The protein encoded by this gene functions to recycle protein-bound biotin by cleaving biocytin (biotin-epsilon-lysine), a normal product of carboxylase degradation, resulting in regeneration of free biotin. The encoded protein has also been shown to have biotinyl transferase activity. Mutations in this gene are associated with biotinidase deficiency. Multiple transcript variants encoding different isoforms have been described. [provided by RefSeq, Aug 2013]

OMIMResearchGenerating clinical summary…
LOFmechanismARLOEUF 1.301 OMIM phenotype
Clinical SummaryBTD
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Gene-Disease Validity (ClinGen)
Leigh syndrome · ARModerate

Moderate evidence — consider for supplementary testing

2 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
1 active or recruiting trial — 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?
1.30LOEUF
pLI 0.000
Z-score 0.70
OE 0.81 (0.521.30)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint?
-0.53Z-score
OE missense 1.09 (0.991.19)
320 obs / 294.6 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios?
LoF OE?0.81 (0.521.30)
00.351.4
Missense OE?1.09 (0.991.19)
00.61.4
Synonymous OE?1.07
01.21.6
LoF obs/exp: 12 / 14.9Missense obs/exp: 320 / 294.6Syn Z: -0.58

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

BTD · protein map & ClinVar variants

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