CNTNAP2

Chr 7AR

contactin associated protein 2

Also known as: AUTS15, CASPR2, CDFE, NRXN4, PTHSL1

This protein functions as a cell adhesion molecule localized at the juxtaparanodes of myelinated axons, where it mediates neuron-glia interactions during nervous system development and localizes potassium channels within differentiating axons. Biallelic mutations cause autosomal recessive Pitt-Hopkins-like syndrome 1, characterized by intellectual disability, epilepsy, and other neurodevelopmental features. The gene is also associated with autism susceptibility through a dominant-negative mechanism.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.552 OMIM phenotypes
Clinical SummaryCNTNAP2
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Gene-Disease Validity (ClinGen)
complex neurodevelopmental disorder · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

2 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
26 unique Pathogenic / Likely Pathogenic· 102 VUS of 300 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
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GeneReview available — CNTNAP2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.55LOEUF
pLI 0.000
Z-score 4.61
OE 0.40 (0.290.55)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
-0.29Z-score
OE missense 1.03 (0.971.09)
772 obs / 749.5 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.40 (0.290.55)
00.351.4
Missense OE1.03 (0.971.09)
00.61.4
Synonymous OE1.12
01.21.6
LoF obs/exp: 27 / 68.0Missense obs/exp: 772 / 749.5Syn Z: -1.60
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveCNTNAP2-related cortical dysplasia-focal epilepsy syndromeLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6936th %ile
GOF
0.5953th %ile
LOF
0.2874th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic19
Likely Pathogenic7
VUS102
Likely Benign155
Benign1
19
Pathogenic
7
Likely Pathogenic
102
VUS
155
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
10
0
9
0
19
Likely Pathogenic
7
0
0
0
7
VUS
1
91
10
0
102
Likely Benign
0
0
59
96
155
Benign
0
0
1
0
1
Total18917996284

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

CNTNAP2 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →