PEX19

Chr 1AR

peroxisomal biogenesis factor 19

Also known as: D1S2223E, HK33, PBD12A, PMP1, PMPI, PXF, PXMP1

PEX19 encodes a cytosolic chaperone and import receptor that is essential for importing peroxisomal membrane proteins and early peroxisomal biogenesis. Mutations cause Zellweger syndrome and other peroxisome biogenesis disorders through an autosomal recessive inheritance pattern, resulting in defective import of peroxisomal matrix proteins and multiple peroxisomal dysfunction. These disorders are characterized as lethal conditions with multiple organ system involvement due to the failure of peroxisome assembly and function.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.941 OMIM phenotype
Clinical SummaryPEX19
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Gene-Disease Validity (ClinGen)
peroxisome biogenesis disorder · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📖
GeneReview available — PEX19
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.94LOEUF
pLI 0.000
Z-score 1.75
OE 0.52 (0.300.94)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.78Z-score
OE missense 1.17 (1.041.32)
190 obs / 162.2 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.52 (0.300.94)
00.351.4
Missense OE1.17 (1.041.32)
00.61.4
Synonymous OE1.11
01.21.6
LoF obs/exp: 8 / 15.4Missense obs/exp: 190 / 162.2Syn Z: -0.67
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePEX19-related peroxisome biogenesis disorderLOFAR

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

DN
0.6745th %ile
GOF
0.6443th %ile
LOF
0.1698th %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

PEX19 · 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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