{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,30]],"date-time":"2026-06-30T06:08:54Z","timestamp":1782799734161,"version":"3.54.5"},"reference-count":68,"publisher":"Oxford University Press (OUP)","issue":"6","license":[{"start":{"date-parts":[[2026,3,4]],"date-time":"2026-03-04T00:00:00Z","timestamp":1772582400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,5,19]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>CYLD cutaneous syndrome (CCS) skin tumours develop from puberty onwards, can number in the hundreds and progressively grow over time. Patients with CCS lack medical therapies and require repeated surgery to control tumour burden. CYLD loss of heterozygosity drives tumour growth, and CCS tumours have previously been shown to demonstrate increased canonical nuclear factor kappa B (NF-\u03baB) and Wnt signalling.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>To investigate NF-\u03baB signalling in CCS tumours and CCS tumour keratinocytes, with the aim of identifying druggable targets.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We used complementary bulk transcriptomics and proteomics in patient-derived CCS tumour cell fractions, as well as single-cell RNA sequencing of CCS tumour cells to investigate aberrant NF-\u03baB signalling. We developed a patient-derived CCS tumour spheroid culture model to determine the utility of targeting aberrant NF-\u03baB cell signalling.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>We demonstrate evidence of non-canonical NF-\u03baB signalling in CCS tumour keratinocytes, with increased p100 to p52 processing and RelB protein expression compared with normal skin. We identify I\u03baB kinase alpha (IKK\u03b1) as a candidate target in the noncanonical NF-\u03baB signalling pathway. A novel, highly selective IKK\u03b1 inhibitor (SU1644) used in patient-derived CCS tumour spheroid cultures demonstrated that IKK\u03b1 inhibition reduced tumour spheroid viability.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>These data provide the preclinical rationale for the assessment of topical IKK\u03b1 inhibitors as a novel preventive treatment for CCS.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1093\/bjd\/ljag044","type":"journal-article","created":{"date-parts":[[2026,2,6]],"date-time":"2026-02-06T12:49:38Z","timestamp":1770382178000},"page":"1130-1141","source":"Crossref","is-referenced-by-count":3,"title":["Targeting noncanonical nuclear factor kappa B signalling in\n                    <i>CYLD<\/i>\n                    cutaneous syndrome by selective inhibition of I\u03baB kinase alpha"],"prefix":"10.1093","volume":"194","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4348-0430","authenticated-orcid":false,"given":"Kirsty","family":"Hodgson","sequence":"first","affiliation":[{"name":"Translational and Clinical Research Institute, Newcastle University , Newcastle upon Tyne ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6761-4179","authenticated-orcid":false,"given":"Joseph","family":"Inns","sequence":"additional","affiliation":[{"name":"Translational and Clinical Research Institute, Newcastle University , Newcastle upon Tyne ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8142-8708","authenticated-orcid":false,"given":"Gary","family":"Reynolds","sequence":"additional","affiliation":[{"name":"Biosciences Institute, Newcastle University , Newcastle upon Tyne ,","place":["UK"]},{"name":"Centre for Immunology and Inflammatory 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