Archives of Pediatrics and Adolescent MedicineGenetic & Genomic MedicinePeer-reviewed journal article
Integrating genomic variants and developmental milestones to predict cognitive and adaptive outcomes in autistic children
Vincent-Raphaël BourqueiD, Zoe Schmilovich, Guillaume HuguetiD, Jade England, Adeniran OkewoleiD, Cécile PoulainiD, Thomas RenneiD, Martineau Jean‐LouisiD, Zohra SaciiD, Xinhe ZhangiD, Thomas RollandiD, Aurélie LabbeiD, Jacob A. S. VorstmaniD, Guy Armand RouleauiD, Simon Baron‐CoheniD, Laurent MottroniD, Richard A. I. BethlehemiD, Varun WarrieriD, Sébastien JacquemontiD
Abstract
Although the first signs of autism are often observed as early as 18-36 months of age, there is a broad uncertainty regarding future development, and clinicians lack predictive tools to identify those who will later be diagnosed with co-occurring intellectual disability (ID). Here, we developed predictive models of ID in autistic children (n=5,633 from three cohorts), integrating different classes of genetic variants alongside developmental milestones. The integrated model yielded an AUC ROC=0.65, with this predictive performance cross-validated and generalised across cohorts. Positive predictive values reached up to 55%, accurately identifying 10% of ID cases. The ability to stratify the probabilities of ID using genetic variants was up to twofold greater in individuals with delayed milestones compared to those with typical development. These findings underscore the potential of models in neurodevelopmental medicine that integrate genomics and clinical observations to predict outcomes and target interventions.
- organism: human
- assay: whole-exome sequencing
- research focus: variant effect prediction
- approach: statistical model
- Citations
- 9
- FWCI
- 5.10
- Field percentile
- top 4%
Reproducibility ledger
Nobody has reported trying to reproduce this paper yet. Did you replicate an experiment or run the code? Your report saves the next lab a week.
Sign in to file a reproducibility report.
Related papers
- Gene functional networks and autism spectrum characteristics in young people with intellectual disability
Diandra Brkić, Elise Ng‐Cordell, … Kate Baker·bioRxiv·May 11, 2020
- Combinatorial approach for complex disorder prediction: Case Study Of Neurodevelopmental Disorders
Linh Ngoc Huynh, Fereydoun Hormozdiari·GeneticsPeer-reviewed·Oct 8, 2018
- Genetic correlates of phenotypic heterogeneity in autism
Varun Warrier, Xinhe Zhang, … Simon Baron‐Cohen·Nature GeneticsPeer-reviewed·Jun 2, 2022
- Gene discoveries in autism are biased towards comorbidity with intellectual disability
Matthew Jensen, Corrine Smolen, Santhosh Girirajan·Journal of Medical GeneticsPeer-reviewed·Mar 9, 2020
- Prematurity and Genetic Liability for Autism Spectrum Disorder
Yali Zhang, Ashraf Yahia, … Kristiina Tammimies·Genome MedicinePeer-reviewed·Oct 2, 2025
- Copy-number variants and polygenic risk for intelligence confer risk for autism spectrum disorder irrespective of their effects on cognitive ability
Zoe Schmilovich, Vincent-Raphaël Bourque, … Guy Armand Rouleau·Frontiers in PsychiatryPeer-reviewed·May 1, 2024