biochem J       Date:  1956-02       Impact factor: 3.857

3.  The syndrome of testicular feminization in male pseudohermaphrodites.

Authors:  J M MORRIS
Journal:  Am J Obstet Gynecol       Date:  1953-06       Impact factor: 8.661

4.  Locus on human X chromosome for dihydrotestosterone receptor and androgen insensitivity.

Authors:  W J Meyer; B R Migeon; C J Migeon
Journal:  Proc Natl Acad Sci U S A       Date:  1975-04       Impact factor: 11.205

5.  Androgen receptor in human skin fibroblasts. Characterization of a specific 17beta-hydroxy-5alpha-androstan-3-one-protein complex in cell sonicates and nuclei.

Authors:  B S Keenan; W J Meyer; A J Hadjian; C J Migeon
Journal:  Steroids       Date:  1975-04       Impact factor: 2.668

6.  Protein measurement with the Folin phenol reagent.

Authors:  O H LOWRY; N J ROSEBROUGH; A L FARR; R J RANDALL
Journal:  J Biol Chem       Date:  1951-11       Impact factor: 5.157

7.  Cytosol androgen receptor from kidney of normal and testicular feminized (Tfm) mice.

Authors:  B Attardi; S Ono
Journal:  Cell       Date:  1974-08       Impact factor: 41.582

8.  Characterization of a hormone receptor defect in the androgen-insensitivity mutant.

Authors:  U Gehring; G M Tomkins
Journal:  Cell       Date:  1974-09       Impact factor: 41.582

9.  Androgen metabolism and mechanism of action in male pseudohermaphroditism: a study of testicular feminization.

Authors:  C W Bardin; L P Bullock; R J Sherins; I Mowszowicz; W R Blackburn
Journal:  Recent Prog Horm Res       Date:  1973

10.  Studies of androgens in the syndrome of male pseudohermaphroditism with testicular feminization.

Authors:  M A Rivarola; J M Saez; W J Meyer; F M Kenny; C J Migeon
Journal:  J Clin Endocrinol Metab       Date:  1967-03       Impact factor: 5.958

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  23 in total

1.  Sequence of the intron/exon junctions of the coding region of the human androgen receptor gene and identification of a point mutation in a family with complete androgen insensitivity.

Authors:  D B Lubahn; T R Brown; J A Simental; H N Higgs; C J Migeon; E M Wilson; F S French
Journal:  Proc Natl Acad Sci U S A       Date:  1989-12       Impact factor: 11.205

Review 2.  The incomplete male.

Authors:  M O Savage; D B Grant
Journal:  Arch Dis Child       Date:  1978-09       Impact factor: 3.791

Review 3.  Molecular genetics of human androgen insensitivity.

Authors:  T R Brown; P A Scherer; Y T Chang; C J Migeon; P Ghirri; K Murono; Z Zhou
Journal:  Eur J Pediatr       Date:  1993       Impact factor: 3.183

4.  Human minimal androgen insensitivity with normal dihydrotestosterone-binding capacity in cultured genital skin fibroblasts: evidence for an androgen-selective qualitative abnormality of the receptor.

Authors:  L Pinsky; M Kaufman; D W Killinger; B Burko; D Shatz; R Volpé
Journal:  Am J Hum Genet       Date:  1984-09       Impact factor: 11.025

5.  Differences in proteins synthesized by fibroblasts from normal individuals and patients with complete testicular feminization.

Authors:  G P Risbridger; B A Khalid; G L Warne; J W Funder
Journal:  J Clin Invest       Date:  1982-01       Impact factor: 14.808

6.  Comments on some genetic abnormalities of sex determination and sex differentiation in Homo sapiens.

Authors:  J M Opitz
Journal:  Eur J Pediatr       Date:  1980-03       Impact factor: 3.183

7.  Testicular feminisation syndrome presenting in the newborn.

Authors:  M Sheridan-Pereira; N O'Brien
Journal:  Arch Dis Child       Date:  1983-05       Impact factor: 3.791

8.  Intersex problems in the neonate.

Authors:  R K Danish
Journal:  Indian J Pediatr       Date:  1982 Jul-Aug       Impact factor: 1.967

9.  Studies on steroid hormone receptors (5alpha-dihidrotesterone, estradiol, and dexamethasone) in cultured human fibroblasts and amniotic fluid cells.

Authors:  T Bauknecht
Journal:  Hum Genet       Date:  1977-12-23       Impact factor: 4.132

10.  Deletion of the steroid-binding domain of the human androgen receptor gene in one family with complete androgen insensitivity syndrome: evidence for further genetic heterogeneity in this syndrome.

Authors:  T R Brown; D B Lubahn; E M Wilson; D R Joseph; F S French; C J Migeon
Journal:  Proc Natl Acad Sci U S A       Date:  1988-11       Impact factor: 11.205

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