FZD3

Chr 8

frizzled class receptor 3

Also known as: Fz-3

The FZD3 protein is a seven-transmembrane receptor for Wnt signaling proteins that controls axon growth and guidance during development of major central nervous system fiber tracts including the corpus callosum, corticospinal tract, and thalamocortical connections, as well as peripheral motor nerves and neural tube closure. Mutations cause autosomal recessive developmental and epileptic encephalopathy with microcephaly, intellectual disability, seizures, and structural brain abnormalities including agenesis of the corpus callosum. This gene is highly intolerant to loss-of-function mutations, indicating that complete loss of protein function is likely pathogenic.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.62
Clinical SummaryFZD3
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
0.62LOEUF
pLI 0.000
Z-score 3.14
OE 0.37 (0.230.62)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
2.64Z-score
OE missense 0.62 (0.560.69)
240 obs / 385.7 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.37 (0.230.62)
00.351.4
Missense OE0.62 (0.560.69)
00.61.4
Synonymous OE0.97
01.21.6
LoF obs/exp: 11 / 29.4Missense obs/exp: 240 / 385.7Syn Z: 0.29
DN
0.76top 25%
GOF
0.77top 25%
LOF
0.2679th %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

FZD3 · 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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