Genetic mechanisms underlying 46,XY DSD with gonadal dysgenesis

Louisa Ludbrook, Vincent R. Harley

Research output: Chapter in Book/Report/Conference proceedingChapter (Book)Otherpeer-review

Abstract

Genetic changes in key sex-determining genes lead to 46,XY disorders of sex development (DSD) in humans. Loss-of-function mutation in the sex-determining region on Y (SRY), steroidogenic factor 1 (SF1), and SRY-Box gene 9 (SOX9) genes causes varying phenotypic changes ranging from complete syndromic gonadal dysgenesis to isolated hypospadias. Similarly, gain of function of dosage-sensitive sex reversal adrenal hypoplasia congenita on X gene 1 (DAX1) through duplications within the Xp21 region also affects typical testicular determination, together demonstrating the critical roles these genes play in determining sex. The molecular mechanisms through which these transcription factors act during sex determination are poorly understood and our current efforts aim at delineating the complex interactions that fail due to mutation or deletion in human DSD.

Original languageEnglish
Title of host publicationHormonal and Genetic Basis of Sexual Differentiation Disorders and Hot Topics in Endocrinology
Subtitle of host publicationProceedings of the 2nd World Conference
EditorsMaria I. New, Joe Leigh Simpson
PublisherSpringer Science + Business Media
Pages87-88
Number of pages2
Volume707
ISBN (Print)9781441980014
DOIs
Publication statusPublished - 2011
Externally publishedYes

Publication series

NameAdvances in Experimental Medicine and Biology
Volume707
ISSN (Print)00652598

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