Language across the Lifespan in Fragile X Syndrome: Characteristics and Considerations for Assessment.

Fragile X syndrome intellectual disability language assessment

Journal

Brain sciences
ISSN: 2076-3425
Titre abrégé: Brain Sci
Pays: Switzerland
ID NLM: 101598646

Informations de publication

Date de publication:
04 Apr 2020
Historique:
received: 03 03 2020
revised: 27 03 2020
accepted: 30 03 2020
entrez: 9 4 2020
pubmed: 9 4 2020
medline: 9 4 2020
Statut: epublish

Résumé

While it is widely acknowledged that language development is delayed for the majority of individuals with fragile X syndrome (FXS), there has been limited research into how best to assess this area. This study aimed to deepen the understanding of standardized language assessment in FXS by addressing the three following objectives: (1) Examine the feasibility and validity of widely-used, standardized assessments in participants with FXS; (2) describe linguistic and cognitive profiles for a large sample of individuals with FXS; and (3) Compare results obtained from objective testing in clinic to those obtained using caregiver report. Results indicate that previous results indicating strong correlations between cognition and language results hold true across a wide range of ages as well as across multiple assessments, with an exception in very young children. Caregiver report tended to give lower estimates of language ability than what was found using an objectively administered assessment. Appropriate assessments remain difficult to find as a significant percentage of individuals scored at floor when scaled scores were calculated. Further, a sub-group of participants were coded for behavioral response to testing demands, the majority being able to complete a standardized assessment. These results speak to the need for assessments that provide a wider range of items so individuals can both achieve a valid score and demonstrate progress in their attainment of language skills.

Identifiants

pubmed: 32260354
pii: brainsci10040212
doi: 10.3390/brainsci10040212
pmc: PMC7226519
pii:
doi:

Types de publication

Journal Article

Langues

eng

Subventions

Organisme : NCBDD CDC HHS
ID : U01 DD001185
Pays : United States
Organisme : CDC HHS
ID : 1 U01 DD001186-01
Pays : United States

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Auteurs

Anne Hoffmann (A)

Departments of Pediatrics and Communication Disorders, Rush University Medical Center, Chicago, IL 60612, USA.

Angel Wang (A)

Department of Pediatrics, Rush University Medical Center, Chicago, IL 60612, USA.

Natalie Berger (N)

Department of Psychiatry, Rush University Medical Center, Chicago, IL 60612, USA.

Lisa Cordeiro (L)

Division of Developmental Pediatrics, Department of Pediatrics, University of Colorado School of Medicine, Aurora, CO 80045, USA.

Rebecca Shaffer (R)

Division of Child and Adolescent Psychiatry, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.

Nicole Tartaglia (N)

Division of Developmental Pediatrics, Department of Pediatrics, University of Colorado School of Medicine, Aurora, CO 80045, USA.

Craig Erickson (C)

Division of Child and Adolescent Psychiatry, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.

Elizabeth Berry-Kravis (E)

Departments of Pediatrics, Neurological Sciences, Biochemistry, Rush University Medical Center, Chicago, IL 60612, USA.

Classifications MeSH