CYP2U1

Chr 4AR

cytochrome P450 family 2 subfamily U member 1

Also known as: P450TEC, SPG49, SPG56

This gene encodes a cytochrome P450 enzyme that hydroxylates long chain fatty acids including arachidonic acid and docosahexaenoic acid. Mutations cause spastic paraplegia 56, an autosomal recessive disorder. The pathogenic mechanism involves loss of function of this fatty acid-metabolizing enzyme.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.751 OMIM phenotype
Clinical SummaryCYP2U1
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Gene-Disease Validity (ClinGen)
hereditary spastic paraplegia · 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.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.75LOEUF
pLI 0.001
Z-score 2.39
OE 0.41 (0.240.75)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.99Z-score
OE missense 0.83 (0.750.93)
233 obs / 279.8 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.41 (0.240.75)
00.351.4
Missense OE0.83 (0.750.93)
00.61.4
Synonymous OE0.92
01.21.6
LoF obs/exp: 8 / 19.3Missense obs/exp: 233 / 279.8Syn Z: 0.64
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveCYP2U1-related spastic paraplegiaLOFAR

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

DN
0.75top 25%
GOF
0.6932th %ile
LOF
0.2776th %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

CYP2U1 · 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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