Literature DB >> 876326

Partial androgen insensitivity: the Reifenstein syndrome revisited.

J A Amrhein, G J Klingensmith, P C Walsh, V A McKusick, C J Migeon.   

Abstract

We investigated eight patients with the Reifenstein syndrome to define the hormonal basis for this condition. The patients had normal or elevated concentrations of plasma androgens, normal production rates of testosterone and dihydrotestosterone, elevated serum levels of luteinizing hormone and normal 5alpha-reductase activity in skin fibroblasts. These findings indicate that the syndrome results from defective androgen action rather than from decreased androgen synthesis. The term "partial androgen insensitivity syndrome" describes this condition more accurately than a term based on clinical phenotype. Dihydrotestosterone binding studies in skin fibroblasts demonstrated two genetic variants similar to those reported in complete androgen insensitivity syndrome. One patient had a partial deficiency of cytoplasmic dihydrotestosterone binding, and four others had normal binding activity. The cause of the androgen insensitivity in the last four cases is unknown. Treatment with testosterone suppressed serum luteinizing hormone levels and promoted mild virilizing effects.

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Year:  1977        PMID: 876326     DOI: 10.1056/NEJM197708182970703

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


  13 in total

Review 1.  The incomplete male.

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

Review 2.  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

3.  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

4.  Receptor dysfunctions in human disease.

Authors:  L H Block; W Tenschert; R Locher; W Siegenthaler; W Vetter
Journal:  Klin Wochenschr       Date:  1981-04-15

5.  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

6.  Intersex problems in the neonate.

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

7.  Primary defect of insulin receptors in skin fibroblasts cultured from an infant with leprechaunism and insulin resistance.

Authors:  E E Schilling; M M Rechler; C Grunfeld; A M Rosenberg
Journal:  Proc Natl Acad Sci U S A       Date:  1979-11       Impact factor: 11.205

Review 8.  The androgen resistance syndromes: clinical and biochemical aspects.

Authors:  H U Schweikert
Journal:  Eur J Pediatr       Date:  1993       Impact factor: 3.183

Review 9.  Cultured human skin fibroblasts: a model for the study of androgen action.

Authors:  T R Brown; C J Migeon
Journal:  Mol Cell Biochem       Date:  1981-04-13       Impact factor: 3.396

10.  Familial incomplete male pseudohermaphroditism associated with impaired nuclear androgen retention. Studies in cultured skin fibroblasts.

Authors:  C Eil
Journal:  J Clin Invest       Date:  1983-04       Impact factor: 14.808

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