A novel WNT10A variant impairs the homeostasis of alveolar bone mesenchymal stem cells.

IF 2.9 3区医学 Q1 DENTISTRY, ORAL SURGERY & MEDICINE Oral diseases Pub Date : 2025-01-01 Epub Date: 2024-06-09 DOI:10.1111/odi.15032

Bichen Lin, Haochen Liu, Hangbo Liu, Lanxin Su, Kai Sun, Hailan Feng, Yang Liu, Miao Yu, Dong Han

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Abstract

Objectives: To explore the influence of a novel WNT10A variant on bone mineral density, proliferation, and osteogenic differentiation capacities of alveolar bone mesenchymal stem cells in humans.

Subjects and methods: Whole-exome sequencing and Sanger sequencing were utilized to detect gene variants in a family with non-syndromic tooth agenesis (NSTA). The panoramic mandibular index was calculated on the proband with WNT10A variant and normal controls to evaluate bone mineral density. Alveolar bone mesenchymal stem cells from the proband with a novel WNT10A variant and normal controls were isolated and cultured, then proliferation and osteogenic differentiation capacities were evaluated and compared.

Results: We identified a novel WNT10A pathogenic missense variant (c.353A > G/p. Tyr118Cys) in a family with NSTA. The panoramic mandibular index of the proband implied a reduction in bone mineral density. Moreover, the proliferation and osteogenic differentiation capacities of alveolar bone mesenchymal stem cells from the proband with WNT10A Tyr118Cys variant were significantly decreased.

Conclusions: Our findings broaden the spectrum of WNT10A variants in patients with non-syndromic oligodontia, suggest an association between WNT10A and the proliferation and osteogenic differentiation of alveolar bone mesenchymal stem cells, and demonstrate that WNT10A is involved in maintaining jaw bone homeostasis.

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一种新型 WNT10A 变体会损害牙槽骨间充质干细胞的稳态。

目的探讨新型 WNT10A 变异对人类牙槽骨间充质干细胞的骨矿密度、增殖和成骨分化能力的影响：利用全外显子组测序和桑格测序检测一个非综合征性牙齿缺失（NSTA）家族的基因变异。计算了WNT10A变异体原型和正常对照组的下颌骨全景指数，以评估骨矿物质密度。从WNT10A新型变异体的疑似患者和正常对照组中分离并培养牙槽骨间充质干细胞，然后对其增殖和成骨分化能力进行评估和比较：结果：我们在一个NSTA家族中发现了一个新型WNT10A致病性错义变体（c.353A > G/p.该患者的下颌骨全景指数显示其骨矿物质密度降低。此外，来自 WNT10A Tyr118Cys 变异原患者的牙槽骨间充质干细胞的增殖和成骨分化能力显著下降：我们的研究结果拓宽了非综合征少骨症患者中WNT10A变异的范围，表明WNT10A与牙槽骨间充质干细胞的增殖和成骨分化之间存在关联，并证明WNT10A参与维持颌骨的稳态。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Oral diseases 医学-牙科与口腔外科

CiteScore

7.60

自引率

5.30%

发文量

325

审稿时长

4-8 weeks

期刊介绍： Oral Diseases is a multidisciplinary and international journal with a focus on head and neck disorders, edited by leaders in the field, Professor Giovanni Lodi (Editor-in-Chief, Milan, Italy), Professor Stefano Petti (Deputy Editor, Rome, Italy) and Associate Professor Gulshan Sunavala-Dossabhoy (Deputy Editor, Shreveport, LA, USA). The journal is pre-eminent in oral medicine. Oral Diseases specifically strives to link often-isolated areas of dentistry and medicine through broad-based scholarship that includes well-designed and controlled clinical research, analytical epidemiology, and the translation of basic science in pre-clinical studies. The journal typically publishes articles relevant to many related medical specialties including especially dermatology, gastroenterology, hematology, immunology, infectious diseases, neuropsychiatry, oncology and otolaryngology. The essential requirement is that all submitted research is hypothesis-driven, with significant positive and negative results both welcomed. Equal publication emphasis is placed on etiology, pathogenesis, diagnosis, prevention and treatment.