The association between WNT10A variants and dental development in patients with isolated oligodontia
The association between WNT10A variants and dental development in patients with isolated oligodontia
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DOI：
10.1038/ejhg.2016.117
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发表时间：
2017-01-01
影响因子：
5.2
通讯作者：
Ongkosuwito, Edwin M.
中科院分区：
文献类型：
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作者：
Dhamo, Brunilda;Fennis, Willem;Ongkosuwito, Edwin M.
在这项研究中，我们的目的是确定WNT10A变异对少牙患者牙齿发育的影响。43人(25名男孩和18名女孩)符合这项研究的条件。每颗牙的发育阶段均采用Demirkin方法进行评估。在没有相应牙齿的情况下，应用回归方程计算牙龄。确定所有象限内每颗现牙的牙根长度与牙冠长度的比值。所有患者均由临床遗传学家进行体格检查，并对WNT10A基因进行DNA分析。采用线性回归模型分析WNT10A基因变异与牙龄的关系。同样的分析被应用于研究WNT10A变异与每颗现有牙齿的牙根伸长之间的关联。采用一元有序回归模型分析WNT10A基因变异与上下颌牙齿发育的关系。36例(84%)患者检测到WNT10A变异，其中6例患者表现出明显的外胚层特征。WNT10A变异患者的牙龄比无变异患者低1.50岁(95%可信区间：-2.59，-0.42)至1.96岁(95%可信区间：-3.76，-0.17)。WNT10A变异者上颌尖牙、上颌第二磨牙和下颌第二磨牙的发育较无变异者有统计学意义的延迟。WNT10A变异体对牙齿发育的影响随着无意义c.(321C>；A p.(C107*))变异体的存在和缺失牙齿的数量而增加。
In this study we aimed to determine the effect of WNT10A variants on dental development in patients with oligodontia. Forty-three (25 boys and 18 girls) individuals were eligible for this study. Stage of development for each present tooth was assessed using the Demirjian method. In case no corresponding tooth was present, regression equations were applied for dental age to be calculated. The ratio between length of root and length of crown was ascertained for each present tooth in all quadrants. All patients were physically examined by a clinical geneticist and DNA analysis of the WNT10A gene was performed. Linear regression models were applied to analyze the association between WNT10A variants and dental age. The same analysis was applied to study the association between WNT10A variants and root elongation for each present tooth. One ordinal regression model was applied to analyze the association between WNT10A variants and development of present maxillary and mandibular teeth. Thirty-six (84%) patients were detected with WNT10A variants of which six patients displayed evident ectodermal features. Dental age was 1.50 (95% confidence interval (CI): -2.59, -0.42) to 1.96 (95% CI: -3.76, -0.17) years lower in patients with WNT10A variants compared with patients without variants. The development of maxillary canine, maxillary second molar and mandibular second molar was statistically significantly delayed in patients with WNT10A variants compared with patients without variants. The impact of WNT10A variants on dental development increases with presence of the nonsense c. (321C>A p.(C107*)) variant and the number of missing teeth.