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A clinical scoring system for congenital contractural arachnodactyly.

Authors :
Meerschaut, Ilse
De Coninck, Shana
Steyaert, Wouter
Barnicoat, Angela
Bayat, Allan
Benedicenti, Francesco
Berland, Siren
Blair, Edward M
Breckpot, Jeroen
de Burca, Anna
Destrée, Anne
García-Miñaúr, Sixto
Green, Andrew J
Hanna, Bernadette C
Keymolen, Kathelijn
Koopmans, Marije
Lederer, Damien
Lees, Melissa
Longman, Cheryl
Lynch, Sally Ann
Male, Alison M
McKenzie, Fiona
Migeotte, Isabelle
Mihci, Ercan
Nur, Banu
Petit, Florence
Piard, Juliette
Plasschaert, Frank FS
Rauch, Anita
Ribai A Nyam, Pascale
Pacheco, Iratxe Salcedo
Stanzial, Franco
Stolte-Dijkstra, Irene
Valenzuela, Irene
Varghese, Vinod
Vasudevan, Pradeep C
Wakeling, Emma L
Wallgren-Pettersson, Carina
Coucke, Paul
De Paepe, Anne
De Wolf, Daniël
Symoens, Sofie
Callewaert, Bert
Meerschaut, Ilse
De Coninck, Shana
Steyaert, Wouter
Barnicoat, Angela
Bayat, Allan
Benedicenti, Francesco
Berland, Siren
Blair, Edward M
Breckpot, Jeroen
de Burca, Anna
Destrée, Anne
García-Miñaúr, Sixto
Green, Andrew J
Hanna, Bernadette C
Keymolen, Kathelijn
Koopmans, Marije
Lederer, Damien
Lees, Melissa
Longman, Cheryl
Lynch, Sally Ann
Male, Alison M
McKenzie, Fiona
Migeotte, Isabelle
Mihci, Ercan
Nur, Banu
Petit, Florence
Piard, Juliette
Plasschaert, Frank FS
Rauch, Anita
Ribai A Nyam, Pascale
Pacheco, Iratxe Salcedo
Stanzial, Franco
Stolte-Dijkstra, Irene
Valenzuela, Irene
Varghese, Vinod
Vasudevan, Pradeep C
Wakeling, Emma L
Wallgren-Pettersson, Carina
Coucke, Paul
De Paepe, Anne
De Wolf, Daniël
Symoens, Sofie
Callewaert, Bert
Source :
Genetics in medicine
Publication Year :
2019

Abstract

Congenital contractural arachnodactyly (CCA) is an autosomal dominant connective tissue disorder manifesting joint contractures, arachnodactyly, crumpled ears, and kyphoscoliosis as main features. Due to its rarity, rather aspecific clinical presentation, and overlap with other conditions including Marfan syndrome, the diagnosis is challenging, but important for prognosis and clinical management. CCA is caused by pathogenic variants in FBN2, encoding fibrillin-2, but locus heterogeneity has been suggested. We designed a clinical scoring system and diagnostic criteria to support the diagnostic process and guide molecular genetic testing.<br />SCOPUS: ar.j<br />info:eu-repo/semantics/published

Details

Database :
OAIster
Journal :
Genetics in medicine
Notes :
1 full-text file(s): application/pdf, English
Publication Type :
Electronic Resource
Accession number :
edsoai.on1192452135
Document Type :
Electronic Resource