Document Detail


SHARPIN forms a linear ubiquitin ligase complex regulating NF-κB activity and apoptosis.
MedLine Citation:
PMID:  21455181     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
SHARPIN is a ubiquitin-binding and ubiquitin-like-domain-containing protein which, when mutated in mice, results in immune system disorders and multi-organ inflammation. Here we report that SHARPIN functions as a novel component of the linear ubiquitin chain assembly complex (LUBAC) and that the absence of SHARPIN causes dysregulation of NF-κB and apoptotic signalling pathways, explaining the severe phenotypes displayed by chronic proliferative dermatitis (cpdm) in SHARPIN-deficient mice. Upon binding to the LUBAC subunit HOIP (also known as RNF31), SHARPIN stimulates the formation of linear ubiquitin chains in vitro and in vivo. Coexpression of SHARPIN and HOIP promotes linear ubiquitination of NEMO (also known as IKBKG), an adaptor of the IκB kinases (IKKs) and subsequent activation of NF-κB signalling, whereas SHARPIN deficiency in mice causes an impaired activation of the IKK complex and NF-κB in B cells, macrophages and mouse embryonic fibroblasts (MEFs). This effect is further enhanced upon concurrent downregulation of HOIL-1L (also known as RBCK1), another HOIP-binding component of LUBAC. In addition, SHARPIN deficiency leads to rapid cell death upon tumour-necrosis factor α (TNF-α) stimulation via FADD- and caspase-8-dependent pathways. SHARPIN thus activates NF-κB and inhibits apoptosis via distinct pathways in vivo.
Authors:
Fumiyo Ikeda; Yonathan Lissanu Deribe; Sigrid S Skånland; Benjamin Stieglitz; Caroline Grabbe; Mirita Franz-Wachtel; Sjoerd J L van Wijk; Panchali Goswami; Vanja Nagy; Janos Terzic; Fuminori Tokunaga; Ariadne Androulidaki; Tomoko Nakagawa; Manolis Pasparakis; Kazuhiro Iwai; John P Sundberg; Liliana Schaefer; Katrin Rittinger; Boris Macek; Ivan Dikic
Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Nature     Volume:  471     ISSN:  1476-4687     ISO Abbreviation:  Nature     Publication Date:  2011 Mar 
Date Detail:
Created Date:  2011-04-01     Completed Date:  2011-05-25     Revised Date:  2012-04-04    
Medline Journal Info:
Nlm Unique ID:  0410462     Medline TA:  Nature     Country:  England    
Other Details:
Languages:  eng     Pagination:  637-41     Citation Subset:  IM    
Affiliation:
Frankfurt Institute for Molecular Life Sciences and Institute of Biochemistry II, Goethe University School of Medicine, Theodor-Stern-Kai 7, D-60590 Frankfurt, Main, Germany.
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MeSH Terms
Descriptor/Qualifier:
Animals
Apoptosis* / drug effects
B-Lymphocytes / metabolism
Carrier Proteins / metabolism
Caspase 8 / metabolism
Cells, Cultured
Dermatitis / genetics,  metabolism,  pathology
Fas-Associated Death Domain Protein / metabolism
Fibroblasts / metabolism
HEK293 Cells
HeLa Cells
Humans
I-kappa B Kinase / metabolism
Intracellular Signaling Peptides and Proteins / metabolism
Macrophages / metabolism
Mice
NF-kappa B / metabolism*
Nerve Tissue Proteins / deficiency,  genetics,  metabolism*
Tumor Necrosis Factor-alpha / metabolism,  pharmacology
Ubiquitin / metabolism*
Ubiquitin-Protein Ligase Complexes / metabolism*
Ubiquitin-Protein Ligases / metabolism
Ubiquitination
Grant Support
ID/Acronym/Agency:
AR049288/AR/NIAMS NIH HHS; R01 AR049288-07/AR/NIAMS NIH HHS
Chemical
Reg. No./Substance:
0/Carrier Proteins; 0/Fadd protein, mouse; 0/Fas-Associated Death Domain Protein; 0/HOIL-1L protein, mouse; 0/Intracellular Signaling Peptides and Proteins; 0/NEMO protein, mouse; 0/NF-kappa B; 0/Nerve Tissue Proteins; 0/Tumor Necrosis Factor-alpha; 0/Ubiquitin; 0/sharpin; EC 2.7.11.10/I-kappa B Kinase; EC 3.4.22.-/Casp8 protein, mouse; EC 3.4.22.-/Caspase 8; EC 6.3.2.19/PAUL protein, mouse; EC 6.3.2.19/Ubiquitin-Protein Ligase Complexes; EC 6.3.2.19/Ubiquitin-Protein Ligases

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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