FGD3

Chr 9

FYVE, RhoGEF and PH domain containing 3

Also known as: ZFYVE5

The FGD3 protein functions as a guanyl-nucleotide exchange factor that activates CDC42 by exchanging GDP for GTP, thereby promoting filopodium formation and regulating actin cytoskeleton organization and cell shape. Gain-of-function mutations in FGD3 cause disease, though the specific neurological phenotypes are not detailed in the available data. The extremely low pLI score (0.0000013) indicates this gene is highly tolerant to loss-of-function variants, consistent with the gain-of-function disease mechanism.

OMIMResearchSummary from RefSeq, UniProt, Mechanism
MultiplemechanismLOEUF 0.73
Clinical SummaryFGD3
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
0.73LOEUF
pLI 0.000
Z-score 2.73
OE 0.48 (0.320.73)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.65Z-score
OE missense 0.78 (0.710.85)
345 obs / 442.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.48 (0.320.73)
00.351.4
Missense OE0.78 (0.710.85)
00.61.4
Synonymous OE0.95
01.21.6
LoF obs/exp: 15 / 31.5Missense obs/exp: 345 / 442.9Syn Z: 0.53
DN
0.6162th %ile
GOF
0.7030th %ile
LOF
0.3452th %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

FGD3 · 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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