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J. Buxbaum

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Open access Aug 2026

Estimating the contribution of coding mutations to autism

De novo mutations in protein-coding regions are strongly associated with autism, and family-based sequencing studies have identified numerous genes that harbor excess mutations in probands. However, the aggregate contribution of this class of variation to autism remains unclear. Here, we model the distribution of de novo autosomal coding variant effect sizes in 38,680 autism trios to estimate fundamental features of de novo genetic architecture. We find that damaging de novo single-nucleotide variants and frameshift indels explain 3.4% (95% CI: 2.1% - 4.7%) of autism variance on the observed scale. Approximately 7.0% (95% CI: 5.6% - 8.4%) of cases carry a large-effect mutation (rate ratio > 5), and most such mutations are incompletely penetrant. Although hundreds of genes make some nonzero contribution, 50% of mutational variance on the autosomes is explained by just 15 genes. De novo enrichments vary across cohorts with different ascertainment strategies; making projections for future trio studies, we show that many large-effect genes remain to be found.

A. Nadig, J. Fu, F. Satterstrom et al. · 0 citations
Open access Jul 2026

The NeuroWES project: lessons learned from comprehensive phenotyping and genetic analysis of neurodevelopmental disorders over a decade

This study showcases the complexities and novel findings derived from a decade-long analysis of 419 Italian NDD patient-parent trios, and underscores that navigating the complexities of large NDD cohorts requires a detailed, expert-driven approach to enhance diagnostic yield.

Simona Cardaropoli, Lisa Pavinato, Slavica Trajkova et al. · 0 citations