Calvarial doughnut lesions with bone fragility in a French-Canadian family; case report and review of the literature.

Bone fragility Calvarial doughnut lesions with bone fragility SGMS2 Skeletal dysplasia

Journal

Bone reports
ISSN: 2352-1872
Titre abrégé: Bone Rep
Pays: United States
ID NLM: 101646176

Informations de publication

Date de publication:
Dec 2021
Historique:
received: 04 05 2021
revised: 03 08 2021
accepted: 20 08 2021
entrez: 10 9 2021
pubmed: 11 9 2021
medline: 11 9 2021
Statut: epublish

Résumé

Calvarial Doughnut Lesions with Bone Fragility (CDL) is an autosomal dominant genetic disease, characterized by low bone mineral density, multiple fractures starting in childhood, and sclerotic doughnut-shaped lesions in the cranial bones. Aubé and colleagues described in 1988 a French-Canadian family of 12 affected members who had a clinical diagnosis of doughnut lesions of the skull, with pathological fractures, osteopenia, "bone in bone" in the vertebral bodies and squaring of metatarsal and metacarpal bones. Herein we study new members of this family. Sequential genetic testing identified a nonsense variant c.148C>T, p. Arg50

Identifiants

pubmed: 34504906
doi: 10.1016/j.bonr.2021.101121
pii: S2352-1872(21)00378-8
pmc: PMC8414042
doi:

Types de publication

Case Reports

Langues

eng

Pagination

101121

Informations de copyright

© 2021 Published by Elsevier Inc.

Déclaration de conflit d'intérêts

The authors declare no conflict of interest.

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Auteurs

Shuaa Basalom (S)

Division of Medical Genetics, Department of Specialized Medicine, McGill University Health Center, Montreal, QC H4A 3J1, Canada.

Mélissa Fiscaletti (M)

Department of Pediatrics, CHU Sainte-Justine, Montreal, QC H3T 1C5, Canada.

Valancy Miranda (V)

Medical Genetics Division, Department of Pediatrics, CHU Sainte-Justine, Montreal, QC H3T 1C5, Canada.

Céline Huber (C)

Department of Medical Genetics, INSERM UMR 1163, Paris Descartes-Sorbonne Paris Cité University, IMAGINE Institute, Necker Enfants Malades Hospital, Paris, France.

Guillaume Couture (G)

Centre de rhumatologie, CHU Purpan, 31300 Toulouse, France.

Régen Drouin (R)

Division of Medical Genetics, Department of Pediatrics, CHU de Quebec - ULaval, Faculty of Medicine, Université Laval, Quebec City, Canada.

Élise Monceau (É)

Department of Pediatrics, CHU Sainte-Justine, Montreal, QC H3T 1C5, Canada.

Sandrine Wavrant (S)

Department of Obstetrics and Gynecology, CHU Sainte-Justine, Montreal, QC H3T 1C5, Canada.

Johanne Dubé (J)

Department of Obstetrics and Gynecology, CHU Sainte-Justine, Montreal, QC H3T 1C5, Canada.

Outi Mäkitie (O)

Children's Hospital and Pediatric Research Center, University of Helsinki and Helsinki University Hospital, FI-00014 Helsinki, Finland.

Valérie Cormier-Daire (V)

Centre de référence MOC, hôpital Necker, 75015 Paris, France.

Philippe M Campeau (PM)

Medical Genetics Division, Department of Pediatrics, CHU Sainte-Justine, Montreal, QC H3T 1C5, Canada.

Classifications MeSH