Winchester syndrome caused by a homozygous mutation affecting the active site of matrix metalloproteinase 2.

Détails

ID Serval
serval:BIB_B5EDDC543658
Type
Article: article d'un périodique ou d'un magazine.
Sous-type
Etude de cas (case report): rapporte une observation et la commente brièvement.
Collection
Publications
Institution
Titre
Winchester syndrome caused by a homozygous mutation affecting the active site of matrix metalloproteinase 2.
Périodique
Clinical Genetics
Auteur⸱e⸱s
Zankl A., Bonafé L., Calcaterra V., Di Rocco M., Superti-Furga A.
ISSN
0009-9163
Statut éditorial
Publié
Date de publication
03/2005
Peer-reviewed
Oui
Volume
67
Numéro
3
Pages
261-266
Langue
anglais
Notes
Publication types: Case Reports ; Journal Article
Résumé
The inherited osteolysis syndromes are a heterogeneous group of skeletal disorders whose classification is still uncertain. Three osteolysis syndromes show autosomal recessive inheritance and multicentric involvement: Torg syndrome (OMIM 259600), Winchester syndrome (OMIM 277950) and Nodulosis-Arthropathy-Osteolysis syndrome (NAO; OMIM 605156). The 2001 Nosology of the International Skeletal Dysplasia Society (Hall CM, Am J Med Genet 2002: 113: 65) classifies NAO as a variant of Torg syndrome, while Winchester syndrome is considered as a separate disorder. Recently, mutations in the matrix metalloproteinase 2 (MMP2) gene were identified in affected individuals with a clinical diagnosis of NAO in two Arab families. We report a homozygous missense mutation (E404K) in the active site of MMP2 in a 21-year-old woman with a severe form of osteolysis best compatible with a diagnosis of Winchester syndrome. The clinical and molecular findings suggest that Torg, NAO and Winchester syndromes are allelic disorders that form a clinical spectrum.
Mots-clé
Adult, Arabs/genetics, DNA Mutational Analysis, Female, Humans, Matrix Metalloproteinase 2/genetics, Mutation, Missense, Osteolysis/genetics
Pubmed
Web of science
Création de la notice
21/01/2008 13:50
Dernière modification de la notice
20/08/2019 16:24
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