ArticleJournal of intellectual disability research : JIDR2014
Consistency between research and clinical diagnoses of autism among boys and girls with fragile X syndrome.
Article in Journal of intellectual disability research : JIDR, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 75 papers, 2 of them syntheses that pooled it.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
75 citing papers in PubMed, 2 syntheses or guidelines pooled it, 96 citations in OpenAlex.
- Autonomic Function in Fragile X Syndrome: A Systematic Review.Journal of intellectual disability research : JIDR · 2026Pooled it
- Autism spectrum disorder in females with fragile X syndrome: a systematic review and meta-analysis of prevalence.Journal of neurodevelopmental disorders · 2021Pooled it
- Short-term and Long-term Stability of the Autism Diagnostic Observation Schedule (ADOS-2) Calibrated Comparison Scores (CCS) and Classification Scores in Youth with Down Syndrome or Fragile X Syndrome with Intellectual Disability.Journal of autism and developmental disorders · 2026Article
- Intergenerational associations in inhibitory control inFrontiers in psychiatry · 2026Article
- Assessing Autism Co-Occurrence in Fragile X Syndrome: Proposing a Preliminary CARS-2 Cut-Off Score.Journal of autism and developmental disorders · 2025Article
- The effect of anxiety and autism symptom severity on restricted and repetitive behaviors over time in children with fragile X syndrome.Journal of neurodevelopmental disorders · 2024Article
- Description and predictors of self-determination in males and females with fragile X syndrome on the verge of adulthood.Journal of intellectual disability research : JIDR · 2024Article
- Developmental associations between motor and communication outcomes in Fragile X syndrome: Variation in the context of co-occurring autism.Autism : the international journal of research and practice · 2024Article
- Autistic Traits Associated with the Fragile X Premutation Allele: The Neurodevelopmental Profile.Developmental neuropsychology · 2024Review
- Characterising the social interaction style of autism in young adult males with fragile X syndrome.Journal of intellectual disability research : JIDR · 2024Article
- Language Skills Influence Transition Planning in Adolescents With Fragile X Syndrome.American journal on intellectual and developmental disabilities · 2024Article
- Adaptive, behavioral, and cognitive outcomes in individuals with fragile X syndrome with varying autism severity.International journal of developmental neuroscience : the official journal of the International Society for Developmental Neuroscience · 2023Article
- The Effect of Anxiety and Autism Symptom Severity on Restricted and Repetitive Behaviors Over Time in Children with Fragile X Syndrome.Research square · 2023Article
- Review
- ADHD and ASD symptoms in young males with fragile X syndrome: associations with early trajectories of inhibitory control.Child neuropsychology : a journal on normal and abnormal development in childhood and adolescence · 2023Article
- Predictors, Parental Views, and Concordance Across Diagnostic Sources of Autism in Male Youth with Fragile X Syndrome: Clinical Best Estimate and Community Diagnoses.Research on child and adolescent psychopathology · 2023Article
- Transactional relationships between maternal requesting and responsivity, adolescent compliance, and task demands in fragile X syndrome.Research in developmental disabilities · 2023Article
- A longitudinal investigation of pragmatic language across contexts in autism and related neurodevelopmental conditions.Frontiers in neurology · 2023Article
- Negative affect and respiratory sinus arrhythmia are differentially related to social anxiety and autism features in autistic preschoolers contrasted to fragile X syndrome.Frontiers in psychiatry · 2023Article
- Neuroanatomical Profile of Young Females with Fragile X Syndrome: A Voxel-Based Morphometry Analysis.Cerebral cortex (New York, N.Y. : 1991) · 2022Article
15 more citing papers are in PubMed but not listed here.
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
3 authors at 2 institutions in 1 country.
Funding
Abstract
backgroundPrior research suggests that 60-74% of males and 16-45% of females with fragile X syndrome (FXS) meet criteria for autism spectrum disorder (ASD) in research settings. However, relatively little is known about the rates of clinical diagnoses in FXS and whether such diagnoses are consistent with those performed in a research setting using gold standard diagnostic tools.
methodThis study explored whether boys and girls with FXS met criteria for ASD in a research setting using the Autism Diagnostic Observation Schedule (ADOS) and the Autism Diagnostic Interview-Revised (ADI-R), and then compared these data with the frequency of parent-reported clinical diagnoses. We also examined child and family characteristics as potential diagnostic predictors across settings. Participants included 35 females and 51 males with FXS (mean age: 10 years), who were from Eastern and Midwestern regions of the USA.
resultsAbout half of the children met criteria for ASD on either the ADOS or ADI-R, with ASD occurring three times more frequently in males than females (∼75% vs. ∼25%). In contrast, ∼25% of participants of both genders had received a clinical diagnosis of ASD. While cognitive and language skills predicted diagnostic outcome on the ADOS and ADI-R, these skills did not predict clinical diagnoses. Executive functions predicted clinical diagnoses, but not diagnoses per the ADOS or ADI-R.
conclusionsASD in FXS may be under-diagnosed in clinical/educational settings, which raises questions regarding access to ASD-related services.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.