VPS4A

Chr 16AD

vacuolar protein sorting 4 homolog A

Also known as: CIMDAG, SKD1, SKD1A, SKD2, VPS4, VPS4-1

The VPS4A protein is an ATPase that disassembles ESCRT-III complexes during endosomal protein trafficking and is essential for cellular processes including cytokinesis, chromosome segregation, and primary cilium organization. Mutations cause CIMDAG syndrome, which is inherited in an autosomal dominant pattern. This gene is highly constrained against loss-of-function variants, indicating that functional copies are critical for normal cellular function.

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismADLOEUF 0.351 OMIM phenotype
Clinical SummaryVPS4A
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.94). One damaged copy is likely sufficient to cause disease.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.35LOEUF
pLI 0.945
Z-score 3.79
OE 0.13 (0.060.35)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
2.68Z-score
OE missense 0.54 (0.470.62)
147 obs / 271.5 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.13 (0.060.35)
00.351.4
Missense OE0.54 (0.470.62)
00.61.4
Synonymous OE1.18
01.21.6
LoF obs/exp: 3 / 22.4Missense obs/exp: 147 / 271.5Syn Z: -1.54
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveVPS4A-related CIMDAG Syndrome (monoallelic)DNAD
limitedVPS4A-related CIMDAG Syndrome (biallelic)OTHERAR
DN
0.5476th %ile
GOF
0.4184th %ile
LOF
0.56top 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.

LOFLOEUF 0.35
DN1 literature citation

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.

Literature Evidence

DNIn cultured cells, overexpression of VPS4A mutants caused enlarged endosomal vacuoles resembling those induced by expression of known dominant-negative ATPase-defective forms of VPS4A.PMID:33186545

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

VPS4A · 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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