Expressive morphosyntax in boys with Fragile X syndrome with and without autism spectrum disorder

被引:25
作者
Estigarribia, Bruno [1 ]
Roberts, Joanne Erwick [1 ,2 ]
Sideris, John [3 ]
Price, Johanna [4 ]
机构
[1] Univ N Carolina, Div Speech & Hearing Sci, Chapel Hill, NC 27599 USA
[2] Univ N Carolina, Dept Pediat, Chapel Hill, NC 27599 USA
[3] FPG Child Dev Inst, Chapel Hill, NC USA
[4] Mississippi Univ Women, Dept Speech Language Pathol, Columbus, OH USA
关键词
Fragile X syndrome; morphosyntax; autism spectrum disorder; X-linked; language disorders; conversation; DOWN-SYNDROME; LANGUAGE IMPAIRMENT; GRAMMATICAL MORPHOLOGY; CHILDREN; SPEECH; SKILLS; MALES; TENSE; DELAY;
D O I
10.3109/13682822.2010.487885
中图分类号
R36 [病理学]; R76 [耳鼻咽喉科学];
学科分类号
100104 ; 100213 ;
摘要
Background: Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability, and the most common single gene disorder associated with autism. Language impairments in this disorder are well documented, but the nature and extent of syntactic impairments are still unclear. Aims: To compare the performance of boys with FXS with and without autism spectrum disorder on measures of verb (VM) and noun (NM) morphosyntax with that of typically developing boys of similar non-verbal mental ages. Methods & Procedures: Conversational samples were obtained from 33 boys with FXS with autism spectrum disorder (FXS-ASD), 35 boys with FXS and no ASD (FXS-O), and 46 typically developing boys (TD). Production of verbal and nominal morphosyntax was assessed separately in these two subdomains. A hierarchical linear model compared morphosyntactic scores in all groups after adjusting for non-verbal cognition, articulatory skill, and caregiver education. The model also tested interactions between group and morphosyntactic subdomain. Outcomes & Results: Boys with FXS in both groups scored lower than the TD boys on both measures. The FXS-O and the FXS-ASD groups did not differ on either composite measure. All covariates were significantly related to morphosyntactic scores. Conclusions & Implications: Part of the morphosyntactic impairment in FXS may be attributable to cognitive, environmental, and speech factors. However, it is clear that boys with FXS perform at levels lower than expected from differences in these extra-linguistic factors alone, across both the verb and the noun domains. Clinical interventions should therefore seek to address specific syntactic targets.
引用
收藏
页码:216 / 230
页数:15
相关论文
共 50 条
[31]   Joint attention performance in preschool-aged boys with autism or fragile X syndrome [J].
Thurman, Angela John ;
Dimachkie Nunnally, Amanda .
FRONTIERS IN PSYCHOLOGY, 2022, 13
[32]   Screening for Fragile X Syndrome Among Filipino Children with Autism Spectrum Disorder [J].
Angel Belle C. Dy ;
Lourdes Bernadette S. Tanchanco ;
Jenica Clarisse Y. Sy ;
Myla Dominicina Levantino ;
Randi J. Hagerman .
Journal of Autism and Developmental Disorders, 2023, 53 :4465-4473
[33]   Early Predictors of Later Expressive Language in Boys With Fragile X Syndrome [J].
Fielding-Gebhardt, Heather ;
Warren, Steven F. .
AJIDD-AMERICAN JOURNAL ON INTELLECTUAL AND DEVELOPMENTAL DISABILITIES, 2019, 124 (01) :11-24
[34]   Language Skills of Males with Fragile X Syndrome or Nonsyndromic Autism Spectrum Disorder [J].
Angela John Thurman ;
Andrea McDuffie ;
Randi J. Hagerman ;
Cynde K. Josol ;
Leonard Abbeduto .
Journal of Autism and Developmental Disorders, 2017, 47 :728-743
[35]   Autism Symptoms in Fragile X Syndrome [J].
Niu, Manman ;
Han, Ying ;
Dy, Angel Belle C. ;
Du, Junbao ;
Jin, Hongfang ;
Qin, Jiong ;
Zhang, Jing ;
Li, Qinrui ;
Hagerman, Randi J. .
JOURNAL OF CHILD NEUROLOGY, 2017, 32 (10) :903-909
[36]   Discourse skills of boys with fragile X syndrome in comparison to boys with Down syndrome [J].
Roberts, Joanne ;
Martin, Gary E. ;
Moskowitz, Lauren ;
Harris, Adrianne A. ;
Foreman, Jamila ;
Nelson, Lauren .
JOURNAL OF SPEECH LANGUAGE AND HEARING RESEARCH, 2007, 50 (02) :475-492
[37]   A Comparison of Pragmatic Language in Boys With Autism and Fragile X Syndrome [J].
Klusek, Jessica ;
Martin, Gary E. ;
Losh, Molly .
JOURNAL OF SPEECH LANGUAGE AND HEARING RESEARCH, 2014, 57 (05) :1692-1707
[38]   An Investigation of Narrative Ability in Boys with Autism and Fragile X Syndrome [J].
Hogan-Brown, Abigail L. ;
Losh, Molly ;
Martin, Gary E. ;
Mueffelmann, Deborah J. .
AJIDD-AMERICAN JOURNAL ON INTELLECTUAL AND DEVELOPMENTAL DISABILITIES, 2013, 118 (02) :77-94
[39]   Brief Report: Implementation of a Specific Carbohydrate Diet for a Child with Autism Spectrum Disorder and Fragile X Syndrome [J].
Barnhill, Kelly ;
Devlin, Morgan ;
Moreno, Hannah Taylor ;
Potts, Amy ;
Richardson, Wendy ;
Schutte, Claire ;
Hewitson, Laura .
JOURNAL OF AUTISM AND DEVELOPMENTAL DISORDERS, 2020, 50 (05) :1800-1808
[40]   Autism spectrum disorder in females with fragile X syndrome: a systematic review and meta-analysis of prevalence [J].
Marlborough, M. ;
Welham, A. ;
Jones, C. ;
Reckless, S. ;
Moss, J. .
JOURNAL OF NEURODEVELOPMENTAL DISORDERS, 2021, 13 (01)