Late diagnosis of complete androgen insensitivity syndrome and transmission/carriers of the condition in a family with mutation c.2495G> T p.(Arg832Leu) in exon 7 of the androgen receptor gene: genetic, clinical and ethical aspects.


Journal

Biomedical papers of the Medical Faculty of the University Palacky, Olomouc, Czechoslovakia
ISSN: 1804-7521
Titre abrégé: Biomed Pap Med Fac Univ Palacky Olomouc Czech Repub
Pays: Czech Republic
ID NLM: 101140142

Informations de publication

Date de publication:
Dec 2019
Historique:
received: 12 06 2018
accepted: 17 10 2018
pubmed: 8 11 2018
medline: 9 6 2020
entrez: 8 11 2018
Statut: ppublish

Résumé

The complete androgen insensitivity syndrome (CAIS) is a rare genetic disorder causing insensitivity to androgens in a person with female phenotype and 46,XY karyotype due to a mutation in the androgen receptor gene located on chromosome X. These children are born with female external genitalia, and females are transmitters. We illustrate an unexpected diagnosis of CAIS in two siblings during examination for short stature, and describe transmission/carriers in the family along with ethical aspects. A genetic examination could have earlier revealed the transmission of c.2495G>Tp.(Arg832Leu) mutation in exon 7. Our experience highlights the possibility of prenatal testing for the management of pregnancy in a family with a history of CAIS. The implications of prenatal testing in relation to CAIS with clearer explication of ethical and clinical issues warrant further investigation.

Sections du résumé

BACKGROUND BACKGROUND
The complete androgen insensitivity syndrome (CAIS) is a rare genetic disorder causing insensitivity to androgens in a person with female phenotype and 46,XY karyotype due to a mutation in the androgen receptor gene located on chromosome X. These children are born with female external genitalia, and females are transmitters.
CASE REPORT METHODS
We illustrate an unexpected diagnosis of CAIS in two siblings during examination for short stature, and describe transmission/carriers in the family along with ethical aspects.
CONCLUSION CONCLUSIONS
A genetic examination could have earlier revealed the transmission of c.2495G>Tp.(Arg832Leu) mutation in exon 7. Our experience highlights the possibility of prenatal testing for the management of pregnancy in a family with a history of CAIS. The implications of prenatal testing in relation to CAIS with clearer explication of ethical and clinical issues warrant further investigation.

Identifiants

pubmed: 30401990
doi: 10.5507/bp.2018.067
doi:

Substances chimiques

Receptors, Androgen 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

379-382

Auteurs

Renata Pomahacova (R)

Department of Pediatrics, Faculty of Medicine in Pilsen, Charles University in Prague and Faculty Hospital, Pilsen, Czech Republic.

Jana Zamboryova (J)

Department of Pediatrics, Faculty of Medicine in Pilsen, Charles University in Prague and Faculty Hospital, Pilsen, Czech Republic.

Petra Paterova (P)

Department of Pediatrics, Faculty of Medicine in Pilsen, Charles University in Prague and Faculty Hospital, Pilsen, Czech Republic.

Anna Krepelova (A)

Department of Biology and Medical Genetics, 2 nd Faculty of Medicine, Charles University in Prague and Motol University Hospital, Czech Republic.

Ivan Subrt (I)

Institute of Medical Genetics, Faculty of Medicine in Pilsen, Charles University in Prague and Faculty Hospital, Pilsen, Czech Republic.

Radka Jaklova (R)

Institute of Medical Genetics, Faculty of Medicine in Pilsen, Charles University in Prague and Faculty Hospital, Pilsen, Czech Republic.

Petra Vohradska (P)

Institute of Medical Genetics, Faculty of Medicine in Pilsen, Charles University in Prague and Faculty Hospital, Pilsen, Czech Republic.

Eliska Hrdonkova (E)

Department of Gynecology, Faculty of Medicine in Pilsen, Charles University in Prague and Faculty Hospital, Pilsen, Czech Republic.

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Classifications MeSH