FGF14

Chr 13AD

fibroblast growth factor 14

Also known as: FGF-14, FHF-4, FHF4, NYS4, SCA27, SCA27A, SCA27B

This protein is a member of the fibroblast growth factor family involved in nervous system development and function. Mutations cause spinocerebellar ataxia 27A and 27B, with 27B being late-onset, inherited in an autosomal dominant pattern. The gene is highly constrained against loss-of-function variants (pLI 0.91, LOEUF 0.39).

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 0.392 OMIM phenotypes
Clinical SummaryFGF14
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.91). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
129 unique Pathogenic / Likely Pathogenic· 112 VUS of 325 total submissions
💊
Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available
📖
GeneReview available — FGF14
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.39LOEUF
pLI 0.909
Z-score 2.96
OE 0.08 (0.030.39)
Highly constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.76Z-score
OE missense 0.57 (0.470.69)
75 obs / 132.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.08 (0.030.39)
00.351.4
Missense OE0.57 (0.470.69)
00.61.4
Synonymous OE0.95
01.21.6
LoF obs/exp: 1 / 12.1Missense obs/exp: 75 / 132.0Syn Z: 0.26
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveFGF14-related episodic ataxiaLOFAD
DN
0.4586th %ile
GOF
0.6150th %ile
LOF
0.66top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to loss-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFprediction above median · LOEUF 0.39
DN1 literature citation

Literature Evidence

DNIn vitro molecular studies in cultured neurons indicate that the FGF14 (F145S) SCA27 allele acts as a dominant negative mutant suppressing the FGF14 wild type function and resulting in inhibition of voltage-gated Na(+) and Ca(2+) channels.PMID:26089778

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

325 submitted variants in ClinVar

Classification Summary

Pathogenic118
Likely Pathogenic11
VUS112
Likely Benign50
Benign29
Conflicting3
118
Pathogenic
11
Likely Pathogenic
112
VUS
50
Likely Benign
29
Benign
3
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
6
2
110
0
118
Likely Pathogenic
8
0
3
0
11
VUS
6
69
32
5
112
Likely Benign
1
7
18
24
50
Benign
0
0
27
2
29
Conflicting
3
Total217819031323

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

FGF14 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗