CPPED1

Chr 16

calcineurin like phosphoesterase domain containing 1

Also known as: CSTP1

The protein is a phosphatase that dephosphorylates AKT at serine-473, blocking cell cycle progression and promoting apoptosis, and may inhibit glucose uptake in adipocytes. Mutations cause autosomal recessive developmental and epileptic encephalopathy with onset in infancy, characterized by severe intellectual disability, seizures, and progressive microcephaly. The gene shows minimal constraint against loss-of-function variants in population databases.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 1.45
Clinical SummaryCPPED1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
16 unique Pathogenic / Likely Pathogenic· 77 VUS of 112 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.45LOEUF
pLI 0.000
Z-score 0.40
OE 0.88 (0.551.45)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.96Z-score
OE missense 1.19 (1.071.33)
232 obs / 194.5 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.88 (0.551.45)
00.351.4
Missense OE1.19 (1.071.33)
00.61.4
Synonymous OE1.21
01.21.6
LoF obs/exp: 11 / 12.5Missense obs/exp: 232 / 194.5Syn Z: -1.56

ClinVar Variant Classifications

112 submitted variants in ClinVar

Classification Summary

Pathogenic14
Likely Pathogenic2
VUS77
Likely Benign4
14
Pathogenic
2
Likely Pathogenic
77
VUS
4
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
14
0
14
Likely Pathogenic
0
0
2
0
2
VUS
0
67
10
0
77
Likely Benign
0
3
1
0
4
Benign
0
0
0
0
0
Total07027097

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

View in ClinVar →

Protein Context — Lollipop Plot

CPPED1 · 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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