NRXN1

Chr 2AR

neurexin 1

Also known as: Hs.22998, PTHSL2, SCZD17

The protein is a presynaptic cell membrane neurexin that binds neuroligins to form calcium-dependent complexes required for efficient neurotransmission and synaptic contact formation. Mutations cause autosomal recessive Pitt-Hopkins-like syndrome 2 and contribute to schizophrenia susceptibility through loss of function mechanisms. The gene is highly intolerant to loss-of-function variants, indicating haploinsufficiency as the likely pathogenic mechanism.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.252 OMIM phenotypes
Clinical SummaryNRXN1
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Gene-Disease Validity (ClinGen)
complex neurodevelopmental disorder · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
29 unique Pathogenic / Likely Pathogenic· 95 VUS of 234 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 — NRXN1
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.25LOEUF
pLI 1.000
Z-score 6.44
OE 0.15 (0.090.25)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
2.56Z-score
OE missense 0.76 (0.710.81)
681 obs / 897.1 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.15 (0.090.25)
00.351.4
Missense OE0.76 (0.710.81)
00.61.4
Synonymous OE1.22
01.21.6
LoF obs/exp: 10 / 66.8Missense obs/exp: 681 / 897.1Syn Z: -3.35
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongNRXN1-related Pitt Hopkins-like syndromeLOFAR
moderateNRXN1-related autismLOFAD
DN
0.5082th %ile
GOF
0.5758th %ile
LOF
0.58top 25%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOF1 literature citation · 17% of P/LP variants are LoF · LOEUF 0.25

Literature Evidence

LOFMarshall et al., 2017. It is the largest-to-date genome-wide schizophrenia cohort (21,094 cases; 20,227 controls). They assessed the contribution of CNV to the risk of SCZ. Odds ratio (95% CI) is 14.4 (4.2-46.9, cases: 35; controls: 3) for losses involving NRXN1 (majority are partial loss of the genPMID:27869829

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

234 submitted variants in ClinVar

Classification Summary

Pathogenic24
Likely Pathogenic5
VUS95
Likely Benign94
Benign4
Conflicting12
24
Pathogenic
5
Likely Pathogenic
95
VUS
94
Likely Benign
4
Benign
12
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
3
0
21
0
24
Likely Pathogenic
2
0
3
0
5
VUS
0
82
8
5
95
Likely Benign
0
3
32
59
94
Benign
0
0
3
1
4
Conflicting
12
Total5856765234

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

View in ClinVar →

Protein Context — Lollipop Plot

NRXN1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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