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De novo gene mutations linked with autism: a family-friendly reference

An original diffrnt. guide to autism-associated genes with strong de novo evidence—drawn from SFARI Gene, large sequencing studies, and public databases. For learning, not diagnosis.

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Original to diffrnt.info

This article was written and published by the creators of diffrnt. It is not syndicated from another outlet. We research and edit these pieces to support San Diego families with practical context on autism and neurodiversity.

Last updated: May 2026 · For informational purposes only, not medical or career advice.

When families receive genetic testing results, words like de novo, likely gene-disrupting, and penetrance can be hard to parse. This article summarizes publicly curated autism gene lists where new (non-inherited) mutations are a major part of the evidence—so you can orient yourself before speaking with a genetic counselor or clinician.

Not medical or genetic advice.This article summarizes public research databases for orientation and learning. It does not interpret your child's test results. Always discuss variants with a genetic counselor or clinician.

Key terms

De novo
A variant that arose new in the child and was not inherited from either parent.
Likely gene-disrupting (LGD)
Often a stop-gain, frameshift, splice, or strongly damaging missense change—variants thought to severely affect the protein.
Penetrance
Not everyone with a given variant develops autism; risk is probabilistic and varies by gene and variant.
NDD
Neurodevelopmental disorder—a broader label that often includes intellectual disability, epilepsy, or speech delay alongside autism traits.

Public studies and databases

Tier 1 — Highest-confidence genes (SFARI Category 1)

SFARI Gene Category 1 lists 245 genes (May 2026) with clear autism links, usually supported by multiple de novo likely-gene-disrupting mutations. Below are grouped highlights; the full alphabetical list is at the end.

Synaptic and neuronal signaling

GeneWhy it appears here
SHANK3Postsynaptic scaffold; 22q13 region; among the strongest ASD signals.
SHANK2Related synaptic scaffold; recurrent de novo hits.
NRXN1Neurexin; presynaptic adhesion.
NRXN2Neurexin family.
NRXN3Neurexin family.
NLGN2Neuroligin.
NLGN3Neuroligin.
NLGN4XX-linked neuroligin.
SYNGAP1Synaptic Ras-GAP; ID, epilepsy, and ASD overlap.
DLG4PSD-95; postsynaptic density.
GRIN2BNMDA receptor subunit; classic recurrent de novo gene.
GRIN2ANMDA receptor subunit.
GRIN1NMDA receptor subunit.
GRIA2AMPA receptor subunit.
GABRB3GABA-A receptor.
GABRB2GABA-A receptor.
SCN2ASodium channel; top penetrant de novo gene in many cohorts.
SCN1ASodium channel; epilepsy and NDD overlap.
SCN8ASodium channel.
CACNA1CCalcium channel.
CACNA1ACalcium channel.
CACNA1ECalcium channel.
STXBP1Synaptic vesicle release.
RELNReelin signaling.

Chromatin remodeling and transcription

GeneWhy it appears here
CHD8Top-tier de novo ASD gene; macrocephaly often reported.
CHD2CHD family helicase.
CHD7CHARGE syndrome region; NDD overlap.
CHD3CHD family helicase.
ARID1BSWI/SNF chromatin remodeling.
SMARCA4SWI/SNF.
SMARCA2SWI/SNF.
SMARCC2SWI/SNF.
KMT2AHistone methyltransferase.
KMT2CHistone methyltransferase.
KMT2EHistone methyltransferase.
KDM5BHistone demethylase.
KDM5CX-linked histone demethylase.
KDM6BHistone demethylase.
DNMT3ADNA methylation.
EHMT1Kleefstra-related chromatin gene.
CREBBPCREB-binding protein.
EP300p300 histone acetyltransferase.
CTCFGenome architecture / insulator protein.
YY1Transcription factor; genome regulation.
ASH1LHistone methyltransferase.
SETD5Chromatin regulator.
SETD1AChromatin regulator.

Brain development and neuronal specification

GeneWhy it appears here
DYRK1A21q22 kinase; microcephaly association in some cases.
TBR1T-box TF; OMIM documents de novo TBR1 variants with autism and speech delay.
FOXP1Forkhead transcription factor.
FOXP2Speech and language development.
FOXG1Forebrain development.
MEF2CCortical development.
SATB2SATB2-associated syndrome.
POGZHigh-impact developmental regulator.
PHIPDevelopmental disorder gene.
ADNPHelsmoortel–van der Aa syndrome; strong ASD link.
BCL11ACortical interneuron development.

Cytoskeleton, migration, and Rho signaling

GeneWhy it appears here
TRIORho GEF; de novo enrichment in autism studies.
NCKAP1Neurite migration.
WDFY3Neurite and synaptic development.
DYNC1H1Dynein; axonal transport.
DPYSL2CRMP family; axon guidance.

RNA processing and regulation

GeneWhy it appears here
DDX3XX-linked; intellectual disability and ASD.
HNRNPUhnRNP family.
HNRNPH2X-linked hnRNP.
CELF4RNA regulation.
UPF3BNonsense-mediated decay; X-linked.
SONRNA-binding; enriched in NDD/ASD cohorts.

Syndromic genes with strong autism and de novo overlap

GeneWhy it appears here
FMR1Fragile X.
MECP2Rett syndrome (X-linked).
TSC1Tuberous sclerosis.
TSC2Tuberous sclerosis.
PTENPTEN hamartoma; macrocephaly/autism.
NF1Neurofibromatosis type 1.
UBE3A15q11 region (Angelman context).
CDKL5Epileptic encephalopathy (X-linked).
PCDH19Epileptic encephalopathy (X-linked).
RAI1Smith–Magenis (17p11.2).
ANKRD11KBG syndrome.

Tier 2 — Landmark recurrent de novo genes

Early large trio exome studies (e.g. Sanders 2012, De Rubeis 2014) highlighted these six genes as recurrent de novo contributors to sporadic autism risk.

GeneWhy it appears here
CHD8Chromatin remodeling.
DYRK1AKinase; 21q22 region.
GRIN2BGlutamate signaling.
TBR1T-box transcription factor.
PTENPI3K pathway / tumor suppressor.
TBL1XR1Transducin beta-like protein.

Tier 3 — Findings from large meta-analyses (2021–2022)

Primarily supported by de novo variants (Zhou et al. 2022)

GeneWhy it appears here
MARK2Primarily de novo; also NDD, epilepsy, Tourette overlap.
ITSN1De novo and inherited; endocytosis.
SCAF1De novo and inherited.
HNRNPUL2De novo and inherited; RNA processing.

Also exome-wide significant in large meta-analyses

Includes established de novo drivers such as CHD8, SHANK3, SCN2A, ADNP, and FOXP1. Some genes (e.g. NAV3) are driven more by inherited loss-of-function than classic de novo hits.

GeneWhy it appears here
SHANK3Established de novo driver.
SCN2AEstablished de novo driver.
ADNPEstablished de novo driver.
FOXP1Established de novo driver.
NAV3Mostly inherited LoF in Zhou 2022—not a classic de novo list gene.

Full list — SFARI Category 1 (alphabetical)

242 gene symbols from the public Category 1 export. Search any symbol on gene.sfari.org for variant reports and scores.

ABCE1, ACTB, ADNP, ADSL, AFF2, AHDC1, ALDH5A1, ANK2, ANK3, ANKRD11, ANP32A, AP2S1, ARF3, ARHGEF9, ARID1B, ARX, ASH1L, ASTN1, ASXL3, ATRX, AUTS2, BAZ2B, BCKDK, BCL11A, BRAF, BRSK2, CACNA1A, CACNA1C, CACNA1E, CACNA2D3, CAMK2A, CAMTA2, CAPRIN1, CASK, CASZ1, CDKL5, CELF4, CHAMP1, CHD2, CHD3, CHD7, CHD8, CIC, CNOT3, CORO1A, CPSF7, CREBBP, CSDE1, CSNK2A1, CTCF, CTNNB1, CUL3, DDX3X, DEAF1, DHCR7, DIP2A, DLG4, DMPK, DNMT3A, DPYSL2, DSCAM, DYNC1H1, DYRK1A, EBF3, EHMT1, EIF3G, ELAVL3, EP300, FMR1, FOXG1, FOXP1, FOXP2, GABRB2, GABRB3, GFAP, GIGYF1, GIGYF2, GNAI1, GRIA2, GRIN1, GRIN2A, GRIN2B, HDLBP, HECTD4, HIVEP2, HNRNPH2, HNRNPU, HRAS, IQSEC2, IRF2BPL, KANSL1, KATNAL2, KCNB1, KCNQ2, KCNQ3, KDM2B, KDM3B, KDM5B, KDM5C, KDM6B, KIAA0232, KLHL20, KMT2A, KMT2C, KMT2E, KMT5B, LDB1, LRRC4C, LZTR1, MAGEC3, MAGEL2, MAP1A, MBD5, MBOAT7, MECP2, MED13, MED13L, MEF2C, MEIS2, MKX, MSL3, MTOR, MYCBP2, MYT1L, NAA15, NACC1, NBEA, NCKAP1, NCOA1, NEXMIF, NF1, NIPBL, NLGN2, NLGN3, NLGN4X, NR3C2, NR4A2, NRXN1, NRXN2, NRXN3, NSD1, NUP155, PACS1, PAH, PAX5, PCCB, PCDH19, PHF12, PHF2, PHF21A, PHF3, PHIP, POGZ, POMGNT1, PPP1R9B, PPP2R5D, PPP5C, PRMT9, PRR12, PRR14L, PSMD11, PSMD12, PSMD6, PTCHD1, PTEN, PTK7, PTPN11, RAI1, RALGAPB, RELN, RERE, RFX3, RIMS1, RORB, RUNX1T1, SATB1, SATB2, SCN1A, SCN2A, SCN8A, SETBP1, SETD1A, SETD2, SETD5, SF1, SHANK2, SHANK3, SIN3A, SKI, SLC6A1, SLC9A6, SMARCA1, SMARCA2, SMARCA4, SMARCC2, SON, SOS2, SOX5, SPAST, SRCAP, SRPRA, STXBP1, SYN1, SYNGAP1, TANC2, TAOK1, TBCEL, TBCK, TBL1XR1, TBR1, TCF20, TCF4, TCF7L2, TEK, TLE3, TLK2, TM9SF4, TRAF7, TRIM23, TRIO, TRIP12, TRRAP, TSC1, TSC2, TSHZ1, TSHZ3, UBAP2L, UBE3A, UBR1, UPF3B, USP9X, VEZF1, VPS13B, WAC, WDFY3, YY1, ZBTB20, ZBTB21, ZMYND8, ZNF292, ZNF462, ZNF865

Category 2 genes (often two reported de novo LGD hits) are not listed here. Browse them on SFARI Category 2.

Look up any gene

  • SFARI GeneAutism-specific gene scores and reported variants.
  • ClinVarClinical classifications of specific variants.
  • gnomADHow rare a variant is in the general population.
  • OMIMGene–disease entries and references.
  • GeneMatcherConnect families and clinicians studying the same gene.
  • DECIPHERDevelopmental disorders, CNVs, and phenotype sharing.

Important limitations

  • This is not a complete catalog of every possible de novo gene. Thousands of genes can carry rare new mutations; only a subset reach statistical and clinical confidence for autism.
  • Many de novo variants are benign. Pathogenicity depends on the specific change, not the gene name alone.
  • Inherited variants also contribute substantially to autism risk in large studies—not only de novo changes.
  • A gene on this list does not mean your child has that mutation. Always interpret results with a qualified genetic counselor or clinician.

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