Kantaputra Piranit Nik, Kapoor Seema, Verma Prashant, Intachai Worrachet, Ketudat Cairns James R
Center of Excellence in Medical Genetics Research, Chiang Mai University, Division of Pediatric Dentistry, Department of Orthodontics and Pediatric Dentistry, Faculty of Dentistry, Chiang Mai University, Chiang Mai, Thailand; DENTALAND CLINIC, Chiang Mai, Thailand.
Pediatrics Research & Genetic Lab, Department of Pediatrics, MAMC & Associated Lok Nayak Hospital, New Delhi, India.
Eur J Med Genet. 2018 Jul;61(7):372-375. doi: 10.1016/j.ejmg.2018.02.001. Epub 2018 Feb 7.
We report an Indian girl with split-hand/foot malformation (SHFM), sparse hair, and interrupted eyebrows, who carries a novel homozygous deletion c.695_697delACA in WNT10B. The variant is deduced to cause an in-frame deletion of Asn residue 232 (p.Asn232del). According to the protein model, this single amino acid deletion at the critical position in the protein structure is likely to severely affect the protein structure and function. This deletion is likely to lead decreased lifetime and make it unable to bind to its receptors and other ligands. The patient and all family members had normal bone density and they were not obese like some of the patients with WNT10B variants. Here we report a patient with SHFM6 who carried a novel WNT10B mutation. Sparse hair and interrupted eyebrows may be associated findings of SHFM6.
我们报告了一名患有手足裂畸形(SHFM)、头发稀疏和眉毛间断的印度女孩,她携带WNT10B基因中一个新的纯合缺失c.695_697delACA。该变异推断导致第232位天冬酰胺残基的框内缺失(p.Asn232del)。根据蛋白质模型,蛋白质结构关键位置的这一单氨基酸缺失可能严重影响蛋白质结构和功能。这种缺失可能导致寿命缩短,并使其无法与其受体及其他配体结合。该患者及其所有家庭成员骨密度正常,且不像一些携带WNT10B变异的患者那样肥胖。在此我们报告一名携带新型WNT10B突变的SHFM6患者。头发稀疏和眉毛间断可能是SHFM6的相关表现。