Centre de Génétique Humaine, Université de Franche-Comté, Besançon, France.
CHU Bordeaux, Service de Génétique Médicale, INSERM U1211, Université de Bordeaux, Bordeaux, France.
Clin Genet. 2018 Oct;94(3-4):373-380. doi: 10.1111/cge.13409. Epub 2018 Jul 23.
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.
通过描述在人类遗传学中心招募的 10 名新患者,我们进一步描述了一种类 Crouzon 颅缝早闭疾病的临床谱,该疾病正式命名为颅缝早闭和牙齿异常(MIM614188)。该疾病为常染色体隐性遗传模式遗传。我们在编码白介素 11 受体α亚基的基因 IL11RA 中发现了六个错义突变,其中四个是新的,包括 2 个在 Ig 样 C2 型结构域。一部分患者伴有结缔组织疾病,表现为关节过度活动和椎间盘脆弱。与之前在牙科研究所评估的两个大型患者系列中报道的牙齿异常数量相比,数量较少,提示存在检出偏倚。