VPS37A

Chr 8AR

VPS37A subunit of ESCRT-I

Also known as: HCRP1, PQBP2, SPG53

VPS37A encodes a component of the ESCRT-I complex that sorts ubiquitinated transmembrane proteins into multivesicular bodies for degradation. Mutations cause autosomal recessive spastic paraplegia-53, a hereditary spastic paraplegia primarily affecting the pyramidal motor system. The gene shows low constraint to loss-of-function variation (pLI 0.0007, LOEUF 0.722), consistent with its recessive inheritance pattern.

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismARLOEUF 0.721 OMIM phenotype
Clinical SummaryVPS37A
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Gene-Disease Validity (ClinGen)
complex hereditary spastic paraplegia · ARLimited

Limited evidence — not for standalone diagnostic reporting

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
0.72LOEUF
pLI 0.001
Z-score 2.53
OE 0.41 (0.250.72)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.88Z-score
OE missense 1.18 (1.061.31)
235 obs / 199.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.41 (0.250.72)
00.351.4
Missense OE1.18 (1.061.31)
00.61.4
Synonymous OE1.27
01.21.6
LoF obs/exp: 9 / 21.7Missense obs/exp: 235 / 199.8Syn Z: -1.82
DN
0.6744th %ile
GOF
0.4874th %ile
LOF
0.2678th %ile

The highest-scoring mechanism for this gene is dominant-negative.

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

VPS37A · 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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