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DOI 10.1159/000527754

Медицинская библиотека · Sexual development : genetics, molecular biology, evolution, endocrinology, embryology, and pathology of sex determination and differentiation · 2023

Genetic Variants in SRD5A2 in a Spectrum of DSD Patients from Australian Clinics Highlight Importance of Genetic Testing alongside Typical First-Line Investigations.

Robevska Gorjana, Hanna Chloe, van den Bergen Jocelyn, Welch John, Couper Jennifer, Harris Shannon, Joshi Kriti, Brown Justin, Sabin Matthew, Sinclair Andrew, O'Connell Michele, Ayers Katie

СтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

INTRODUCTION

Steroid 5-alpha reductase deficiency (5α-R2D) is a rare condition caused by genetic variants that reduce the activity of the enzyme that converts testosterone into dihydrotestosterone. The clinical spectrum of 5α-R2D is known to overlap with other 46,XY differences of sex development (DSD) such as androgen insensitivity or gonadal dysgenesis. However, the clinical trajectories of the aetiologies can differ, with 5α-R2D presenting its own challenges.

METHODS

In this study, we have collated clinical information for five individuals with variants in SRD5A2 identified using research genetic testing in an Australian paediatric setting.

RESULTS

We describe how a genetic finding resolved or confirmed a diagnosis for these individuals and how it guided clinical management and family counselling.

CONCLUSION

This work highlights the importance of early genetic testing in children born with 46,XY DSD where it complements traditional first-line testing.

Для цитирования: Robevska Gorjana, Hanna Chloe, van den Bergen Jocelyn, Welch John, Couper Jennifer, Harris Shannon, Joshi Kriti, Brown Justin, Sabin Matthew, Sinclair Andrew, O'Connell Michele, Ayers Katie. Genetic Variants in SRD5A2 in a Spectrum of DSD Patients from Australian Clinics Highlight Importance of Genetic Testing alongside Typical First-Line Investigations.. Sexual development : genetics, molecular biology, evolution, endocrinology, embryology, and pathology of sex determination and differentiation. 2023. DOI: 10.1159/000527754.

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