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STAMBP

Domain

The JAMM motif is essential for the protease activity.

Function

Zinc metalloprotease that specifically cleaves 'Lys-63'-linked polyubiquitin chains (PubMed:15314065, PubMed:23542699, PubMed:34425109). Does not cleave 'Lys-48'-linked polyubiquitin chains (PubMed:15314065). Plays a role in signal transduction for cell growth and MYC induction mediated by IL-2 and GM-CSF (PubMed:10383417). Potentiates BMP (bone morphogenetic protein) signaling by antagonizing the inhibitory action of SMAD6 and SMAD7 (PubMed:11483516). Has a key role in regulation of cell surface receptor-mediated endocytosis and ubiquitin-dependent sorting of receptors to lysosomes (PubMed:15314065, PubMed:17261583). Endosomal localization of STAMBP is required for efficient EGFR degradation but not for its internalization (PubMed:15314065, PubMed:17261583). Involved in the negative regulation of PI3K-AKT-mTOR and RAS-MAP signaling pathways (PubMed:23542699).

Involvement in disease

Microcephaly-capillary malformation syndrome

MICCAP

A congenital disorder characterized by severe progressive microcephaly, early-onset refractory epilepsy, profound developmental delay, and multiple small capillary malformations spread diffusely on the body. Additional more variable features include dysmorphic facial features, distal limb abnormalities, and mild heart defects.

None

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

Post-translational modifications

Phosphorylated after BMP type I receptor activation.

Ubiquitinated by SMURF2 in the presence of RNF11.

Sequence Similarities

Belongs to the peptidase M67C family.

Tissue Specificity

Ubiquitously expressed.

Cellular localization

Alternative names

AMSH, STAMBP, STAM-binding protein, Associated molecule with the SH3 domain of STAM, Endosome-associated ubiquitin isopeptidase

swissprot:O95630 omim:606247 entrezGene:10617