NETO1

Chr 18

neuropilin and tolloid like 1

Also known as: BCTL1, BTCL1

The protein functions as an accessory subunit of neuronal NMDA receptors, maintaining GRIN2A-containing NMDARs in the postsynaptic density and regulating synaptic plasticity critical for spatial learning and memory. Mutations cause autosomal recessive intellectual disability and epileptic encephalopathy with onset in infancy or early childhood. The gene is highly constrained against loss-of-function variants (LOEUF 0.496), indicating that complete loss of protein function is likely not tolerated.

Summary from RefSeq, UniProt
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0
Active trials
3
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.50
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryNETO1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.27) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.50LOEUF
pLI 0.053
Z-score 3.62
OE 0.27 (0.160.50)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.80Z-score
OE missense 0.71 (0.630.79)
213 obs / 300.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.27 (0.160.50)
00.351.4
Missense OE0.71 (0.630.79)
00.61.4
Synonymous OE1.04
01.21.6
LoF obs/exp: 8 / 29.1Missense obs/exp: 213 / 300.7Syn Z: -0.30
DN
0.74top 25%
GOF
0.74top 25%
LOF
0.3647th %ile

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

GOFprediction above median
DNprediction 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

NETO1 · protein map & ClinVar variants

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

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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