Psychometric Study of the Social Responsiveness Scale in Phelan-McDermid Syndrome

被引：19

作者：

Gergoudis, Kellie ^{[1
]}

Weinberg, Alan ^{[2
]}

Templin, Jonathan ^{[3
]}

Farmer, Cristan ^{[4
]}

Durkin, Alison ^{[4
,5
]}

Weissman, Jordana ^{[5
]}

Siper, Paige ^{[5
]}

Foss-Feig, Jennifer ^{[5
]}

Trelles, Maria del Pilar ^{[5
]}

Bernstein, Jonathan A. ^{[6
]}

Buxbaum, Joseph D. ^{[5
,7
,8
]}

Berry-Kravis, Elizabeth ^{[9
,10
,11
]}

Powell, Craig M. ^{[12
]}

Sahin, Mustafa ^{[13
,14
]}

Soorya, Latha ^{[15
]}

Thurm, Audrey ^{[16
]}

Kolevzon, Alexander ^{[5
]}

机构：

[1] Icahn Sch Med Mt Sinai, New York, NY 10029 USA

[2] Icahn Sch Med Mt Sinai, Dept Populat Hlth Sci & Policy, New York, NY 10029 USA

[3] Univ Kansas, Dept Psychol & Res Educ, Lawrence, KS 66045 USA

[4] NIMH, Pediat & Dev Neurosci Branch, NIH, Bethesda, MD 20892 USA

[5] Icahn Sch Med Mt Sinai, Dept Psychiat, Seaver Autism Ctr, New York, NY 10029 USA

[6] Stanford Univ, Dept Pediat, Sch Med, Stanford, CA 94305 USA

[7] Icahn Sch Med Mt Sinai, Dept Genet & Genom Sci, New York, NY 10029 USA

[8] Icahn Sch Med Mt Sinai, Dept Neurosci, New York, NY 10029 USA

[9] Rush Univ, Med Ctr, Dept Pediat, Chicago, IL 60612 USA

[10] Rush Univ, Med Ctr, Dept Neurol Sci, Chicago, IL 60612 USA

[11] Rush Univ, Med Ctr, Dept Biochem, Chicago, IL 60612 USA

[12] Univ Alabama Birmingham, Dept Neurobiol, Birmingham, AL USA

[13] Harvard Med Sch, Boston Childrens Hosp, FM Kirby Neurobiol Ctr, Boston, MA 02115 USA

[14] Harvard Med Sch, Boston Childrens Hosp, Dept Neurol, Boston, MA 02115 USA

[15] Rush Univ, Med Ctr, Dept Psychiat, Chicago, IL 60612 USA

[16] Icahn Sch Med Mt Sinai, Dept Pediat, New York, NY 10029 USA

来源：

AUTISM RESEARCH | 2020年 / 13卷 / 08期

关键词：

autism spectrum disorder; Phelan-McDermid syndrome; behavioral measures; Item Response Theory; intellectual disability; FRAGILE-X-SYNDROME; AUTISM; CHILDREN; PSYCHOPATHOLOGY; INSTRUMENTS; PHENOTYPE; BEHAVIOR; QUALITY;

D O I：

10.1002/aur.2299

中图分类号：

B84 [心理学]; C [社会科学总论]; Q98 [人类学];

学科分类号：

03 ; 0303 ; 030303 ; 04 ; 0402 ;

摘要：

The Social Responsiveness Scale-2 (SRS-2) is a quantitative measure used to characterize symptoms of autism spectrum disorder (ASD). However, research suggests that SRS-2 scores are significantly influenced by language ability and intellectual disability (ID). Efforts to refine the SRS-2 by Sturm, Kuhfeld, Kasari, and Mccracken [Journal of Child Psychology and Psychiatry, 58(9), 1053-1061] yielded a shortened form, yet its psychometric properties in populations with severe ID remain unknown. This study aims to examine the psychometric properties of the SRS-2 in Phelan-McDermid syndrome (PMS), a genetic condition associated with ASD and ID, thereby guiding score interpretation in this population and future development of targeted scales. Analyses, including Item Response Theory (IRT), were conducted on a sample of individuals with PMS (n = 91) recruited at six sites nationally. Psychometric properties evaluated include measures of reliability (internal consistency, test-retest reliability) and validity (structural, construct, content). While both SRS-2 forms are reliable, the shortened SRS-2 shows superior validity to the full SRS-2 for measuring ASD symptoms in PMS. On IRT analysis, the shortened SRS-2 shows excellent discrimination and precisely evaluates respondents across a wide range of ASD symptomatology but interpretation is limited by uncertain content validity and small sample size. The shortened SRS-2 shows some promise for use in PMS, but future refinements and additions are needed to develop items that are tailored to identify ASD in children with severe ID and specifically PMS. Lay Summary This study determined that a shortened form of the Social Responsiveness Scale, Second Edition (SRS-2) shows both promise and limitations for the characterization of autism symptomatology in individuals with Phelan-McDermid syndrome (PMS), a population characterized by intellectual disability (ID). Caution should be used when interpreting SRS-2 scores in individuals with ID and future research should modify existing items and develop new items to improve the SRS-2's ability to accurately characterize autism symptomatology in PMS.

引用

页码：1383 / 1396

页数：14

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