Literature DB >> 28690896

Late Diagnosis of 5-α-Reductase Type 2 Deficiency in an Adolescent Girl with Primary Amenorrhoea.

Abdul-Rahman A Hummadi1, Ayel O Yahya1, Awad M Al-Qahtani2.   

Abstract

Deficiency of the 5-α-reductase enzyme has been found to affect male sexual development. We report an 18-year-old patient who was referred to an endocrinology clinic in Jizan, Saudi Arabia, in April 2014 with primary amenorrhoea, virilisation and a lack of secondary sex characteristics. As female external genitalia were present at birth, she had been raised as a female. Magnetic resonance imaging revealed no uterine or ovarian tissue in the pelvis and the presence of a scrotal sac. She was diagnosed with 5-α-reductase type 2 deficiency, a 46,XY disorder of sexual development. Typically, affected males have pseudovaginal perineoscrotal hypospadias and ambiguous genitalia at birth. Individuals who have been raised as female manifest characteristics of virilisation at puberty, including deepening of the vocal tone, phallus enlargement, scrotal hyperpigmentation and increased muscle mass.

Entities:  

Keywords:  46; Case Report; Dihydrotestosterone; Hypospadias; Puberty; Saudi Arabia; Testosterone 5-alpha-Reductase; XY Disorders of Sex Development

Mesh:

Substances:

Year:  2017        PMID: 28690896      PMCID: PMC5488825          DOI: 10.18295/squmj.2016.17.02.014

Source DB:  PubMed          Journal:  Sultan Qaboos Univ Med J        ISSN: 2075-051X


  8 in total

1.  Phenotypic classification of male pseudohermaphroditism due to steroid 5 alpha-reductase 2 deficiency.

Authors:  G H Sinnecker; O Hiort; L Dibbelt; N Albers; H G Dörr; H Hauss; U Heinrich; M Hemminghaus; W Hoepffner; M Holder; D Schnabel; K Kruse
Journal:  Am J Med Genet       Date:  1996-05-03

2.  Difficulties in diagnosis and treatment of 5alpha-reductase type 2 deficiency in a newborn with 46,XY DSD.

Authors:  Kerstin N Walter; Frederike B Kienzle; Alexander Frankenschmidt; Olaf Hiort; Stefan A Wudy; Natascha van der Werf-Grohmann; Andrea Superti-Furga; Karl Otfried Schwab
Journal:  Horm Res Paediatr       Date:  2010-04-16       Impact factor: 2.852

3.  The IVS1-2A>G mutation in the SRD5A2 gene predominates in Cypriot patients with 5α reductase deficiency.

Authors:  N Skordis; V Neocleous; A Kyriakou; E Efstathiou; A Sertedaki; P Philibert; L A Phylactou; S Lumbroso; C Sultan
Journal:  J Endocrinol Invest       Date:  2010-05-28       Impact factor: 4.256

Review 4.  Steroid 5 alpha-reductase 2 deficiency.

Authors:  J D Wilson; J E Griffin; D W Russell
Journal:  Endocr Rev       Date:  1993-10       Impact factor: 19.871

5.  Male pseudohermaphroditism due to steroid 5alpha-reductase 2 deficiency. Diagnosis, psychological evaluation, and management.

Authors:  B B Mendonca; M Inacio; E M Costa; I J Arnhold; F A Silva; W Nicolau; W Bloise; D W Russel; J D Wilson
Journal:  Medicine (Baltimore)       Date:  1996-03       Impact factor: 1.889

6.  Early diagnosis of 5alpha-reductase deficiency in newborns.

Authors:  S Bertelloni; R T Scaramuzzo; D Parrini; F Baldinotti; S Tumini; P Ghirri
Journal:  Sex Dev       Date:  2007       Impact factor: 1.824

7.  Steroid 5alpha-reductase deficiency in man: an inherited form of male pseudohermaphroditism.

Authors:  J Imperato-McGinley; L Guerrero; T Gautier; R E Peterson
Journal:  Science       Date:  1974-12-27       Impact factor: 47.728

8.  5-alpha-reductase 2 deficiency in a woman with primary amenorrhea.

Authors:  Nasrollah Maleki; Mohammadreza Kalantar Hormozi; Manouchehr Iranparvar Alamdari; Zahra Tavosi
Journal:  Case Rep Endocrinol       Date:  2013-12-07
  8 in total

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