CEP170

Chr 1

centrosomal protein 170

Also known as: FAM68A, KAB, KIAA0470

CEP170 encodes a centrosomal protein required for centriole subdistal appendage assembly and microtubule organization during both interphase and mitosis. Mutations cause autosomal recessive primary microcephaly with seizures and intellectual disability, reflecting the gene's critical role in proper cell division and brain development. This gene is highly constrained against loss-of-function variants in the general population.

OMIMResearchSummary from RefSeq, UniProt
LOFmechanismLOEUF 0.19
Clinical SummaryCEP170
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.19LOEUF
pLI 1.000
Z-score 6.58
OE 0.10 (0.050.19)
Highly constrained

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

Missense Constraint
2.20Z-score
OE missense 0.78 (0.720.83)
593 obs / 763.9 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.10 (0.050.19)
00.351.4
Missense OE0.78 (0.720.83)
00.61.4
Synonymous OE0.85
01.21.6
LoF obs/exp: 6 / 61.8Missense obs/exp: 593 / 763.9Syn Z: 1.92
DN
0.3892th %ile
GOF
0.4381th %ile
LOF
0.79top 5%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.19

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CEP170 · 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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