SYT4

Chr 18

synaptotagmin 4

Also known as: HsT1192

Synaptotagmin-4 is a calcium-independent synaptotagmin family member that regulates dense core vesicle transport at synapses through interaction with the motor protein KIF1A and is involved in dendrite formation. Biallelic mutations in SYT4 cause autosomal recessive neurodevelopmental disorder with seizures and absent language, typically presenting in infancy with severe developmental delays and intractable epilepsy. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.549), consistent with its role in critical synaptic functions.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
5
Pubs (1 yr)
46
P/LP submissions
0%
P/LP missense
0.55
LOEUF
Multiple*
Mechanism· predicted
Clinical SummarySYT4
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.21) despite low pLI — interpret in context.
📋
ClinVar Variants
44 unique Pathogenic / Likely Pathogenic· 43 VUS of 94 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.55LOEUF
pLI 0.439
Z-score 2.74
OE 0.21 (0.100.55)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.06Z-score
OE missense 0.99 (0.881.11)
213 obs / 215.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.21 (0.100.55)
00.351.4
Missense OE0.99 (0.881.11)
00.61.4
Synonymous OE1.16
01.21.6
LoF obs/exp: 3 / 14.1Missense obs/exp: 213 / 215.6Syn Z: -1.17
DN
0.6841th %ile
GOF
0.6834th %ile
LOF
0.4825th %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

94 submitted variants in ClinVar

Classification Summary

Pathogenic42
Likely Pathogenic2
VUS43
Likely Benign5
42
Pathogenic
2
Likely Pathogenic
43
VUS
5
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
42
0
42
Likely Pathogenic
0
0
2
0
2
VUS
0
39
4
0
43
Likely Benign
0
3
2
0
5
Benign
0
0
0
0
0
Total04250092

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

View in ClinVar →

Protein Context — Lollipop Plot

SYT4 · protein map & ClinVar variants

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

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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