Cospain Auriane, Schaefer Elise, Faoucher Marie, Dubourg Christèle, Carré Wilfrid, Bizaoui Varoona, Assoumani Jessica, Van Maldergem Lionel, Piton Amélie, Gérard Bénédicte, Tran Mau-Them Frédéric, Bruel Ange-Line, Faivre Laurence, Demurger Florence, Pasquier Laurent, Odent Sylvie, Fradin Mélanie, Lavillaureix Alinoë
CHU Rennes, Service de Génétique Clinique, Centre de Référence Maladies Rares CLAD-Ouest, ERN ITHACA, Hôpital Sud, Rennes, France.
Service de Génétique Médicale, Institut de Génétique Médicale d'Alsace, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.
Clin Genet. 2021 May;99(5):732-739. doi: 10.1111/cge.13933. Epub 2021 Feb 8.
Skraban-Deardorff syndrome (a disease related to variations in the WDR26 gene; OMIM #617616) was first described in a cohort of 15 individuals in 2017. The syndrome comprises intellectual deficiency, severe speech impairment, ataxic gait, seizures, mild hypotonia with feeding difficulties during infancy, and dysmorphic features. Here, we report on six novel heterozygous de novo pathogenic variants in WDR26 in six probands. The patients' phenotypes were consistent with original publication. One patient displayed marked hypotonia with an abnormal muscle biopsy; this finding warrants further investigation. Gait must be closely monitored, in order to highlight any musculoskeletal or neurological abnormalities and prompt further examinations. Speech therapy and alternative communication methods should be initiated early in the clinical follow-up, in order to improve language and oral eating and drinking.
Skraban-Deardorff综合征(一种与WDR26基因变异相关的疾病;OMIM编号#617616)于2017年在一组15名个体中首次被描述。该综合征包括智力缺陷、严重言语障碍、共济失调步态、癫痫发作、婴儿期轻度肌张力减退伴喂养困难以及畸形特征。在此,我们报告了6名先证者中WDR26基因的6种新的杂合性新生致病性变异。患者的表型与最初的报道一致。一名患者表现出明显的肌张力减退,肌肉活检异常;这一发现值得进一步研究。必须密切监测步态,以发现任何肌肉骨骼或神经异常并促使进一步检查。在临床随访早期就应开始言语治疗和采用替代沟通方法,以改善语言及经口进食和饮水能力。