LITERATURE · PEER REVIEWED

Transcriptomic and phenotypic convergence of neurodevelopmental disorder risk genes in vitro and in vivo

Nature Neuroscience · 2026 · Peer reviewed

COMPLETE CITATION

Citation and identifiers

Fernandez Garcia M, Retallick-Townsley K, Pruitt A, et al.. “Transcriptomic and phenotypic convergence of neurodevelopmental disorder risk genes in vitro and in vivo.” Nature Neuroscience. 29:1079–1094 · doi:10.1038/s41593-026-02247-7.

PubMed ID
42032432

RESEARCH QUESTION

How does ASH1L loss alter proliferation and cell-state programs across neural progenitors, excitatory neurons, inhibitory neurons, and an in vivo zebrafish model?

The study directly perturbed ASH1L across human neural cell types and zebrafish, finding increased Ki-67-positive neural progenitors while placing broader network results within a 23-gene convergence analysis.

INTERPRETATION BOUNDARY

Read the finding and limit together.

What this study supports

A Yale-led study directly perturbed ASH1L within a 23-gene CRISPR program across neural progenitors and glutamatergic and GABAergic neurons, with arrayed ASH1L assays and an ash1l zebrafish model. ASH1L loss increased Ki-67-positive neural progenitors and contributed to cell-state-specific convergence analyses.

What it cannot establish

This is a gene-knockout study, not a graded ASH1L dosage-response experiment or a functional classification of patient alleles. Most network findings are cross-gene convergence results; the reported Seahorse assay did not identify a significant respiratory change for ASH1L.

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