Literature DB >> 22212252

17-β-hydroxysteroid dehydrogenase type 3 deficiency in three adult Iranian siblings.

M D Omrani1, T Adamovic, U Grandell, S Saleh-Gargari, Agneta Nordenskjöld.   

Abstract

17-β-hydroxysteroid dehydrogenase type 3 (17-β-HSD 3) deficiency is an autosomal recessive form of 46,XY disorder of sex development (DSD). To date, a total of 27 HSD17B3 gene mutations have been described in 46,XY patients exhibiting different phenotypes at birth and virilization at puberty, sometimes in association with gynecomastia. Herein, we investigate the 46,XY DSD in an Iranian family consisting of 7 siblings, 3 of which are affected and virilized at puberty. We clinically characterized these patients and performed direct DNA sequencing of the steroid 5-α-reductase type 2 (SRD5A2) and the HSD17B3 gene, respectively. We identified a homozygous mutation in the HSD17B3 gene (R80W; c.238C>G) in all affected siblings. No mutation was detected in the SRD5A2 gene. The detected mutation in the HSD17B3 gene was previously described in a newborn child, who died from other congenital malformations, and in a 12-year-old girl. Hence, our report adds novel value to the phenotype classification of 17-β-HSD 3 deficiency.
Copyright © 2011 S. Karger AG, Basel.

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Year:  2011        PMID: 22212252     DOI: 10.1159/000335006

Source DB:  PubMed          Journal:  Sex Dev        ISSN: 1661-5425            Impact factor:   1.824


  3 in total

1.  A Novel Mutation Causing 17-β-Hydroxysteroid Dehydrogenase Type 3 Deficiency in an Omani Child: First Case Report and Review of Literature.

Authors:  Aisha Al-Sinani; Waad-Allah S Mula-Abed; Manal Al-Kindi; Ghariba Al-Kusaibi; Hanan Al-Azkawi; Nahid Nahavandi
Journal:  Oman Med J       Date:  2015-03

2.  Complexities of gender assignment in 17β-hydroxysteroid dehydrogenase type 3 deficiency: is there a role for early orchiectomy?

Authors:  Janet Chuang; Amy Vallerie; Lesley Breech; Howard M Saal; Shumyle Alam; Peggy Crawford; Meilan M Rutter
Journal:  Int J Pediatr Endocrinol       Date:  2013-09-12

Review 3.  Disorder of Sex Development Due to 17-Beta-Hydroxysteroid Dehydrogenase Type 3 Deficiency: A Case Report and Review of 70 Different HSD17B3 Mutations Reported in 239 Patients.

Authors:  Catarina I Gonçalves; Josianne Carriço; Margarida Bastos; Manuel C Lemos
Journal:  Int J Mol Sci       Date:  2022-09-02       Impact factor: 6.208

  3 in total

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