HAPSTR1

Chr 16

HUWE1 associated protein modifying stress responses

Also known as: C16orf72, PRO0149, TAPR1

HAPSTR1 encodes a protein that negatively regulates p53-mediated stress response pathways and cellular responses to telomere erosion and DNA damage through interactions with the E3 ubiquitin ligase HUWE1. The gene is highly constrained against loss-of-function variants (pLI 0.98, LOEUF 0.29), but no human diseases have been definitively associated with HAPSTR1 mutations to date. Given its critical role in DNA damage response and high constraint scores, pathogenic variants would likely cause severe developmental or neurological phenotypes.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 0.29
Clinical SummaryHAPSTR1
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.98). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
24 unique Pathogenic / Likely Pathogenic· 28 VUS of 61 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.29LOEUF
pLI 0.980
Z-score 3.53
OE 0.06 (0.020.29)
Highly constrained

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

Missense Constraint
1.95Z-score
OE missense 0.56 (0.470.67)
87 obs / 155.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.06 (0.020.29)
00.351.4
Missense OE0.56 (0.470.67)
00.61.4
Synonymous OE1.62
01.21.6
LoF obs/exp: 1 / 16.4Missense obs/exp: 87 / 155.3Syn Z: -3.70

ClinVar Variant Classifications

61 submitted variants in ClinVar

Classification Summary

Pathogenic22
Likely Pathogenic2
VUS28
Likely Benign1
Benign1
22
Pathogenic
2
Likely Pathogenic
28
VUS
1
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories· variant type breakdown unavailable

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
22
Likely Pathogenic
2
VUS
28
Likely Benign
1
Benign
1
Total54

Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

HAPSTR1 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC