In vitro functional characterization of androgen receptor gene mutations at arginine p.856 of the ligand-binding-domain associated with androgen insensitivity syndrome.


Journal

The Journal of steroid biochemistry and molecular biology
ISSN: 1879-1220
Titre abrégé: J Steroid Biochem Mol Biol
Pays: England
ID NLM: 9015483

Informations de publication

Date de publication:
04 2021
Historique:
received: 03 11 2020
revised: 12 01 2021
accepted: 29 01 2021
pubmed: 7 2 2021
medline: 8 5 2021
entrez: 6 2 2021
Statut: ppublish

Résumé

Androgens are critical for male sex differentiation. Their actions are mediated by the androgen receptor (AR). Mutations disrupting AR function result in the androgen insensitivity syndrome (AIS). In this study, we identified in a patient with complete AIS, a novel AR mutation p.R856L. To investigate the functional properties of p.R856L, we performed functional studies. In comparison, we have characterized two already described mutations: p.R856H and p.R856C. We used a model composed of two different promoters fused to a reporter gene, two cell lines, and showed that all mutations were able to transactivate the (ARE)

Identifiants

pubmed: 33548461
pii: S0960-0760(21)00027-3
doi: 10.1016/j.jsbmb.2021.105834
pii:
doi:

Substances chimiques

AR protein, human 0
Androgens 0
Ligands 0
Receptors, Androgen 0
Arginine 94ZLA3W45F

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

105834

Informations de copyright

Copyright © 2021 Elsevier Ltd. All rights reserved.

Auteurs

Asma Tajouri (A)

University of Tunis El Manar, Faculty of Medicine of Tunis, LR99ES10 Human Genetics Laboratory, 1007, Tunis, Tunisia; Department of Paediatric and Adolescent Medicine, Division of Paediatric Endocrinology and Diabetes, University of Luebeck, 23562, Luebeck, Germany.

Maher Kharrat (M)

University of Tunis El Manar, Faculty of Medicine of Tunis, LR99ES10 Human Genetics Laboratory, 1007, Tunis, Tunisia. Electronic address: maher.kharrat@fmt.rnu.tn.

Mediha Trabelsi (M)

University of Tunis El Manar, Faculty of Medicine of Tunis, LR99ES10 Human Genetics Laboratory, 1007, Tunis, Tunisia; Department of Congenital and Hereditary Diseases, Charles Nicolle Hospital, Tunis, Tunisia.

Ridha M'rad (R)

University of Tunis El Manar, Faculty of Medicine of Tunis, LR99ES10 Human Genetics Laboratory, 1007, Tunis, Tunisia; Department of Congenital and Hereditary Diseases, Charles Nicolle Hospital, Tunis, Tunisia.

Olaf Hiort (O)

Department of Paediatric and Adolescent Medicine, Division of Paediatric Endocrinology and Diabetes, University of Luebeck, 23562, Luebeck, Germany.

Ralf Werner (R)

Department of Paediatric and Adolescent Medicine, Division of Paediatric Endocrinology and Diabetes, University of Luebeck, 23562, Luebeck, Germany; Institute of Molecular Medicine, University of Luebeck, 23562, Luebeck, Germany. Electronic address: ralf.werner@uni-luebeck.de.

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Classifications MeSH