CHEK1

Chr 11AD

checkpoint kinase 1

Also known as: CHK1, OZEMA21

This serine/threonine kinase regulates cell cycle checkpoints and DNA damage repair by phosphorylating key substrates including CDC25 phosphatases, TP53, and RAD51. Mutations cause oocyte/zygote/embryo maturation arrest 21, an autosomal dominant condition affecting early reproductive development. The gene is highly intolerant to loss-of-function mutations (pLI near 0, LOEUF 0.931), suggesting that complete protein loss is likely incompatible with normal development.

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

Oocyte/zygote/embryo maturation arrest 21MIM #620610
AD
5
Active trials
80
Pubs (1 yr)
68
P/LP submissions
4%
P/LP missense
0.93
LOEUF
DN
Mechanism· predicted
Clinical SummaryCHEK1
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Gene-Disease Validity (ClinGen)
familial ovarian cancer · ADNo Known Disease Relationship

No known disease relationship

2 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
67 unique Pathogenic / Likely Pathogenic· 77 VUS of 202 total submissions
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Clinical Trials
5 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.93LOEUF
pLI 0.000
Z-score 1.83
OE 0.61 (0.410.93)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.78Z-score
OE missense 0.68 (0.600.78)
170 obs / 249.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.61 (0.410.93)
00.351.4
Missense OE0.68 (0.600.78)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 16 / 26.1Missense obs/exp: 170 / 249.1Syn Z: -0.48
DN
0.6939th %ile
GOF
0.5170th %ile
LOF
0.3550th %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

202 submitted variants in ClinVar

Classification Summary

Pathogenic64
Likely Pathogenic3
VUS77
Likely Benign38
Benign10
64
Pathogenic
3
Likely Pathogenic
77
VUS
38
Likely Benign
10
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
3
60
0
64
Likely Pathogenic
0
0
3
0
3
VUS
1
63
12
1
77
Likely Benign
1
1
11
25
38
Benign
0
1
8
1
10
Total3689427192

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

View in ClinVar →

Protein Context — Lollipop Plot

CHEK1 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

Clinical Literature
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