PLPBP

Chr 8AR

pyridoxal phosphate binding protein

Also known as: EPEO1, EPVB6D, PROSC

The protein binds pyridoxal 5'-phosphate and regulates intracellular homeostasis of this active form of vitamin B6. Mutations cause early-onset, vitamin B6-dependent epilepsy that follows autosomal recessive inheritance. The gene shows tolerance to loss-of-function variants (low pLI score), which is consistent with the recessive inheritance pattern observed clinically.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.701 OMIM phenotype
Clinical SummaryPLPBP
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.33) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.70LOEUF
pLI 0.040
Z-score 2.41
OE 0.33 (0.170.70)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.98Z-score
OE missense 0.78 (0.680.91)
125 obs / 159.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.33 (0.170.70)
00.351.4
Missense OE0.78 (0.680.91)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 5 / 15.1Missense obs/exp: 125 / 159.9Syn Z: -0.38
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongPLPBP-related vitamin-B6-dependent epilepsyLOFAR

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

DN
0.6357th %ile
GOF
0.4480th %ile
LOF
0.3355th %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.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PLPBP · 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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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC