CEP192

Chr 18

centrosomal protein 192

Also known as: PPP1R62

CEP192 encodes a centrosome protein required for mitotic centrosome maturation, bipolar spindle assembly, and serves as an activating scaffold for key mitotic kinases AURKA and PLK1. Mutations cause autosomal recessive primary microcephaly with onset apparent at birth, reflecting the gene's critical role in proper cell division during brain development. This gene is highly constrained against loss-of-function variants in the general population.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
8
Pubs (1 yr)
89
P/LP submissions
0%
P/LP missense
0.60
LOEUF
Mechanism
Clinical SummaryCEP192
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
85 unique Pathogenic / Likely Pathogenic· 234 VUS of 406 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.60LOEUF
pLI 0.000
Z-score 5.20
OE 0.48 (0.390.60)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.00Z-score
OE missense 0.92 (0.880.97)
1193 obs / 1294.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.48 (0.390.60)
00.351.4
Missense OE0.92 (0.880.97)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 57 / 118.0Missense obs/exp: 1193 / 1294.0Syn Z: 0.76

ClinVar Variant Classifications

406 submitted variants in ClinVar

Classification Summary

Pathogenic83
Likely Pathogenic2
VUS234
Likely Benign24
Benign6
83
Pathogenic
2
Likely Pathogenic
234
VUS
24
Likely Benign
6
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
83
0
83
Likely Pathogenic
0
0
2
0
2
VUS
0
230
4
0
234
Likely Benign
0
20
1
3
24
Benign
0
4
0
2
6
Total0254905349

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

View in ClinVar →

Protein Context — Lollipop Plot

CEP192 · 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.

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Clinical Literature
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