| Literature DB >> 29926465 |
E Brischoux-Boucher1, A Trimouille2, G Baujat3, A Goldenberg4, E Schaefer5, B Guichard6, P Hannequin7, G Paternoster8, S Baer5, C Cabrol1, E Weber9, G Godfrin10, M Lenoir11, D Lacombe2, C Collet12, L Van Maldergem1,13,14.
Abstract
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.Entities:
Keywords: Crouzon syndrome; IL11RA; craniosynostosis; hyperlaxity
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Year: 2018 PMID: 29926465 DOI: 10.1111/cge.13409
Source DB: PubMed Journal: Clin Genet ISSN: 0009-9163 Impact factor: 4.438