Cognitive, language and social-cognitive skills of individuals with fragile X syndrome with and without autism
Cognitive, language and social-cognitive skills of individuals with fragile X syndrome with and without autism
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DOI：
10.1111/j.1365-2788.2006.00803.x
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发表时间：
2006-07-01
影响因子：
3.6
通讯作者：
Schroeder, S.
中科院分区：
文献类型：
--
作者：
Lewis, P.;Abbeduto, L.;Schroeder, S.
背景：目前尚不清楚脆性X染色体综合征（FXS）和自闭症共病者是否代表一个不同的FXS亚型；在研究中，与仅患有FXS的儿童相比，患有FXS和自闭症的幼儿中特别严重的认知延迟是否会持续到青春期和青年期；以及FXS患者的自闭症是否是“真正的自闭症”，即反映了与特发性自闭症相同的潜在问题。方法将FXS合并自闭症的青少年和青壮年（n = 10）与仅FXS的青少年和青壮年（n = 44）的非语言智商进行比较。然后，我们创建了一个仅患有FXS的子样本，将非语言智商、心理年龄和性别（n = 21）与同时患有FXS和自闭症的子样本相匹配。我们比较了两组的表达性语言、接受性语言（词汇、语法形态和句法模式）和心理理论任务。结果FXS合并自闭症组的非语言智商低于单纯FXS组。在接受性语言三项测试和心理理论测试中，FXS合并自闭症的受试者表现不如认知能力和性别匹配的FXS合并自闭症的受试者；两组在表达性语言测试上没有差异。我们的研究结果支持了这样一种观点，即那些同时患有FXS和自闭症的人代表了一种独特的FXS亚型，即使在控制了他们相对于只有FXS的人较低的非语言智商的情况下，他们在接受性语言和心理理论方面的损害也更大。同时患有FXS和自闭症的患者在青春期和青年期会出现更大的认知障碍；而FXS患者的自闭症似乎与特发性自闭症相同。
Background It is not known whether those with co-morbid fragile X syndrome (FXS) and autism represent a distinct subtype of FXS; whether the especially severe cognitive delays seen in studies of young children with co-morbid FXS and autism compared with those with only FXS continue into adolescence and young adulthood; and whether autism in those with FXS is 'true autism', i.e. reflects the same underlying problems as idiopathic autism.Method We compared the non-verbal IQ of adolescents and young adults with co-morbid FXS and autism (n = 10) with those with only FXS (n = 44). We then created a subsample of those with FXS only, matched on non-verbal IQ, mental age and gender (n = 21) to the subsample of those with co-morbid FXS and autism. We compared the two groups on measures of expressive language, receptive language (lexical, grammatical morphology and syntactic patterns), and a theory of mind task.Results Those with co-morbid FXS and autism had lower non-verbal IQs than those with only FXS. The participants with co-morbid FXS and autism did not perform as well as the cognitive ability- and gender-matched participants with only FXS on the three measures of receptive language or the theory of mind task; there were no differences on the expressive language measure.Conclusions Our findings support the notion that those with co-morbid FXS and autism represent a distinct subtype of FXS, with more impairment in receptive language and theory of mind even when controlling for their lower non-verbal IQ relative to those with only FXS. The greater cognitive impairments observed in those with co-morbid FXS and autism continues into adolescence and young adulthood; and the autism seen in those with FXS appears to be the same as idiopathic autism.