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Clin Genet ; 94(3-4): 373-380, 2018 10.
Artigo em Inglês | MEDLINE | ID: mdl-29926465

RESUMO

By describing 10 new patients recruited in centres for Human Genetics, we further delineate the clinical spectrum of a Crouzon-like craniosynostosis disorder, officially termed craniosynostosis and dental anomalies (MIM614188). Singularly, it is inherited according to an autosomal recessive mode of inheritance. We identified six missense mutations in IL11RA, a gene encoding the alpha subunit of interleukin 11 receptor, 4 of them being novel, including 2 in the Ig-like C2-type domain. A subset of patients had an associated connective tissue disorder with joint hypermobility and intervertebral discs fragility. A smaller number of teeth anomalies than that previously reported in the two large series of patients evaluated in dental institutes points toward an ascertainment bias.


Assuntos
Disostose Craniofacial/genética , Genes Recessivos , Subunidade alfa de Receptor de Interleucina-11/genética , Adolescente , Adulto , Criança , Pré-Escolar , Disostose Craniofacial/diagnóstico por imagem , Feminino , Humanos , Imageamento por Ressonância Magnética , Masculino , Mutação de Sentido Incorreto
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