CEP55

Chr 10AR

centrosomal protein 55

Also known as: C10orf3, CT111, MARCH, URCC6

CEP55 encodes a protein that recruits PDCD6IP and TSG101 to the midbody during cytokinesis and is required for successful completion of cell division. Mutations cause multinucleated neurons, anhydramnios, renal dysplasia, cerebellar hypoplasia, and hydranencephaly through autosomal recessive inheritance. This gene is extremely intolerant to loss-of-function variants and affects both brain and kidney development.

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismARLOEUF 1.081 OMIM phenotype
Clinical SummaryCEP55
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
1 active or recruiting trial — 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
1.08LOEUF
pLI 0.000
Z-score 1.22
OE 0.76 (0.541.08)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.50Z-score
OE missense 0.91 (0.811.02)
211 obs / 232.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.76 (0.541.08)
00.351.4
Missense OE0.91 (0.811.02)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 22 / 29.1Missense obs/exp: 211 / 232.3Syn Z: 0.68
DN
0.6356th %ile
GOF
0.5367th %ile
LOF
0.4038th %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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CEP55 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
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 ↗