CEP78

Chr 9AR

centrosomal protein 78

Also known as: C9orf81, CRDHL, IP63

This centrosomal protein regulates centriole duplication and cilia biogenesis by controlling the stability of key centrosomal proteins and facilitating PLK4 localization. Autosomal recessive mutations cause cone-rod dystrophy and hearing loss, affecting both retinal and auditory function through defects in primary cilia. The gene shows extremely low constraint against loss-of-function variants (pLI near zero), consistent with its recessive inheritance pattern.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 1.021 OMIM phenotype
Clinical SummaryCEP78
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Gene-Disease Validity (ClinGen)
cone-rod dystrophy and hearing loss · ARStrong

Strong evidence — appropriate for clinical testing

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.02LOEUF
pLI 0.000
Z-score 1.48
OE 0.71 (0.511.02)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.58Z-score
OE missense 1.09 (1.001.18)
383 obs / 352.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.71 (0.511.02)
00.351.4
Missense OE1.09 (1.001.18)
00.61.4
Synonymous OE0.81
01.21.6
LoF obs/exp: 22 / 30.9Missense obs/exp: 383 / 352.4Syn Z: 1.71
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveCEP78-related cone-rod dystrophy and hearing lossLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6259th %ile
GOF
0.6443th %ile
LOF
0.4233th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CEP78 · 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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