Mounoud R L, Klein D, Bettschart W, Cabrol C
J Genet Hum. 1976 Dec;24(4):297-335.
A clinical and cytogenetic investigation carried out in a special institution for mentally retarded patients revealed 82 cases of oligophrenia, amongst whom were found 56 normal karyotypes (68.3%). Out of 25 karyotypes with chromosome anomalies or variants there were 18 cases of trisomy 21 and 7 others: one case of mosaicism with balanced translocation, 46,XX/46,XX,6p+,17q-; one case of partial trisomy, 46,XX,11q+; one case of pericentric inversion, 46,XY,inv(1) (p13,q21); one case with 8% chromosome breaks; three cases of marker chromosomes, of which one was of karyotype 46,XX,1qh+, and two (oligophrenic sisters) 46,XX,21p+. Moreover, there was an interesting case of testicular feminisation in a 9-year-old girl with karyotype 46,XY. The authors' results corroborate those obtained in several important previous studies based on much larger numbers of patients. Amongst the 56 cases where the karyotype was shown to be normal, there were 15 for whom a probably exogenic cause of the oligophrenia could be established, occurring mainly during the perinatal period. The authors were also able to confirm that the genetic factor plays an important role in the incidence of mental retardation, since in 22 examined patients, i.e. 26.8% of all cases, the condition was of familial type. Some interesting observations of idiopathic oligophrenia are reported, as well as several cases with well-known syndromes (Crouzon's and Cornelia de Lange's syndromes, hypothyroidism). Two cases of incest between father and daughter, which had produced children with serious oligophrenia associated, in one case, with deaf-mutism, microphthalmia, microcephaly and sclerocornea, are also discussed. The data show that mental retardation can frequently have a genetic cause, either of mendelian, chromosomal or multifactorial origin.
在一家针对智障患者的特殊机构中进行的一项临床和细胞遗传学调查显示,有82例智力低下患者,其中56例核型正常(68.3%)。在25例有染色体异常或变异的核型中,有18例21三体,另外7例分别为:1例嵌合型伴平衡易位,核型为46,XX/46,XX,6p+,17q-;1例部分三体,核型为46,XX,11q+;1例臂间倒位,核型为46,XY,inv(1) (p13,q21);1例有8%的染色体断裂;3例标记染色体,其中1例核型为46,XX,1qh+,2例(智障姐妹)核型为46,XX,21p+。此外,还有1例9岁女孩的睾丸女性化病例,其核型为46,XY。作者的结果证实了先前基于更多患者数量的几项重要研究所得出的结果。在56例核型显示正常的病例中,有15例可能确定了智力低下的外源性病因,主要发生在围产期。作者还能够证实遗传因素在智力低下的发病率中起重要作用,因为在22例接受检查的患者中,即所有病例的26.8%,病情为家族性类型。报告了一些关于特发性智力低下的有趣观察结果,以及几例患有已知综合征(克鲁宗综合征和科妮莉亚·德·朗格综合征、甲状腺功能减退症)的病例。还讨论了2例父女乱伦病例,所生孩子患有严重智力低下,其中1例伴有聋哑、小眼症、小头畸形和角膜硬化症。数据表明,智力低下常常有遗传原因,其遗传方式可为孟德尔遗传、染色体遗传或多因素遗传。