NPFFR2

Chr 4

neuropeptide FF receptor 2

Also known as: GPR74, HLWAR77, NPFF2, NPGPR

The protein functions as a G-protein-coupled receptor that binds neuropeptides NPAF and NPFF to regulate pain modulation and opioid system function through phosphatidylinositol-calcium signaling. Biallelic mutations cause autosomal recessive normosmic congenital hypogonadotropic hypogonadism, a disorder affecting reproductive hormone regulation that typically presents with delayed or absent puberty. The gene shows very low constraint against loss-of-function variants, consistent with recessive inheritance where one functional copy is insufficient.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.13
Clinical SummaryNPFFR2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.13LOEUF
pLI 0.000
Z-score 1.15
OE 0.71 (0.461.13)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-1.17Z-score
OE missense 1.20 (1.091.31)
336 obs / 281.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.71 (0.461.13)
00.351.4
Missense OE1.20 (1.091.31)
00.61.4
Synonymous OE1.20
01.21.6
LoF obs/exp: 13 / 18.3Missense obs/exp: 336 / 281.0Syn Z: -1.62
DN
0.78top 25%
GOF
0.7125th %ile
LOF
0.3162th %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

NPFFR2 · 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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