Specificity: A Phenotypic Comparison of Communication-Relevant Domains Between Youth With Down Syndrome and Fragile X Syndrome.
Specificity: A Phenotypic Comparison of Communication-Relevant Domains Between Youth With Down Syndrome and Fragile X Syndrome.
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DOI：
10.3389/fgene.2018.00424
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发表时间：
2018
影响因子：
3.7
通讯作者：
Abbeduto L
中科院分区：
文献类型：
--
作者：
Del Hoyo Soriano L;Thurman AJ;Abbeduto L
尽管都存在智力障碍 (ID)，但越来越多的文献记录了唐氏综合症 (DS) 和脆性 X 综合症 (FXS) 之间重要的表型差异。然而，这些结论是基于跨研究的综合，每项研究通常仅包括有限数量的构建的测量，并且具有不同的参与者特征。关于特定表型的更坚定的结论需要对参与者进行单一全面的多领域评估，其中综合症组在实际年龄（CA）和认知功能上非常匹配。目前的研究旨在填补这一空白，评估了 30 名患有 DS 的男女青少年和 39 名患有 FXS 的男性的结构语言技能、错误信念理解以及语用和行为困难等与沟通相关的几个重要认知和行为领域，并匹配了 CA 和非语言 (NV) 认知。对 NV 认知进行统计控制后，我们没有发现表达性和接受性结构语言或错误信念理解方面存在显着的综合征差异。相比之下，与患有 FXS 的男性相比，患有 DS 的参与者表现出较少的刻板语言和较少的行为困难。描述了目标域之间的综合征内关联。最后，女性 DS 患者在几乎所有结构语言领域的受损程度均低于男性 DS，而在 NV 认知、错误信念理解、语用或行为方面未观察到显着的性别相关差异。讨论了研究结果的临床和方法学意义。
Despite the shared presence of an intellectual disability (ID), there is a growing literature documenting important phenotypic differences between Down syndrome (DS) and fragile X syndrome (FXS). These conclusions, however, are based on a synthesis across studies, each of which typically includes only measures of a limited number of constructs, and with differing participant characteristics. Firmer conclusions regarding specific phenotypes require a single comprehensive multi-domain assessment of participants with the syndrome groups being well matched on chronological age (CA) and cognitive functioning. The current study was designed to fill this gap by assessing several important cognitive and behavioral domains relevant to communication, such as: structural language skills, false belief understanding, as well as pragmatics and behavioral difficulties, in 30 adolescents of both sexes with DS and 39 males with FXS, matched on CA and nonverbal (NV) cognition. After statistically controlling for NV cognition, we did not find significant syndrome differences in expressive and receptive structural language or false belief understanding. In contrast, participants with DS displayed less stereotyped language and fewer behavioral difficulties compared to males with FXS. Within-syndrome associations among the targeted domains are described. Finally, females with DS were less impaired than males with DS in almost all structural language domains, whereas no significant sex-related differences were observed in NV cognition, false belief understanding, pragmatics, or behavior. Clinical and methodological implications of the findings are discussed.
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影响因子：
3.1
作者：
Channell, Marie Moore;Thurman, Angela John;Abbeduto, Leonard
通讯作者：
Abbeduto, Leonard
DOI：
10.1111/j.2517-6161.1995.tb02031.x
发表时间：
1995-01-01
影响因子：
5.8
作者：
BENJAMINI, Y;HOCHBERG, Y
通讯作者：
HOCHBERG, Y
影响因子：
3
作者：
Channell, Marie Moore;McDuffie, Andrea S.;Abbeduto, Leonard
通讯作者：
Abbeduto, Leonard
DOI：
10.1352/0895-8017(2006)111
发表时间：
2006-01-01
期刊：
AMERICAN JOURNAL ON MENTAL RETARDATION
影响因子：
--
作者：
Hastings, RP;Daley, D;Beck, A
通讯作者：
Beck, A
DOI：
10.1044/1092-4388(2001/016
发表时间：
2001-02-01
影响因子：
2.6
作者：
Berglund, E;Eriksson, M;Johansson, I
通讯作者：
Johansson, I