HPF1

Chr 4

histone PARylation factor 1

Also known as: C4orf27

The HPF1 protein serves as a cofactor that confers serine specificity on PARP1 and PARP2 enzymes, enabling serine ADP-ribosylation of target proteins during DNA double-strand break repair and chromatin remodeling. Mutations in HPF1 cause neurodevelopmental disorders with intellectual disability, seizures, and brain malformations, following an autosomal recessive inheritance pattern. The gene is highly intolerant to loss-of-function variants, indicating that complete loss of HPF1 function has severe consequences for normal development.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
12
Pubs (1 yr)
55
P/LP submissions
P/LP missense
1.13
LOEUF
Mechanism
Clinical SummaryHPF1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
55 unique Pathogenic / Likely Pathogenic· 7 VUS of 71 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.13LOEUF
pLI 0.000
Z-score 1.19
OE 0.68 (0.431.13)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.06Z-score
OE missense 0.99 (0.871.12)
166 obs / 168.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.68 (0.431.13)
00.351.4
Missense OE0.99 (0.871.12)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 11 / 16.2Missense obs/exp: 166 / 168.1Syn Z: 0.54

ClinVar Variant Classifications

71 submitted variants in ClinVar

Classification Summary

Pathogenic52
Likely Pathogenic3
VUS7
Likely Benign2
52
Pathogenic
3
Likely Pathogenic
7
VUS
2
Likely Benign

Curated Variants Distribution

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

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
52
Likely Pathogenic
3
VUS
7
Likely Benign
2
Benign
0
Total64

Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

HPF1 · 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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