{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,28]],"date-time":"2026-07-28T16:22:04Z","timestamp":1785255724142,"version":"3.55.0"},"reference-count":62,"publisher":"The Company of Biologists","issue":"13","license":[{"start":{"date-parts":[[2023,7,1]],"date-time":"2023-07-01T00:00:00Z","timestamp":1688169600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["T32GM007739"],"award-info":[{"award-number":["T32GM007739"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"crossref","award":["R35 GM134907"],"award-info":[{"award-number":["R35 GM134907"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"crossref"}]},{"name":"National Cancer Institute","award":["P30 CA008748"],"award-info":[{"award-number":["P30 CA008748"]}]},{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"crossref","award":["EXC 2145 SyNergy ID 390857198"],"award-info":[{"award-number":["EXC 2145 SyNergy ID 390857198"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,7,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Tylosis with oesophageal cancer (TOC) is a rare familial disorder caused by cytoplasmic mutations in inactive rhomboid 2 (iRhom2 or iR2, encoded by Rhbdf2). iR2 and the related iRhom1 (or iR1, encoded by Rhbdf1) are key regulators of the membrane-anchored metalloprotease ADAM17, which is required for activating EGFR ligands and for releasing pro-inflammatory cytokines such as TNF\u03b1 (or TNF). A cytoplasmic deletion in iR2, including the TOC site, leads to curly coat or bare skin (cub) in mice, whereas a knock-in TOC mutation (toc) causes less severe alopecia and wavy fur. The abnormal skin and hair phenotypes of iR2cub\/cub and iR2toc\/toc mice depend on amphiregulin (Areg) and Adam17, as loss of one allele of either gene rescues the fur phenotypes. Remarkably, we found that iR1\u2212\/\u2212 iR2cub\/cub mice survived, despite a lack of mature ADAM17, whereas iR2cub\/cub Adam17\u2212\/\u2212 mice died perinatally, suggesting that the iR2cub gain-of-function mutation requires the presence of ADAM17, but not its catalytic activity. The iR2toc mutation did not substantially reduce the levels of mature ADAM17, but instead affected its function in a substrate-selective manner. Our findings provide new insights into the role of the cytoplasmic domain of iR2 in vivo, with implications for the treatment of TOC patients.<\/jats:p>","DOI":"10.1242\/jcs.260910","type":"journal-article","created":{"date-parts":[[2023,6,7]],"date-time":"2023-06-07T08:11:04Z","timestamp":1686125464000},"source":"Crossref","is-referenced-by-count":10,"title":["Analysis of the function of ADAM17 in iRhom2 curly-bare and tylosis with esophageal cancer mutant mice"],"prefix":"10.1242","volume":"136","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0896-2119","authenticated-orcid":false,"given":"Ariana I.","family":"Rabinowitsch","sequence":"first","affiliation":[{"name":"Memorial Sloan Kettering Cancer Center, Weill Cornell Medicine, The Rockefeller University 1 Tri-Institutional MD\/PhD Program , , New York, NY 10021 , USA"},{"name":"Weill Cornell Medicine 2 Program in Biochemistry, Cellular and Molecular Biology , , New York, NY 10021 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1282-6080","authenticated-orcid":false,"given":"Thorsten","family":"Maretzky","sequence":"additional","affiliation":[{"name":"Roy J. and Lucille A. Carver College of Medicine, University of Iowa 3 Inflammation Program and Department of Internal Medicine , , Iowa City, IA 52242 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gisela","family":"Weskamp","sequence":"additional","affiliation":[{"name":"Hospital for Special Surgery 4 Arthritis and Tissue Degeneration Program , , New York, NY 10021 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5463-3546","authenticated-orcid":false,"given":"Coline","family":"Haxaire","sequence":"additional","affiliation":[{"name":"Hospital for Special Surgery 4 Arthritis and Tissue Degeneration Program , , New York, NY 10021 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johanna","family":"Tueshaus","sequence":"additional","affiliation":[{"name":"German Center for Neurodegenerative Diseases (DZNE) 5 , 81377 Munich , Germany"},{"name":"Neuroproteomics, School of Medicine, Klinikum rechts der Isar, Technical University of Munich 6 , 81675 Munich , Germany"},{"name":"Institute for Advanced Study, Technical University of Munich 7 , 85748 Garching , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2211-2575","authenticated-orcid":false,"given":"Stefan F.","family":"Lichtenthaler","sequence":"additional","affiliation":[{"name":"German Center for Neurodegenerative Diseases (DZNE) 5 , 81377 Munich , Germany"},{"name":"Neuroproteomics, School of Medicine, Klinikum rechts der Isar, Technical University of Munich 6 , 81675 Munich , Germany"},{"name":"Institute for Advanced Study, Technical University of Munich 7 , 85748 Garching , Germany"},{"name":"Munich Cluster for Systems Neurology (SyNergy) 8 , 81377 Munich , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2841-6887","authenticated-orcid":false,"given":"S\u00e9bastien","family":"Monette","sequence":"additional","affiliation":[{"name":"Hospital for Special Surgery, Memorial Sloan Kettering Cancer Center, The Rockefeller University, Weill Cornell Medicine 9 Tri-Institutional Laboratory of Comparative Pathology , , New York, NY 10021 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5053-3137","authenticated-orcid":false,"given":"Carl P.","family":"Blobel","sequence":"additional","affiliation":[{"name":"Memorial Sloan Kettering Cancer Center, Weill Cornell Medicine, The Rockefeller University 1 Tri-Institutional MD\/PhD Program , , New York, NY 10021 , USA"},{"name":"Hospital for Special Surgery 4 Arthritis and Tissue Degeneration Program , , New York, NY 10021 , USA"},{"name":"Institute for Advanced Study, Technical University of Munich 7 , 85748 Garching , Germany"},{"name":"Weill Cornell Medicine 10 Department of Physiology, Biophysics and Systems Biology , , New York, NY 10021 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2023,7,7]]},"reference":[{"key":"2023070709252974200_JCS260910C1","doi-asserted-by":"publisher","first-page":"489","DOI":"10.1038\/nrm3392","article-title":"New lives for old: evolution of pseudoenzyme function illustrated by iRhoms","volume":"13","author":"Adrain","year":"2012","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2023070709252974200_JCS260910C2","doi-asserted-by":"publisher","first-page":"225","DOI":"10.1126\/science.1214400","article-title":"Tumor necrosis factor signaling requires iRhom2 to promote trafficking and activation of TACE","volume":"335","author":"Adrain","year":"2012","journal-title":"Science"},{"key":"2023070709252974200_JCS260910C3","doi-asserted-by":"publisher","first-page":"67","DOI":"10.1016\/j.molmet.2019.10.006","article-title":"Deletion of iRhom2 protects against diet-induced obesity by increasing thermogenesis","volume":"31","author":"Badenes","year":"2020","journal-title":"Mol. Metab."},{"key":"2023070709252974200_JCS260910C4","doi-asserted-by":"publisher","first-page":"e202201644","DOI":"10.26508\/lsa.202201644","article-title":"The ADAM17 sheddase complex regulator iTAP modulates inflammation, epithelial repair, and tumor growth","volume":"6","author":"Badenes","year":"2023","journal-title":"Life Sci. Alliance"},{"key":"2023070709252974200_JCS260910C5","doi-asserted-by":"publisher","first-page":"729","DOI":"10.1038\/385729a0","article-title":"A metalloprotease disintegrin that releases tumour-necrosis factor-a from cells","volume":"385","author":"Black","year":"1997","journal-title":"Nature"},{"key":"2023070709252974200_JCS260910C6","doi-asserted-by":"publisher","first-page":"340","DOI":"10.1016\/j.ajhg.2011.12.008","article-title":"RHBDF2 mutations are associated with tylosis, a familial esophageal cancer syndrome","volume":"90","author":"Blaydon","year":"2012","journal-title":"Am. J. Hum. Genet."},{"key":"2023070709252974200_JCS260910C7","doi-asserted-by":"publisher","first-page":"32","DOI":"10.1038\/nrm1548","article-title":"ADAMs: key components in EGFR signalling and development","volume":"6","author":"Blobel","year":"2005","journal-title":"Nat. Rev. Mol. Cell. Biol."},{"key":"2023070709252974200_JCS260910C8","doi-asserted-by":"publisher","first-page":"1114","DOI":"10.1093\/emboj\/cdg111","article-title":"TACE is required for the activation of the EGFR by TGF-alpha in tumors","volume":"22","author":"Borrell-Pag\u00e8s","year":"2003","journal-title":"EMBO J."},{"key":"2023070709252974200_JCS260910C9","doi-asserted-by":"publisher","first-page":"4064","DOI":"10.1093\/hmg\/ddu120","article-title":"iRHOM2-dependent regulation of ADAM17 in cutaneous disease and epidermal barrier function","volume":"23","author":"Brooke","year":"2014","journal-title":"Hum. Mol. Genet."},{"key":"2023070709252974200_JCS260910C10","doi-asserted-by":"publisher","first-page":"415","DOI":"10.1007\/s00335-021-09910-3","article-title":"Inactive rhomboid proteins RHBDF1 and RHBDF2 (iRhoms): a decade of research in murine models","volume":"32","author":"Burzenski","year":"2021","journal-title":"Mamm. Genome"},{"key":"2023070709252974200_JCS260910C11","doi-asserted-by":"publisher","first-page":"884","DOI":"10.1038\/embor.2013.128","article-title":"Mammalian iRhoms have distinct physiological functions including an essential role in TACE regulation","volume":"14","author":"Christova","year":"2013","journal-title":"EMBO Rep."},{"key":"2023070709252974200_JCS260910C12","doi-asserted-by":"publisher","first-page":"331","DOI":"10.1016\/S0006-291X(03)01381-0","article-title":"TACE\/ADAM-17 enzymatic activity is increased in response to cellular stimulation","volume":"308","author":"Doedens","year":"2003","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"2023070709252974200_JCS260910C13","doi-asserted-by":"publisher","first-page":"190003","DOI":"10.1098\/rsob.190003","article-title":"The molecular, cellular and pathophysiological roles of iRhom pseudoproteases","volume":"9","author":"Dulloo","year":"2019","journal-title":"Open Biol."},{"key":"2023070709252974200_JCS260910C14","doi-asserted-by":"publisher","first-page":"2022.11.28.518246","DOI":"10.1101\/2022.11.28.518246","article-title":"Cleavage of the pseudoprotease iRhom2 by the signal peptidase complex reveals an ER-to-nucleus signalling pathway","author":"Dulloo","year":"2022","journal-title":"bioRxiv"},{"key":"2023070709252974200_JCS260910C15","doi-asserted-by":"publisher","first-page":"3077","DOI":"10.1128\/MCB.00291-13","article-title":"ADAM17 controls endochondral ossification by regulating terminal differentiation of chondrocytes","volume":"33","author":"Hall","year":"2013","journal-title":"Mol. Cell. Biol."},{"key":"2023070709252974200_JCS260910C16","doi-asserted-by":"publisher","first-page":"1064","DOI":"10.1182\/blood-2017-12-820571","article-title":"Blood-induced bone loss in murine hemophilic arthropathy is prevented by blocking the iRhom2\/ADAM17\/TNF-\u03b1 pathway","volume":"132","author":"Haxaire","year":"2018","journal-title":"Blood"},{"key":"2023070709252974200_JCS260910C17","doi-asserted-by":"publisher","first-page":"2686","DOI":"10.4049\/jimmunol.179.5.2686","article-title":"Cutting edge: TNF-\u03b1-converting enzyme (TACE\/ADAM17) inactivation in mouse myeloid cells prevents lethality from endotoxin shock","volume":"179","author":"Horiuchi","year":"2007","journal-title":"J. Immunol."},{"key":"2023070709252974200_JCS260910C18","doi-asserted-by":"publisher","first-page":"E2200","DOI":"10.1073\/pnas.1323908111","article-title":"Rhbdf2 mutations increase its protein stability and drive EGFR hyperactivation through enhanced secretion of amphiregulin","volume":"111","author":"Hosur","year":"2014","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2023070709252974200_JCS260910C19","doi-asserted-by":"publisher","first-page":"1174","DOI":"10.1242\/bio.026260","article-title":"Genetic deletion of amphiregulin restores the normal skin phenotype in a mouse model of the human skin disease tylosis","volume":"6","author":"Hosur","year":"2017","journal-title":"Biol. Open"},{"key":"2023070709252974200_JCS260910C20","doi-asserted-by":"publisher","first-page":"702","DOI":"10.1002\/2211-5463.12407","article-title":"ADAM17 is essential for ectodomain shedding of the EGF-receptor ligand amphiregulin","volume":"8","author":"Hosur","year":"2018","journal-title":"FEBS Open Bio."},{"key":"2023070709252974200_JCS260910C21","doi-asserted-by":"publisher","first-page":"233","DOI":"10.3389\/fgene.2018.00233","article-title":"RHBDF2-regulated growth factor signaling in a rare human disease, tylosis with esophageal cancer: what can we learn from murine models?","volume":"9","author":"Hosur","year":"2018","journal-title":"Front. Genet."},{"key":"2023070709252974200_JCS260910C22","doi-asserted-by":"publisher","first-page":"168","DOI":"10.1186\/s13059-020-02086-0","article-title":"Genes adapt to outsmart gene-targeting strategies in mutant mouse strains by skipping exons to reinitiate transcription and translation","volume":"21","author":"Hosur","year":"2020","journal-title":"Genome Biol."},{"key":"2023070709252974200_JCS260910C23","doi-asserted-by":"publisher","first-page":"928","DOI":"10.1172\/JCI66168","article-title":"iRHOM2 is a critical pathogenic mediator of inflammatory arthritis","volume":"123","author":"Issuree","year":"2013","journal-title":"J. Clin. Invest."},{"key":"2023070709252974200_JCS260910C24","doi-asserted-by":"publisher","first-page":"2704","DOI":"10.1093\/emboj\/cdg264","article-title":"Defective valvulogenesis in HB-EGF and TACE-null mice is associated with aberrant BMP signaling","volume":"22","author":"Jackson","year":"2003","journal-title":"EMBO J."},{"key":"2023070709252974200_JCS260910C25","doi-asserted-by":"publisher","first-page":"6","DOI":"10.1016\/S0888-7543(02)00013-7","article-title":"Curly bare (cub), a new mouse mutation on chromosome 11 causing skin and hair abnormalities, and a modifier gene (mcub) on chromosome 5","volume":"81","author":"Johnson","year":"2003","journal-title":"Genomics"},{"key":"2023070709252974200_JCS260910C26","doi-asserted-by":"publisher","first-page":"135","DOI":"10.21203\/rs.3.rs-2526407\/v1","article-title":"A structural model of the iRhom-ADAM17 sheddase complex reveals functional insights into its trafficking and activity","volume":"80","author":"Kahveci-T\u00fcrk\u00f6z","year":"2022","journal-title":"Res. Square"},{"key":"2023070709252974200_JCS260910C27","doi-asserted-by":"publisher","first-page":"e35012","DOI":"10.7554\/eLife.35012","article-title":"FRMD8 promotes inflammatory and growth factor signalling by stabilising the iRhom\/ADAM17 sheddase complex","volume":"7","author":"Kunzel","year":"2018","journal-title":"eLife"},{"key":"2023070709252974200_JCS260910C28","doi-asserted-by":"publisher","first-page":"3913","DOI":"10.1242\/jcs.069997","article-title":"ADAM17 is regulated by a rapid and reversible mechanism that controls access to its catalytic site","volume":"123","author":"Le Gall","year":"2010","journal-title":"J. Cell Sci."},{"key":"2023070709252974200_JCS260910C29","doi-asserted-by":"publisher","first-page":"6080","DOI":"10.1073\/pnas.1505649112","article-title":"iRhoms 1 and 2 are essential upstream regulators of ADAM17-dependent EGFR signaling","volume":"112","author":"Li","year":"2015","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2023070709252974200_JCS260910C30","doi-asserted-by":"publisher","first-page":"868","DOI":"10.1242\/jcs.196436","article-title":"Structural modeling defines transmembrane residues in ADAM17 that are crucial for Rhbdf2\/ADAM17-dependent proteolysis","volume":"130","author":"Li","year":"2017","journal-title":"J. Cell Sci."},{"key":"2023070709252974200_JCS260910C31","doi-asserted-by":"publisher","first-page":"2022.10.22.513345","DOI":"10.1101\/2022.10.22.513345","article-title":"Structural basis for selective proteolysis of ADAM10 substrates at membrane- proximal sites","author":"Lipper","year":"2022","journal-title":"bioRxiv"},{"key":"2023070709252974200_JCS260910C32","doi-asserted-by":"publisher","first-page":"100733","DOI":"10.1016\/j.jbc.2021.100733","article-title":"Targeted truncation of the ADAM17 cytoplasmic domain in mice results in protein destabilization and a hypomorphic phenotype","volume":"296","author":"Lora","year":"2021","journal-title":"J. Biol. Chem."},{"key":"2023070709252974200_JCS260910C33","doi-asserted-by":"publisher","first-page":"26236","DOI":"10.1074\/jbc.273.40.26236","article-title":"Intracellular maturation of the mouse metalloprotease disintegrin MDC15","volume":"273","author":"Lum","year":"1998","journal-title":"J. Biol. Chem."},{"key":"2023070709252974200_JCS260910C34","doi-asserted-by":"publisher","first-page":"ra109","DOI":"10.1126\/scisignal.aac5356","article-title":"Deletions in the cytoplasmic domain of iRhom1 and iRhom2 promote shedding of the TNF receptor by the protease ADAM17","volume":"8","author":"Maney","year":"2015","journal-title":"Sci. Signal."},{"key":"2023070709252974200_JCS260910C35","doi-asserted-by":"publisher","first-page":"11433","DOI":"10.1073\/pnas.1302553110","article-title":"iRhom2 controls the substrate selectivity of stimulated ADAM17-dependent ectodomain shedding","volume":"110","author":"Maretzky","year":"2013","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2023070709252974200_JCS260910C36","doi-asserted-by":"publisher","first-page":"229","DOI":"10.1126\/science.1214448","article-title":"iRhom2 regulation of TACE controls TNF-mediated protection against Listeria and responses to LPS","volume":"335","author":"McIlwain","year":"2012","journal-title":"Science"},{"key":"2023070709252974200_JCS260910C37","doi-asserted-by":"publisher","first-page":"4317","DOI":"10.1242\/dev.02030","article-title":"HB-EGF promotes epithelial cell migration in eyelid development","volume":"132","author":"Mine","year":"2005","journal-title":"Development"},{"key":"2023070709252974200_JCS260910C38","doi-asserted-by":"publisher","first-page":"1114","DOI":"10.1111\/cge.13161","article-title":"Tylosis associated with squamous cell carcinoma of the oesophagus (TOC): report of an African family with a novel RHBDF2 variant","volume":"93","author":"Mokoena","year":"2018","journal-title":"Clin. Genet."},{"key":"2023070709252974200_JCS260910C39","doi-asserted-by":"publisher","first-page":"733","DOI":"10.1038\/385733a0","article-title":"Cloning of a disintegrin metalloproteinase that processes precursor tumour-necrosis factor-\u03b1","volume":"385","author":"Moss","year":"1997","journal-title":"Nature"},{"key":"2023070709252974200_JCS260910C40","doi-asserted-by":"publisher","first-page":"e35032","DOI":"10.7554\/eLife.35032","article-title":"itap, a novel iRhom interactor, controls TNF secretion by policing the stability of iRhom\/TACE","volume":"7","author":"Oikonomidi","year":"2018","journal-title":"eLife"},{"key":"2023070709252974200_JCS260910C41","doi-asserted-by":"publisher","first-page":"1281","DOI":"10.1126\/science.282.5392.1281","article-title":"An essential role for ectodomain shedding in mammalian development","volume":"282","author":"Peschon","year":"1998","journal-title":"Science"},{"key":"2023070709252974200_JCS260910C42","doi-asserted-by":"publisher","first-page":"2737","DOI":"10.1002\/eji.201646482","article-title":"iRhom2 regulates CSF1R cell surface expression and non-steady state myelopoiesis in mice","volume":"46","author":"Qing","year":"2016","journal-title":"Eur. J. Immunol."},{"key":"2023070709252974200_JCS260910C43","doi-asserted-by":"publisher","first-page":"1397","DOI":"10.1172\/JCI97650","article-title":"iRhom2 promotes lupus nephritis through TNF-\u03b1 and EGFR signaling","volume":"128","author":"Qing","year":"2018","journal-title":"J. Clin. Invest."},{"key":"2023070709252974200_JCS260910C44","doi-asserted-by":"publisher","first-page":"699","DOI":"10.2340\/00015555-3189","article-title":"Whole exome sequencing identified a novel mutation of the RHBDF2 gene in a chinese family of tylosis with esophageal cancer","volume":"99","author":"Qu","year":"2019","journal-title":"Acta Derm. Venereol."},{"key":"2023070709252974200_JCS260910C45","doi-asserted-by":"publisher","first-page":"769","DOI":"10.1083\/jcb.200307137","article-title":"Distinct roles for ADAM10 and ADAM17 in ectodomain shedding of six EGFR-ligands","volume":"164","author":"Sahin","year":"2004","journal-title":"J. Cell Biol."},{"key":"2023070709252974200_JCS260910C46","doi-asserted-by":"publisher","first-page":"99","DOI":"10.1385\/1-59745-012-X:99","article-title":"A sensitive method to monitor ectodomain shedding of ligands of the epidermal growth factor receptor","volume":"327","author":"Sahin","year":"2006","journal-title":"Methods Mol. Biol."},{"key":"2023070709252974200_JCS260910C47","doi-asserted-by":"publisher","first-page":"e54853","DOI":"10.1371\/journal.pone.0054853","article-title":"Conditional inactivation of TNF\u03b1-converting enzyme in chondrocytes results in an elongated growth plate and shorter long bones","volume":"8","author":"Saito","year":"2013","journal-title":"PLoS ONE"},{"key":"2023070709252974200_JCS260910C48","doi-asserted-by":"publisher","first-page":"1826","DOI":"10.1074\/jbc.M408804200","article-title":"ADAM10 mediates ectodomain shedding of the betacellulin precursor activated by p-aminophenylmercuric acetate and extracellular calcium influx","volume":"280","author":"Sanderson","year":"2005","journal-title":"J. Biol. Chem."},{"key":"2023070709252974200_JCS260910C49","doi-asserted-by":"publisher","first-page":"131","DOI":"10.1042\/bj3470131","article-title":"Intracellular maturation and localization of the tumour necrosis factor \u03b1 convertase (TACE)","volume":"347","author":"Schl\u00f6ndorff","year":"2000","journal-title":"Biochem. J."},{"key":"2023070709252974200_JCS260910C50","doi-asserted-by":"publisher","first-page":"jcs259949","DOI":"10.1242\/jcs.259949","article-title":"iRhom2 regulates ERBB signalling to promote KRAS-driven tumour growth of lung cancer cells","volume":"135","author":"Sieber","year":"2022","journal-title":"J. Cell Sci."},{"key":"2023070709252974200_JCS260910C51","doi-asserted-by":"publisher","first-page":"5769","DOI":"10.1182\/blood-2012-03-417949","article-title":"iRhom2 is required for the secretion of mouse TNF\u03b1","volume":"119","author":"Siggs","year":"2012","journal-title":"Blood"},{"key":"2023070709252974200_JCS260910C52","doi-asserted-by":"publisher","first-page":"1151","DOI":"10.1242\/bio.201410116","article-title":"Genetic interaction implicates iRhom2 in the regulation of EGF receptor signalling in mice","volume":"3","author":"Siggs","year":"2014","journal-title":"Biol. Open"},{"key":"2023070709252974200_JCS260910C53","doi-asserted-by":"publisher","first-page":"154194","DOI":"10.1016\/j.metabol.2020.154194","article-title":"Loss of iRhom2 accelerates fat gain and insulin resistance in diet-induced obesity despite reduced adipose tissue inflammation","volume":"106","author":"Skurski","year":"2020","journal-title":"Metabolism"},{"key":"2023070709252974200_JCS260910C54","doi-asserted-by":"publisher","first-page":"3923","DOI":"10.1242\/dev.01966","article-title":"Mammary ductal morphogenesis requires paracrine activation of stromal EGFR via ADAM17-dependent shedding of epithelial amphiregulin","volume":"132","author":"Sternlicht","year":"2005","journal-title":"Development"},{"key":"2023070709252974200_JCS260910C55","doi-asserted-by":"publisher","first-page":"12838","DOI":"10.1074\/jbc.M112050200","article-title":"Tumor necrosis factor-\u03b1 converting enzyme (TACE) regulates epidermal growth factor receptor ligand availability","volume":"277","author":"Sunnarborg","year":"2002","journal-title":"J. Biol. Chem."},{"key":"2023070709252974200_JCS260910C56","doi-asserted-by":"publisher","first-page":"4813","DOI":"10.1096\/fj.201902649R","article-title":"Substrate-selective protein ectodomain shedding by ADAM17 and iRhom2 depends on their juxtamembrane and transmembrane domains","volume":"34","author":"Tang","year":"2020","journal-title":"FASEB J."},{"key":"2023070709252974200_JCS260910C57","doi-asserted-by":"publisher","first-page":"131","DOI":"10.1016\/j.bone.2012.04.012","article-title":"Transforming growth factor alpha controls the transition from hypertrophic cartilage to bone during endochondral bone growth","volume":"51","author":"Usmani","year":"2012","journal-title":"Bone"},{"key":"2023070709252974200_JCS260910C58","doi-asserted-by":"publisher","first-page":"4350","DOI":"10.1074\/jbc.RA119.011136","article-title":"ADAM17 stabilizes its interacting partner inactive Rhomboid 2 (iRhom2) but not inactive Rhomboid 1 (iRhom1)","volume":"295","author":"Weskamp","year":"2020","journal-title":"J. Biol. Chem."},{"key":"2023070709252974200_JCS260910C59","doi-asserted-by":"publisher","first-page":"1317","DOI":"10.1083\/jcb.143.5.1317","article-title":"Removal of the membrane-anchoring domain of epidermal growth factor leads to intracrine signaling and disruption of mammary epithelial cell organization","volume":"143","author":"Wiley","year":"1998","journal-title":"J. Cell Biol."},{"key":"2023070709252974200_JCS260910C60","doi-asserted-by":"publisher","first-page":"469","DOI":"10.1083\/jcb.200307035","article-title":"Mice with defects in HB-EGF ectodomain shedding show severe developmental abnormalities","volume":"163","author":"Yamazaki","year":"2003","journal-title":"J. Cell Biol."},{"key":"2023070709252974200_JCS260910C61","doi-asserted-by":"publisher","first-page":"996","DOI":"10.1126\/science.abo5672","article-title":"The human signal peptidase complex acts as a quality control enzyme for membrane proteins","volume":"378","author":"Zanotti","year":"2022","journal-title":"Science"},{"key":"2023070709252974200_JCS260910C62","doi-asserted-by":"publisher","first-page":"12796","DOI":"10.3390\/ijms232112796","article-title":"Identification of molecular determinants in iRhoms1 and 2 that contribute to the substrate selectivity of stimulated ADAM17","volume":"23","author":"Zhao","year":"2022","journal-title":"Int. J. Mol. Sci."}],"container-title":["Journal of Cell Science"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.biologists.com\/jcs\/article-pdf\/doi\/10.1242\/jcs.260910\/2947351\/jcs260910.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/jcs\/article-pdf\/doi\/10.1242\/jcs.260910\/3030951\/jcs260910.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/jcs\/article-pdf\/doi\/10.1242\/jcs.260910\/3030951\/jcs260910.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,7,7]],"date-time":"2023-07-07T09:25:42Z","timestamp":1688721942000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/jcs\/article\/136\/13\/jcs260910\/323168\/Analysis-of-the-function-of-ADAM17-in-iRhom2-curly"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,7,1]]},"references-count":62,"journal-issue":{"issue":"13","published-print":{"date-parts":[[2023,7,1]]}},"URL":"https:\/\/doi.org\/10.1242\/jcs.260910","relation":{},"ISSN":["0021-9533","1477-9137"],"issn-type":[{"value":"0021-9533","type":"print"},{"value":"1477-9137","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,7,1]]},"article-number":"jcs260910"}}