UPB1

Chr 22AR

beta-ureidopropionase 1

Also known as: BUP1

Beta-ureidopropionase catalyzes the final step in pyrimidine degradation, converting N-carbamoyl-beta-alanine to beta-alanine, ammonia, and carbon dioxide. Mutations cause beta-ureidopropionase deficiency, an autosomal recessive disorder associated with N-carbamyl-beta-amino aciduria and neurological abnormalities. The gene shows low constraint to loss-of-function variation (pLI near zero), consistent with recessive inheritance.

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismARLOEUF 1.261 OMIM phenotype
Clinical SummaryUPB1
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
1.26LOEUF
pLI 0.000
Z-score 0.63
OE 0.86 (0.601.26)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.87Z-score
OE missense 1.16 (1.051.29)
261 obs / 224.5 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.86 (0.601.26)
00.351.4
Missense OE1.16 (1.051.29)
00.61.4
Synonymous OE1.19
01.21.6
LoF obs/exp: 19 / 22.2Missense obs/exp: 261 / 224.5Syn Z: -1.42
DN
0.6162th %ile
GOF
0.5661th %ile
LOF
0.3842th %ile

The highest-scoring mechanism for this gene is dominant-negative.

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

UPB1 · 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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