Cited by 41Genetics and Neurodevelopmental DisordersLanguage Development and DisordersFamily and Disability Support Research

A Spoken-Language Intervention for School-Aged Boys With Fragile X Syndrome

Open access
McDuffie, Andrea, Machalicek, Wendy, Bullard, Lauren, Nelson, Sarah, Mello, Melissa, Tempero-Feigles, Robyn, Castignetti, Nancy, Abbeduto, Leonard·2016·American Journal on Intellectual and Developmental Disabilities

Abstract

Using a single case design, a parent-mediated spoken-language intervention was delivered to three mothers and their school-aged sons with fragile X syndrome, the leading inherited cause of intellectual disability. The intervention was embedded in the context of shared storytelling using wordless picture books and targeted three empirically derived language-support strategies. All sessions were implemented through distance videoteleconferencing. Parent education sessions were followed by 12 weekly clinician coaching and feedback sessions. Data were collected weekly during independent homework and clinician observation sessions. Relative to baseline, mothers increased their use of targeted strategies, and dyads increased the frequency and duration of story-related talking. Generalized effects of the intervention on lexical diversity and grammatical complexity were observed. Implications for practice are discussed.

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  • Expressive Language Sampling has excellent test-retest validity after weeks and has been validated against the expressive language subtest of the Vineland Adaptive Behavior Scales.

    “ELS has excellent test and retest validity after weeks, and it has been validated against the expressive language subtest of the VABS94.”

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Research works printed but not linked: 3

  1. Brown W Hagerman R Hagerman P The molecular biology of the fragile X mutation Fragile X Syndrome: Diagnosis, Treatment and Research 2002 110 135 The Johns Hopkins University Press Baltimore, MD 3rd ed.
  2. Roberts J Mirrett P Burchinal M Receptive and expressive communication development of young males with fragile X syndrome American Journal on Mental Retardation 2001 106 216 230 11389664
  3. Zucker T Justice L Piasta S Kaderavek J Preschool teachers’ literal and inferential questions and children’s responses during whole-class shared reading Early Childhood Research Quarterly 2010 25 65 83

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Authors
McDuffie, A., Machalicek, W., Bullard, L., Nelson, S., Mello, M., Tempero-Feigles, R., Castignetti, N., & Abbeduto, L.
Affiliations
  • University of California, Davis (US)
  • University of Oregon (US)
  • Lauren Bullard, Sarah Nelson, Melissa Mello, Robyn Tempero-Feigles, Nancy Castignetti, and Leonard Abbeduto, University of California–Davis, Department of Psychiatry and Behavioral Sciences, MIND Institute, Sacramento, California.
Corresponding author
McDuffie, Andrea
Venue
American Journal on Intellectual and Developmental Disabilities
Published
2016-05-01
Volume
121
Issue
3
Pages
236-265
ISSN
  • 1944-7558 (ISSN-L)
  • 1944-7515
OpenAlex
W2343643553
Cited by
41
References
83
Citations by year
2026: 2 · 2025: 1 · 2024: 2 · 2023: 3 · 2022: 6 · 2021: 6 · 2020: 4 · 2019: 4 · 2018: 3 · 2017: 8 · 2016: 2
FWCI
4.58
Type
journal article
Language
en
Open access
Locations
  • American Journal on Intellectual and Developmental Disabilities (publishedVersion)
  • PubMed (publishedVersion)
  • PubMed Central (acceptedVersion)
  • eScholarship (California Digital Library) (submittedVersion)
Funders
Eunice Kennedy Shriver National Institute of Child Health and Human Development (U54HD079125)
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
  • Social Sciences › Psychology › Developmental and Educational Psychology › Language Development and Disorders
  • Social Sciences › Psychology › Clinical Psychology › Family and Disability Support Research
Keywords
fragile X syndrome, parent-mediated intervention, wordless picture books, language support strategies, lexical diversity, grammatical complexity, intellectual disability, mothers
MeSH terms
Adult, Child, Female, Fragile X Syndrome, Humans, Language Therapy, Male, Mothers, Treatment Outcome, Videoconferencing

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