Strømme Petter, Mangelsdorf Marie E, Scheffer Ingrid E, Gécz Jozef
Department of Cytogenetics and Molecular Genetics, Women's and Children's Hospital, North Adelaide, SA 5006, Australia.
Brain Dev. 2002 Aug;24(5):266-8. doi: 10.1016/s0387-7604(02)00079-7.
Clinical data from 50 mentally retarded (MR) males in nine X-linked MR families, syndromic and non-specific, with mutations (duplication, expansion, missense, and deletion mutations) in the Aristaless related homeobox gene, ARX, were analysed. Seizures were observed with all mutations and occurred in 29 patients, including one family with a novel myoclonic epilepsy syndrome associated with the missense mutation. Seventeen patients had infantile spasms. Other phenotypes included mild to moderate MR alone, or with combinations of dystonia, ataxia or autism. These data suggest that mutations in the ARX gene are important causes of MR, often associated with diverse neurological manifestations.
分析了来自9个X连锁智力迟钝(MR)男性家系中50名男性患者的临床数据,这些家系有综合征型和非特异性型,其无尾相关同源盒基因(ARX)存在突变(重复、扩增、错义及缺失突变)。所有突变类型均观察到癫痫发作,29名患者出现癫痫,其中一个家系的错义突变与一种新型肌阵挛癫痫综合征相关。17名患者有婴儿痉挛症。其他表型包括单独的轻度至中度智力迟钝,或伴有肌张力障碍、共济失调或自闭症的组合。这些数据表明,ARX基因突变是智力迟钝的重要原因,常伴有多种神经学表现。