PPM1D

Chr 17AD

protein phosphatase, Mg2+/Mn2+ dependent 1D

Also known as: IDDGIP, JDVS, PP2C-DELTA, WIP1

This Ser/Thr protein phosphatase negatively regulates the p53 stress response pathway by dephosphorylating p53, CHEK1, and p38 MAP kinase, thereby controlling cell cycle checkpoints and apoptosis. Mutations cause Jansen-de Vries syndrome, a neurodevelopmental disorder, through autosomal dominant inheritance. The gene is extremely intolerant to loss-of-function variants (pLI ~0), indicating that complete loss of protein function is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 1.102 OMIM phenotypes
Clinical SummaryPPM1D
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Gene-Disease Validity (ClinGen)
syndromic intellectual disability · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

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
1.10LOEUF
pLI 0.000
Z-score 1.17
OE 0.75 (0.531.10)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
2.70Z-score
OE missense 0.59 (0.520.66)
200 obs / 340.3 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.75 (0.531.10)
00.351.4
Missense OE0.59 (0.520.66)
00.61.4
Synonymous OE0.86
01.21.6
LoF obs/exp: 20 / 26.5Missense obs/exp: 200 / 340.3Syn Z: 1.22
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePPM1D-related developmental disorder (Jansen-de Vries syndrome)LOFAD
DN
0.5279th %ile
GOF
0.5857th %ile
LOF
0.56top 25%

The highest-scoring mechanism for this gene is gain-of-function.

GOF1 literature citation

Literature Evidence

GOFThese mutants code for gain-of-function PPM1D with retained phosphatase activity.PMID:25742468

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PPM1D · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗