GINS1

Chr 20AR

GINS complex subunit 1

Also known as: IMD55, PSF1

GINS1 encodes a subunit of the GINS complex, which is essential for DNA replication initiation and progression as part of the CMG helicase that unwinds template DNA during replication. Biallelic mutations cause autosomal recessive immunodeficiency 55, affecting the immune system. The gene is not highly constrained against loss-of-function variants (pLI near 0, LOEUF 1.28).

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Immunodeficiency 55MIM #617827
AR
0
Active trials
14
Pubs (1 yr)
22
P/LP submissions
5%
P/LP missense
1.28
LOEUF
Mechanism
Clinical SummaryGINS1
🧬
Gene-Disease Validity (ClinGen)
combined immunodeficiency due to GINS1 deficiency · ARModerate

Moderate evidence — consider for supplementary testing

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
21 unique Pathogenic / Likely Pathogenic· 145 VUS of 244 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.28LOEUF
pLI 0.000
Z-score 0.80
OE 0.77 (0.481.28)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.22Z-score
OE missense 0.94 (0.801.11)
101 obs / 107.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.77 (0.481.28)
00.351.4
Missense OE0.94 (0.801.11)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 11 / 14.3Missense obs/exp: 101 / 107.5Syn Z: -0.49

ClinVar Variant Classifications

244 submitted variants in ClinVar

Classification Summary

Pathogenic17
Likely Pathogenic4
VUS145
Likely Benign59
Benign4
Conflicting2
17
Pathogenic
4
Likely Pathogenic
145
VUS
59
Likely Benign
4
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
17
0
17
Likely Pathogenic
0
1
3
0
4
VUS
10
99
33
3
145
Likely Benign
0
0
30
29
59
Benign
0
2
2
0
4
Conflicting
2
Total101028532231

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

View in ClinVar →

Protein Context — Lollipop Plot

GINS1 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗