PI4KA

Chr 22AR

phosphatidylinositol 4-kinase alpha

Also known as: GIDID2, PI4K-ALPHA, PIK4CA, PMGYCHA, SPG84, pi4K230

This protein catalyzes the phosphorylation of phosphatidylinositol to produce phosphatidylinositol 4-phosphate, the first committed step in generating the second messenger inositol-1,4,5-trisphosphate. Autosomal recessive mutations cause a spectrum of disorders including gastrointestinal defects with immunodeficiency, perisylvian polymicrogyria with cerebellar hypoplasia and arthrogryposis, and spastic paraplegia. Loss of function disrupts phosphoinositide signaling pathways critical for normal development and cellular function.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.463 OMIM phenotypes
Clinical SummaryPI4KA
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
34 unique Pathogenic / Likely Pathogenic· 128 VUS of 300 total submissions
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GeneReview available — PI4KA
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Missense constrained — critical functional residues
LoF Constraint
0.46LOEUF
pLI 0.000
Z-score 6.53
OE 0.36 (0.280.46)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
3.53Z-score
OE missense 0.72 (0.680.76)
883 obs / 1231.3 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.36 (0.280.46)
00.351.4
Missense OE0.72 (0.680.76)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 43 / 120.3Missense obs/exp: 883 / 1231.3Syn Z: -0.24
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePI4KA-related polymicrogyria, perisylvian, with cerebellar hypoplasia and arthrogryposisLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6453th %ile
GOF
0.6150th %ile
LOF
0.3842th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

Pathogenic22
Likely Pathogenic12
VUS128
Likely Benign71
Benign7
Conflicting1
22
Pathogenic
12
Likely Pathogenic
128
VUS
71
Likely Benign
7
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
0
21
0
22
Likely Pathogenic
8
4
0
0
12
VUS
5
107
16
0
128
Likely Benign
0
10
9
52
71
Benign
0
1
4
2
7
Conflicting
1
Total141225054241

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

View in ClinVar →

Protein Context — Lollipop Plot

PI4KA · 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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