MacLean J E, Teshima I E, Szatmari P, Nowaczyk M J
Department of Pathology and Molecular Medicine, McMaster University, and Hamilton Health Sciences Corporation, Ontario, Canada.
Am J Med Genet. 2000 Feb 28;90(5):382-5.
Ring chromosome 22 has been described in over 50 cases. A characteristic phenotype has not been fully delineated; however, long face, thick eyebrows, 2-3 toe syndactyly, mental retardation, adequate somatic growth and the absence of major malformations are noted in many cases. An 11-year-old boy with ring chromosome 22 and 46,XY,r(22)(p11.31-q13.31 approximately q13.33) karyotype presented with global developmental delay, autistic disorder, and dolichocephaly, apparently low-set and large ears, midface hypoplasia, and 2-3 toe syndactyly. This is the second report of a ring chromosome 22 with autistic disorder. There appears to be an association between abnormalities of chromosome 22, including r(22), and autistic disorder; however, this occurrence may be a result of the association of autistic disorder with mental retardation rather than specifically due to r(22). The physical findings in this case also suggest that ring chromosome 22 causes a subtle but distinct phenotype which has previously been proposed.
已有超过50例关于22号环状染色体的报道。其特征性表型尚未完全明确;然而,许多病例中可见长脸、浓眉、2-3趾并指、智力发育迟缓、体格生长正常且无重大畸形。一名11岁男孩,核型为46,XY,r(22)(p11.31-q13.31约q13.33),表现为全面发育迟缓、自闭症谱系障碍、长头畸形,明显低位且大的耳朵,面中部发育不全以及2-3趾并指。这是第二例伴有自闭症谱系障碍的22号环状染色体的报道。22号染色体异常(包括r(22))与自闭症谱系障碍之间似乎存在关联;然而,这种情况可能是自闭症谱系障碍与智力发育迟缓相关联的结果,而非特别归因于r(22)。该病例的体格检查结果还表明,22号环状染色体导致了一种先前已被提出的细微但独特的表型。