PEX1

Chr 7AR

peroxisomal biogenesis factor 1

Also known as: HMLR1, PBD1A, PBD1B, ZWS, ZWS1

The protein functions as an AAA ATPase that forms heteromeric complexes at peroxisomal membranes to import proteins into peroxisomes and facilitate peroxisome biogenesis. Mutations cause autosomal recessive peroxisome biogenesis disorders including Zellweger syndrome, neonatal adrenoleukodystrophy, infantile Refsum disease, and Heimler syndrome 1. The pathogenic mechanism involves loss of function leading to defective peroxisomal protein import and impaired peroxisome assembly.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.663 OMIM phenotypes
Clinical SummaryPEX1
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Gene-Disease Validity (ClinGen)
peroxisome biogenesis disorder due to PEX1 defect · 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
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GeneReview available — PEX1
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
0.66LOEUF
pLI 0.000
Z-score 3.86
OE 0.50 (0.380.66)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.14Z-score
OE missense 0.88 (0.820.94)
582 obs / 664.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.50 (0.380.66)
00.351.4
Missense OE0.88 (0.820.94)
00.61.4
Synonymous OE0.92
01.21.6
LoF obs/exp: 35 / 69.8Missense obs/exp: 582 / 664.4Syn Z: 0.96
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongPEX1-related Heimler syndromeOTHERAR
definitivePEX1-related peroxisome biogenesis disorderLOFAR

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

DN
0.6839th %ile
GOF
0.4777th %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

PEX1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗
Key Publications
Landmark & review papers · by relevance
PubMed
Heimler Syndrome.
Mechaussier S et al.·Adv Exp Med Biol
2020Review
Ophthalmic Manifestations of Heimler Syndrome in Two Siblings With PEX1 Variants.
Miranda V et al.·J Pediatr Ophthalmol Strabismus
2024Review
Neonatal Cholestasis: Exploring Genetic Causes and Clinical Outcomes.
Gürcan Kaya N et al.·J Paediatr Child Health
2025
Top 5 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC