Wang H, Zhao S, Zhao W, Feng G, Jiang S, Liu W, Li S, Xue H, He L
Shanghai Institute of Physiology, Chinese Academy of Sciences, Shanghai.
Am J Med Genet. 2000 Jan 31;90(3):193-8.
We report on rare, heritable, permanent tooth agenesis in a large Chinese kindred. The congenital absence of permanent teeth except the first and second accessory teeth was observed in 52 individuals through six successive generations in the kindred comprising 328 members. Clinical assessments were carried out, and inheritance mode and spousal influence of the anomaly on their offspring were analyzed. Consequently, the anomaly was transmitted in an autosomal dominant fashion with incomplete penetrance (P = 0.88), and no significant clinical manifestations other than the oligodontia were found. A geographical or environmental effect on the affected individuals was obviously eliminated, because any who are related to the kindred but live under the same conditions are fully healthy. The disorder we describe, therefore, differs from any previously reported oligodontia/anodontia syndromes. The oligodontia ranged from a few teeth to the whole set of teeth, and usually occurred at a period from age 7 or 8 years, the time when primary teeth are normally replaced by permanent teeth, to the forties. Roentgenography of the affected persons indicated that only the first and/or second accessory teeth with tooth buds developed as permanent teeth. In fact, the diphyodontic germination sometimes occurred in the oral cavity of the affected individuals.
我们报告了一个大型中国家系中罕见的、可遗传的恒牙先天缺失情况。在这个由328名成员组成的家系中,经过连续六代观察,发现52人除第一和第二副磨牙外恒牙先天性缺失。我们进行了临床评估,并分析了该异常的遗传模式以及配偶对其后代的影响。结果显示,该异常以常染色体显性方式遗传,外显率不完全(P = 0.88),且除少牙症外未发现其他明显临床表现。对患病个体而言,地理或环境影响显然已被排除,因为任何与该家系有亲缘关系但生活在相同条件下的人都完全健康。因此,我们所描述的这种病症与之前报道的任何少牙症/无牙症综合征都不同。少牙症的范围从几颗牙齿到整套牙齿,通常发生在7、8岁(乳牙正常替换为恒牙的时期)到40多岁之间。对患病个体的X线检查表明,只有带有牙胚的第一和/或第二副磨牙发育为恒牙。实际上,患病个体口腔中有时会出现双牙萌出的情况。