Literature DB >> 9245853

Phenotypic diversity in siblings with partial androgen insensitivity syndrome.

B A Evans1, I A Hughes, C L Bevan, M N Patterson, J W Gregory.   

Abstract

The androgen insensitivity syndrome is a heterogeneous disorder with a wide spectrum of phenotypic abnormalities, ranging from complete female to ambiguous forms that more closely resemble males. The primary abnormality is a defective androgen receptor protein due to a mutation of the androgen receptor gene. This prevents normal androgen action and thus leads to impaired virilisation. A point mutation of the androgen receptor gene affecting two siblings with partial androgen insensitivity syndrome is described. One had cliteromegaly and labial fusion and was raised as a girl, whereas the other sibling had micropenis and penoscrotal hypospadias and was raised as a boy. Both were shown to have the arginine 840 to cysteine mutation. The phenotypic variation in this family is thus dependent on factors other than abnormalities of the androgen receptor gene alone.

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Year:  1997        PMID: 9245853      PMCID: PMC1717223          DOI: 10.1136/adc.76.6.529

Source DB:  PubMed          Journal:  Arch Dis Child        ISSN: 0003-9888            Impact factor:   3.791


  23 in total

1.  Mutations in the ligand-binding domain of the androgen receptor gene cluster in two regions of the gene.

Authors:  M J McPhaul; M Marcelli; S Zoppi; C M Wilson; J E Griffin; J D Wilson
Journal:  J Clin Invest       Date:  1992-11       Impact factor: 14.808

2.  A new point mutation in the luteinising hormone receptor gene in familial and sporadic male limited precocious puberty: genotype does not always correlate with phenotype.

Authors:  B A Evans; D J Bowen; P J Smith; P E Clayton; J W Gregory
Journal:  J Med Genet       Date:  1996-02       Impact factor: 6.318

3.  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

4.  Familial incomplete male pseudohermaphroditism, type 1. Evidence for androgen resistance and variable clinical manifestations in a family with the Reifenstein syndrome.

Authors:  J D Wilson; M J Harrod; J L Goldstein; D L Hemsell; P C MacDonald
Journal:  N Engl J Med       Date:  1974-05-16       Impact factor: 91.245

5.  Prenatal diagnosis of androgen insensitivity.

Authors:  I A Hughes; M N Patterson
Journal:  Clin Endocrinol (Oxf)       Date:  1994-03       Impact factor: 3.478

6.  Phenotypic variation and detection of carrier status in the partial androgen insensitivity syndrome.

Authors:  J A Batch; H R Davies; B A Evans; I A Hughes; M N Patterson
Journal:  Arch Dis Child       Date:  1993-04       Impact factor: 3.791

7.  Studies of the androgen receptor in dispersed fibroblasts: investigation of patients with androgen insensitivity.

Authors:  B A Evans; T R Jones; I A Hughes
Journal:  Clin Endocrinol (Oxf)       Date:  1984-01       Impact factor: 3.478

8.  Domains of the human androgen receptor involved in steroid binding, transcriptional activation, and subcellular localization.

Authors:  G Jenster; H A van der Korput; C van Vroonhoven; T H van der Kwast; J Trapman; A O Brinkmann
Journal:  Mol Endocrinol       Date:  1991-10

Review 9.  Androgen resistance due to mutation of the androgen receptor.

Authors:  L Pinsky; M Trifiro; M Kaufman; L K Beitel; A Mhatre; P Kazemi-Esfarjani; N Sabbaghian; R Lumbroso; C Alvarado; M Vasiliou
Journal:  Clin Invest Med       Date:  1992-10       Impact factor: 0.825

10.  Androgen receptor gene mutations identified by SSCP in fourteen subjects with androgen insensitivity syndrome.

Authors:  J A Batch; D M Williams; H R Davies; B D Brown; B A Evans; I A Hughes; M N Patterson
Journal:  Hum Mol Genet       Date:  1992-10       Impact factor: 6.150

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  9 in total

1.  Inherited human sex reversal due to impaired nucleocytoplasmic trafficking of SRY defines a male transcriptional threshold.

Authors:  Yen-Shan Chen; Joseph D Racca; Nelson B Phillips; Michael A Weiss
Journal:  Proc Natl Acad Sci U S A       Date:  2013-09-03       Impact factor: 11.205

2.  Androgen insensitivity syndrome: a survey of diagnostic procedures and management in the UK.

Authors:  R M Viner; Y Teoh; D M Williams; M N Patterson; I A Hughes
Journal:  Arch Dis Child       Date:  1997-10       Impact factor: 3.791

3.  Three novel and two known androgen receptor gene mutations associated with androgen insensitivity syndrome in sex-reversed XY female patients.

Authors:  Balachandran Saranya; Gunasekaran Bhavani; Brindha Arumugam; Meena Jayashankar; Sathiyavedu Thyagarajan Santhiya
Journal:  J Genet       Date:  2016-12       Impact factor: 1.166

4.  Isolated micropenis reveals partial androgen insensitivity syndrome confirmed by molecular analysis.

Authors:  Amrit Bhangoo; Francoise Paris; Pascal Philibert; Francoise Audran; Svetlana Ten; Charles Sultan
Journal:  Asian J Androl       Date:  2010-03-22       Impact factor: 3.285

Review 5.  Mutation of the androgen receptor (R840S) in an Egyptian patient with partial androgen insensitivity syndrome: review of the literature on the clinical expression of different R840 substitutions.

Authors:  I Mazen; S Lumbroso; S Abdel Ghaffar; N Salah; C Sultan
Journal:  J Endocrinol Invest       Date:  2004-01       Impact factor: 4.256

6.  Apolipoprotein D (APOD) is a putative biomarker of androgen receptor function in androgen insensitivity syndrome.

Authors:  Mahesh Appari; Ralf Werner; Lutz Wünsch; Gunnar Cario; Janos Demeter; Olaf Hiort; Felix Riepe; James D Brooks; Paul-Martin Holterhus
Journal:  J Mol Med (Berl)       Date:  2009-03-30       Impact factor: 4.599

7.  Severe forms of partial androgen insensitivity syndrome due to p.L830F novel mutation in androgen receptor gene in a Brazilian family.

Authors:  Reginaldo J Petroli; Andréa T Maciel-Guerra; Fernanda C Soardi; Flávia L de Calais; Gil Guerra-Junior; Maricilda Palandi de Mello
Journal:  BMC Res Notes       Date:  2011-06-06

8.  Genetic analysis of a family with complete androgen insensitivity syndrome.

Authors:  Sunil Kumar Kota; Kotni Gayatri; Siva Krishna Kota; Sruti Jammula
Journal:  Indian J Hum Genet       Date:  2013-07

9.  A Novel Mutation in Human Androgen Receptor Gene Causing Partial Androgen Insensitivity Syndrome in a Patient Presenting with Gynecomastia at Puberty.

Authors:  Cemil Koçyiğit; Serdar Sarıtaş; Gönül Çatlı; Hüseyin Onay; Bumin Nuri Dündar
Journal:  J Clin Res Pediatr Endocrinol       Date:  2016-04-18
  9 in total

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