PHLPP1

Chr 18

PH domain and leucine rich repeat protein phosphatase 1

Also known as: PHLPP, PLEKHE1, PPM3A, SCOP

This gene encodes a serine/threonine protein phosphatase that regulates cell survival and apoptosis by dephosphorylating AKT, protein kinase C, and other signaling proteins involved in neuronal function and memory formation. Mutations cause autosomal dominant intellectual disability with seizures and behavioral abnormalities, typically presenting in early childhood. The gene is highly constrained against loss-of-function mutations, indicating that proper dosage is critical for normal neurodevelopment.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
18
Pubs (1 yr)
61
P/LP submissions
0%
P/LP missense
0.26
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryPHLPP1
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
59 unique Pathogenic / Likely Pathogenic· 210 VUS of 300 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.26LOEUF
pLI 0.999
Z-score 5.95
OE 0.14 (0.080.26)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
1.75Z-score
OE missense 0.83 (0.780.88)
691 obs / 833.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.14 (0.080.26)
00.351.4
Missense OE0.83 (0.780.88)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 8 / 56.1Missense obs/exp: 691 / 833.0Syn Z: -0.26
DN
0.3892th %ile
GOF
0.5661th %ile
LOF
0.73top 10%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.26

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic54
Likely Pathogenic5
VUS210
Likely Benign8
Benign4
54
Pathogenic
5
Likely Pathogenic
210
VUS
8
Likely Benign
4
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
54
0
54
Likely Pathogenic
0
0
5
0
5
VUS
0
206
4
0
210
Likely Benign
0
7
1
0
8
Benign
0
1
1
2
4
Total0214652281

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

View in ClinVar →

Protein Context — Lollipop Plot

PHLPP1 · 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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