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HIBCH

Function

Hydrolyzes 3-hydroxyisobutyryl-CoA (HIBYL-CoA), a saline catabolite. Has high activity toward isobutyryl-CoA. Could be an isobutyryl-CoA dehydrogenase that functions in valine catabolism. Also hydrolyzes 3-hydroxypropanoyl-CoA.

Involvement in disease

3-hydroxyisobutryl-CoA hydrolase deficiency

HIBCHD

An autosomal recessive inborn error of valine metabolism. It causes severely delayed psychomotor development, neurodegeneration, increased lactic acid, and brain lesions in the basal ganglia.

None

The disease is caused by variants affecting the gene represented in this entry.

Pathway

Amino-acid degradation; L-valine degradation.

Sequence Similarities

Belongs to the enoyl-CoA hydratase/isomerase family.

Tissue Specificity

Highly expressed in liver and kidney, also detected in heart, muscle and brain (at protein level). Not detected in lung.

Cellular localization

Alternative names

3-hydroxyisobutyryl-coenzyme A hydrolase, HIB-CoA hydrolase, HIBYL-CoA-H, HIBCH

swissprot:Q6NVY1 entrezGene:26275 omim:610690