PEX5L

Chr 3

peroxisomal biogenesis factor 5 like

Also known as: PEX5R, PEX5RP, PXR2, PXR2B, TRIP8b

Enables peroxisome matrix targeting signal-1 binding activity and small GTPase binding activity. Predicted to be involved in protein import into peroxisome matrix, docking. Predicted to act upstream of or within maintenance of protein location and regulation of membrane potential. Located in cytosol. Part of receptor complex. [provided by Alliance of Genome Resources, Jul 2025]

OMIMResearchGenerating clinical summary…
MultiplemechanismLOEUF 0.49
Clinical SummaryPEX5L
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.30) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint?
0.49LOEUF
pLI 0.008
Z-score 3.98
OE 0.30 (0.180.49)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint?
2.85Z-score
OE missense 0.57 (0.500.64)
193 obs / 340.8 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios?
LoF OE?0.30 (0.180.49)
00.351.4
Missense OE?0.57 (0.500.64)
00.61.4
Synonymous OE?0.96
01.21.6
LoF obs/exp: 11 / 37.2Missense obs/exp: 193 / 340.8Syn Z: 0.37

This gene — mechanism propensity

DN
0.6454th %ile
GOF
0.6833th %ile
LOF
0.3550th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median
DNprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PEX5L · 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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