CNTN2

Chr 1AR

contactin 2

Also known as: AXT, EPEO5, FAME5, TAG-1, TAX, TAX1

This protein is a GPI-anchored neuronal membrane glycoprotein that organizes axonal domains at nodes of Ranvier by maintaining voltage-gated potassium channels at the juxtaparanodal region and functions in neuronal proliferation, migration, and axon guidance during cerebellar development. Autosomal recessive mutations cause early-onset epilepsy with or without developmental delay. The gene is highly constrained against loss-of-function variants (LOEUF 0.437), indicating that complete loss of protein function is likely pathogenic.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismARLOEUF 0.441 OMIM phenotype
Clinical SummaryCNTN2
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Gene-Disease Validity (ClinGen)
complex neurodevelopmental disorder · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.29) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.44LOEUF
pLI 0.002
Z-score 4.92
OE 0.29 (0.190.44)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.59Z-score
OE missense 0.72 (0.670.78)
481 obs / 669.4 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.29 (0.190.44)
00.351.4
Missense OE0.72 (0.670.78)
00.61.4
Synonymous OE0.92
01.21.6
LoF obs/exp: 16 / 55.6Missense obs/exp: 481 / 669.4Syn Z: 1.02
DN
0.76top 25%
GOF
0.6639th %ile
LOF
0.3550th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOFprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CNTN2 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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