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Skeletal abnormalities, pediatric-onset severe osteoporosis, and multiple fragility fractures in a patient with a novel CTNNB1 de novo variant

Authors :
Olga Lesnyak
Francesca Marini
Polina Sokolnikova
Margarita Sorokina
Kseniya Sukhareva
Irina Artamonova
Vladimir Kenis
Olga Tkach
Anna Kostareva
Maria Luisa Brandi
Source :
Bone Reports, Vol 21, Iss , Pp 101777- (2024)
Publication Year :
2024
Publisher :
Elsevier, 2024.

Abstract

We report a case of a patient with a de novo germline heterozygous truncating variant of CTNNB1 gene (c.2172del, p.Tyr724Ter) causing neurodevelopmental disorder with spastic diplegia and visual defects syndrome (NEDSDV) associated with a new clinical feature — severe pediatric-onset osteoporosis and multiple fractures. A functional effect of the identified variant was demonstrated using adipose-tissue derived primary mesenchymal stem cells, where we detected the alteration of CTNNB1mRNA and β-catenin protein levels using real-time PCR and Western blot analysis.

Details

Language :
English
ISSN :
23521872
Volume :
21
Issue :
101777-
Database :
Directory of Open Access Journals
Journal :
Bone Reports
Publication Type :
Academic Journal
Accession number :
edsdoj.0d254560ddb448c189ae537ba88345dc
Document Type :
article
Full Text :
https://doi.org/10.1016/j.bonr.2024.101777