Kalscheuer Vera M, FitzPatrick David, Tommerup Niels, Bugge Merete, Niebuhr Erik, Neumann Luitgard M, Tzschach Andreas, Shoichet Sarah A, Menzel Corinna, Erdogan Fikret, Arkesteijn Ger, Ropers Hans-Hilger, Ullmann Reinhard
Max-Planck-Institute for Molecular Genetics, Ihnestrasse 73, 14195, Berlin, Germany.
Hum Genet. 2007 May;121(3-4):501-9. doi: 10.1007/s00439-006-0284-0. Epub 2007 Jan 9.
We report on three unrelated mentally disabled patients, each carrying a de novo balanced translocation that truncates the autism susceptibility candidate 2 (AUTS2) gene at 7q11.2. One of our patients shows relatively mild mental retardation; the other two display more profound disorders. One patient is also physically disabled, exhibiting urogenital and limb malformations in addition to severe mental retardation. The function of AUTS2 is presently unknown, but it has been shown to be disrupted in monozygotic twins with autism and mental retardation, both carrying a translocation t(7;20)(q11.2;p11.2) (de la Barra et al. in Rev Chil Pediatr 57:549-554, 1986; Sultana et al. in Genomics 80:129-134, 2002). Given the overlap of this autism/mental retardation (MR) phenotype and the MR-associated disorders in our patients, together with the fact that mapping of the additional autosomal breakpoints involved did not disclose obvious candidate disease genes, we ascertain with this study that AUTS2 mutations are clearly linked to autosomal dominant mental retardation.
我们报告了三名无血缘关系的智力残疾患者,他们每人都携带一种新生的平衡易位,该易位在7q11.2处截断了自闭症易感候选基因2(AUTS2)。我们的一名患者表现出相对轻度的智力迟钝;另外两名患者则表现出更严重的障碍。一名患者还存在身体残疾,除了严重智力迟钝外,还伴有泌尿生殖系统和肢体畸形。目前尚不清楚AUTS2的功能,但已发现在患有自闭症和智力迟钝的同卵双胞胎中,该基因因携带t(7;20)(q11.2;p11.2)易位而受到破坏(de la Barra等人,《智利儿科学杂志》57:549 - 554,1986年;Sultana等人,《基因组学》80:129 - 134,2002年)。鉴于我们患者中这种自闭症/智力迟钝(MR)表型与MR相关疾病的重叠,以及所涉及的额外常染色体断点定位未揭示明显的候选疾病基因这一事实,我们通过本研究确定AUTS2突变与常染色体显性智力迟钝明显相关。