PEX2

Chr 8AR

peroxisomal biogenesis factor 2

Also known as: PAF1, PBD5A, PBD5B, PMP3, PMP35, PXMP3, RNF72, ZWS3

The protein is an integral peroxisomal membrane protein required for peroxisome biogenesis and peroxisomal matrix protein import. Mutations cause peroxisome biogenesis disorders including Zellweger syndrome and infantile Refsum disease through autosomal recessive inheritance. The pathogenic mechanism involves dominant-negative effects disrupting normal peroxisome function.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 1.202 OMIM phenotypes
Clinical SummaryPEX2
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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.
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Clinical Trials
3 active or recruiting trials — 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
1.20LOEUF
pLI 0.000
Z-score 1.11
OE 0.64 (0.361.20)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.05Z-score
OE missense 0.99 (0.871.13)
152 obs / 153.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.64 (0.361.20)
00.351.4
Missense OE0.99 (0.871.13)
00.61.4
Synonymous OE0.85
01.21.6
LoF obs/exp: 7 / 11.0Missense obs/exp: 152 / 153.6Syn Z: 0.92
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePEX2-related peroxisome biogenesis disorder 5A (Zellweger)LOFAR
definitivePEX2-related peroxisome biogenesis disorder 5BLOFAR

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

DN
0.7130th %ile
GOF
0.4875th %ile
LOF
0.3259th %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

PEX2 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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