CCDC15

Chr 11

coiled-coil domain containing 15

CCDC15 encodes a protein that regulates primary cilium assembly, maintenance, and length by interacting with centriole inner scaffold proteins to promote proper centriole size and integrity. Mutations cause autosomal recessive primary ciliary dyskinesia with left-right asymmetry defects. The gene shows tolerance to loss-of-function variants (LOEUF 1.057), consistent with the recessive inheritance pattern observed clinically.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
2
Pubs (1 yr)
60
P/LP submissions
0%
P/LP missense
1.06
LOEUF
DN
Mechanism· predicted
Clinical SummaryCCDC15
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
60 unique Pathogenic / Likely Pathogenic· 131 VUS of 214 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.06LOEUF
pLI 0.000
Z-score 1.24
OE 0.80 (0.611.06)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.13Z-score
OE missense 0.98 (0.911.06)
420 obs / 427.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.80 (0.611.06)
00.351.4
Missense OE0.98 (0.911.06)
00.61.4
Synonymous OE0.77
01.21.6
LoF obs/exp: 35 / 43.9Missense obs/exp: 420 / 427.6Syn Z: 2.25
DN
0.7131th %ile
GOF
0.6247th %ile
LOF
0.3647th %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

214 submitted variants in ClinVar

Classification Summary

Pathogenic58
Likely Pathogenic2
VUS131
Likely Benign9
Benign1
Conflicting1
58
Pathogenic
2
Likely Pathogenic
131
VUS
9
Likely Benign
1
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
58
0
58
Likely Pathogenic
0
0
2
0
2
VUS
1
123
7
0
131
Likely Benign
0
8
0
1
9
Benign
0
0
0
1
1
Conflicting
1
Total1131672202

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

View in ClinVar →

Protein Context — Lollipop Plot

CCDC15 · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 3 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC