Literature DB >> 32643049

A non-surgical approach to 46,XY differences in sex development through hormonal suppression at puberty: a single-center case series study.

Katie L Canalichio1,2, Margarett Shnorhavorian3,4, Anne-Marie Amies Oelschlager3,4, Linda Ramsdell3, Christina Fisher3, Margaret P Adam3,4, Patricia Y Fechner3,4.   

Abstract

PURPOSE: We aim to report outcomes and safety with hormonal suppression to facilitate gonadal preservation in a select group of patients with 46,XY differences in sex development (DSD) who are raised and identify as female yet have diagnoses with potential for androgenization at puberty.
METHODS: We performed a retrospective review of the past 10 years of DSD patients treated by a multidisciplinary program. Inclusion criteria were 46,XY DSD, female sex of rearing, risk of androgenization at puberty, and plan for hormonal suppression at puberty. Patients on hormonal suppression had at least 6 months of follow-up from initiation. We excluded those with complete gonadal dysgenesis or complete androgen insensitivity.
RESULTS: Four patients met inclusion criteria. Initial evaluation by DSD team was at a mean age of 6.6 years (3 weeks-16 years). All patients were evaluated in a coordinated multidisciplinary clinic. The diagnoses are listed in Table 1. Mean follow-up was 5.7 years (1.2-10.9 years). One patient presented as an infant, and is being monitored until Tanner stage 2 and/or serum hormonal evidence to initiate hormonal suppression. Three patients have been receiving hormonal suppression for 1.4 years (1.1-1.9 years) without side effects or complication. Three patients were initiated with estrogen replacement to promote desired breast development. At last follow-up, all patients had retained their gonads, all have female gender identity with no reported gender dysphoria, and no progression of androgenization.
CONCLUSIONS: In our initial experience, gonadal preservation with hormonal suppression is a tool in multidisciplinary management of select DSD patients with female gender identity with conditions associated with androgenization at puberty. Patients' growth, bone health, and overall psychosocial well-being will need to be monitored closely.

Entities:  

Keywords:  Differences in sex development; Gender identity; Gonadectomy; Gonadotropin releasing hormone; Hormonal suppression

Mesh:

Year:  2020        PMID: 32643049     DOI: 10.1007/s12020-020-02409-y

Source DB:  PubMed          Journal:  Endocrine        ISSN: 1355-008X            Impact factor:   3.633


  20 in total

1.  A mutation in the gene encoding steroidogenic factor-1 causes XY sex reversal and adrenal failure in humans.

Authors:  J C Achermann; M Ito; M Ito; P C Hindmarsh; J L Jameson
Journal:  Nat Genet       Date:  1999-06       Impact factor: 38.330

Review 2.  46,XY disorder of sex development (DSD) due to 17β-hydroxysteroid dehydrogenase type 3 deficiency.

Authors:  Berenice B Mendonca; Nathalia Lisboa Gomes; Elaine M F Costa; Marlene Inacio; Regina M Martin; Mirian Y Nishi; Filomena Marino Carvalho; Francisco Denes Tibor; Sorahia Domenice
Journal:  J Steroid Biochem Mol Biol       Date:  2016-05-06       Impact factor: 4.292

Review 3.  The effect of 5α-reductase-2 deficiency on human fertility.

Authors:  Hey-Joo Kang; Julianne Imperato-McGinley; Yuan-Shan Zhu; Zev Rosenwaks
Journal:  Fertil Steril       Date:  2014-01-08       Impact factor: 7.329

4.  Fertility preservation in a family with a novel NR5A1 mutation.

Authors:  Hiroko Yagi; Masaki Takagi; Masafumi Kon; Maki Igarashi; Maki Fukami; Yukihiro Hasegawa
Journal:  Endocr J       Date:  2014-12-12       Impact factor: 2.349

Review 5.  Spontaneous virilization around puberty in NR5A1-related 46,XY sex reversal: additional case and a literature review.

Authors:  Masanori Adachi; Tomonobu Hasegawa; Yukichi Tanaka; Yumi Asakura; Junko Hanakawa; Koji Muroya
Journal:  Endocr J       Date:  2018-09-15       Impact factor: 2.349

6.  Analysis of DAX1 (NR0B1) and steroidogenic factor-1 (NR5A1) in children and adults with primary adrenal failure: ten years' experience.

Authors:  Lin Lin; Wen-Xia Gu; Gokhan Ozisik; Wing S To; Catherine J Owen; J Larry Jameson; John C Achermann
Journal:  J Clin Endocrinol Metab       Date:  2006-05-09       Impact factor: 5.958

7.  Summary of consensus statement on intersex disorders and their management. International Intersex Consensus Conference.

Authors:  Christopher P Houk; Ieuan A Hughes; S Faisal Ahmed; Peter A Lee
Journal:  Pediatrics       Date:  2006-08       Impact factor: 7.124

Review 8.  Steroidogenic factor-1 (SF-1, NR5A1) and human disease.

Authors:  Bruno Ferraz-de-Souza; Lin Lin; John C Achermann
Journal:  Mol Cell Endocrinol       Date:  2010-11-13       Impact factor: 4.102

Review 9.  Wide spectrum of NR5A1-related phenotypes in 46,XY and 46,XX individuals.

Authors:  Sorahia Domenice; Aline Zamboni Machado; Frederico Moraes Ferreira; Bruno Ferraz-de-Souza; Antonio Marcondes Lerario; Lin Lin; Mirian Yumie Nishi; Nathalia Lisboa Gomes; Thatiana Evelin da Silva; Rosana Barbosa Silva; Rafaela Vieira Correa; Luciana Ribeiro Montenegro; Amanda Narciso; Elaine Maria Frade Costa; John C Achermann; Berenice Bilharinho Mendonca
Journal:  Birth Defects Res C Embryo Today       Date:  2016-12

10.  Phenotype and Molecular Characterizations of 30 Children From China With NR5A1 Mutations.

Authors:  Yanning Song; Lijun Fan; Chunxiu Gong
Journal:  Front Pharmacol       Date:  2018-10-30       Impact factor: 5.810

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