KREMEN2

Chr 16

kringle containing transmembrane protein 2

Also known as: KRM2

This gene encodes a transmembrane receptor for Dickkopf proteins that cooperates with DKK1/2 to inhibit Wnt/beta-catenin signaling by promoting endocytosis of Wnt receptors LRP5 and LRP6, playing important roles in limb development and bone formation regulation. Mutations in KREMEN2 cause autosomal recessive ectodermal dysplasia with syndactyly. The gene shows tolerance to loss-of-function variants (LOEUF 0.917), suggesting that complete loss of function may be required for disease manifestation.

OMIMResearchSummary from RefSeq, UniProt
GOFmechanismLOEUF 0.92
Clinical SummaryKREMEN2
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
0.92LOEUF
pLI 0.000
Z-score 1.83
OE 0.54 (0.330.92)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.51Z-score
OE missense 0.73 (0.640.82)
178 obs / 244.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.54 (0.330.92)
00.351.4
Missense OE0.73 (0.640.82)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 10 / 18.5Missense obs/exp: 178 / 244.5Syn Z: -0.56
DN
0.5968th %ile
GOF
0.6931th %ile
LOF
0.3258th %ile

The highest-scoring mechanism for this gene is gain-of-function.

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

KREMEN2 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →