NDFIP2

Chr 13

Nedd4 family interacting protein 2

Also known as: N4WBP5A

The NDFIP2 protein activates HECT domain-containing E3 ubiquitin ligases and regulates the stability of multiple cellular targets, including potassium channels and cytokine signaling components. Mutations cause autosomal recessive neurodevelopmental disorder with seizures and dysmorphic features, typically presenting in early childhood. The gene shows significant constraint against loss-of-function variants (LOEUF 0.514), indicating intolerance to protein-disrupting mutations.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.51
Clinical SummaryNDFIP2
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.53) — some intolerance to loss-of-function variants.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.51LOEUF
pLI 0.527
Z-score 2.88
OE 0.20 (0.090.51)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.62Z-score
OE missense 0.87 (0.760.99)
150 obs / 172.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.20 (0.090.51)
00.351.4
Missense OE0.87 (0.760.99)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 3 / 15.1Missense obs/exp: 150 / 172.9Syn Z: 0.08
DN
0.6261th %ile
GOF
0.6834th %ile
LOF
0.4627th %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

NDFIP2 · 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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