A Spoken-Language Intervention for School-Aged Boys With Fragile X Syndrome
Open accessAbstract
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.”
From Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome (2018) · cites this paper as [94]
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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
- Publisher
- Informa UK Limited
- Published
- 2016-05-01
- Volume
- 121
- Issue
- 3
- Pages
- 236-265
- ISSN
- 1944-7558 (ISSN-L)
- 1944-7515
- PMC
- PMC4849176
- 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
- green
- www.ncbi.nlm.nih.gov
- 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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