CCDC102B

Chr 18

coiled-coil domain containing 102B

Also known as: ACY1L, AN, C18orf14, HsT1731

The protein maintains centrosome cohesion by forming fibers at the proximal ends of centrioles during interphase and dissociates during mitosis to allow centrosome separation. Mutations cause autosomal recessive retinal dystrophy and Bardet-Biedl syndrome, both ciliopathies affecting the retina and other organ systems. The gene shows tolerance to loss-of-function variation, consistent with recessive inheritance patterns.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
4
Pubs (1 yr)
133
P/LP submissions
0%
P/LP missense
1.29
LOEUF
DN
Mechanism· predicted
Clinical SummaryCCDC102B
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
131 unique Pathogenic / Likely Pathogenic· 104 VUS of 265 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.29LOEUF
pLI 0.000
Z-score 0.35
OE 0.93 (0.671.29)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.55Z-score
OE missense 1.10 (0.991.21)
280 obs / 255.2 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.93 (0.671.29)
00.351.4
Missense OE1.10 (0.991.21)
00.61.4
Synonymous OE0.90
01.21.6
LoF obs/exp: 25 / 27.0Missense obs/exp: 280 / 255.2Syn Z: 0.75
DN
0.77top 25%
GOF
0.6150th %ile
LOF
0.3745th %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

265 submitted variants in ClinVar

Classification Summary

Pathogenic120
Likely Pathogenic11
VUS104
Likely Benign11
Benign8
Conflicting1
120
Pathogenic
11
Likely Pathogenic
104
VUS
11
Likely Benign
8
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
120
0
120
Likely Pathogenic
0
0
11
0
11
VUS
0
81
23
0
104
Likely Benign
0
5
4
2
11
Benign
0
5
2
1
8
Conflicting
1
Total0911603255

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

View in ClinVar →

Protein Context — Lollipop Plot

CCDC102B · 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 →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC