CKAP2

Chr 13

cytoskeleton associated protein 2

Also known as: LB1, TMAP, se20-10

This gene encodes a cytoskeleton-associated protein that stabilizes microtubules and regulates cell division, cell cycling, and cell death in a p53-dependent manner. Mutations cause autosomal recessive neurodevelopmental disorders with intellectual disability, developmental delay, and seizures. The gene is extremely intolerant to loss-of-function variants, indicating that complete loss of protein function is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 1.13
Clinical SummaryCKAP2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
58 unique Pathogenic / Likely Pathogenic· 95 VUS of 171 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 1.04
OE 0.79 (0.561.13)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.40Z-score
OE missense 1.06 (0.971.16)
376 obs / 354.6 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.79 (0.561.13)
00.351.4
Missense OE1.06 (0.971.16)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 22 / 27.9Missense obs/exp: 376 / 354.6Syn Z: -0.44

ClinVar Variant Classifications

171 submitted variants in ClinVar

Classification Summary

Pathogenic56
Likely Pathogenic2
VUS95
Likely Benign9
Benign5
56
Pathogenic
2
Likely Pathogenic
95
VUS
9
Likely Benign
5
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
56
0
56
Likely Pathogenic
0
0
2
0
2
VUS
0
85
10
0
95
Likely Benign
0
9
0
0
9
Benign
0
3
1
1
5
Total097691167

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

View in ClinVar →

Protein Context — Lollipop Plot

CKAP2 · 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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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC