BCKDHB

Chr 6

branched chain keto acid dehydrogenase E1 subunit beta

Also known as: BCKDE1B, BCKDH E1-beta, E1B, MSUD1B, OVD1B

This gene encodes the E1 beta subunit of branched-chain keto acid dehydrogenase, which is a multienzyme complex associated with the inner membrane of mitochondria. This enzyme complex functions in the catabolism of branched-chain amino acids. Mutations in this gene have been associated with maple syrup urine disease (MSUD), type 1B, a disease characterized by a maple syrup odor to the urine in addition to mental and physical retardation and feeding problems. Alternative splicing at this locus results in multiple transcript variants. [provided by RefSeq, Jan 2016]

GeneReviewsResearchGenerating clinical summary…

Primary Disease Associations & Inheritance

UniProtMaple syrup urine disease 1B

Clinical highlights

Gene-disease validity (ClinGen)
maple syrup urine disease type 1B · ARDefinitivesufficient evidence for diagnostic panels
Interpreting a novel variant
Loss of function is the curated mechanism (Gene2Phenotype), though the gene is not strongly LoF-constrained in the population — weigh truncating variants against that tolerance.Curated gene-level mechanism — a prior for triage, not a per-variant call.
0
Active trials
19
Pubs (1 yr)
P/LP submissions
P/LP missense
1.10
LOEUF
LOF
Mechanism· G2P
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GeneReview available — BCKDHB
Authoritative clinical overview · Recommended first read
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Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?
1.10LOEUF
pLI 0.000
Z-score 1.19
OE 0.73 (0.491.10)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint?
-0.18Z-score
OE missense 1.03 (0.931.15)
226 obs / 218.7 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios?
LoF OE?0.73 (0.491.10)
00.351.4
Missense OE?1.03 (0.931.15)
00.61.4
Synonymous OE?1.11
01.21.6
LoF obs/exp: 16 / 22.0Missense obs/exp: 226 / 218.7Syn Z: -0.77

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

BCKDHB · 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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