{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,7]],"date-time":"2026-11-07T13:34:47Z","timestamp":1794058487164,"version":"4.8.0"},"reference-count":49,"publisher":"Oxford University Press (OUP)","issue":"12","license":[{"start":{"date-parts":[[2022,6,30]],"date-time":"2022-06-30T00:00:00Z","timestamp":1656547200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"name":"UCSF Department of Pathology"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,12,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background and Aims<\/jats:title><jats:p>Patients with primary sclerosing cholangitis and inflammatory bowel disease [termed PSC-IBD] have a higher risk of developing colorectal neoplasia than those with IBD alone. The mechanism by which concomitant PSC increases the risk of colorectal neoplasia remains unknown. Seven distinct non-conventional dysplastic subtypes have been recently described in IBD, including crypt cell dysplasia, hypermucinous dysplasia, goblet cell-deficient dysplasia, dysplasia with increased Paneth cell differentiation [DPD], sessile serrated lesion [SSL]-like dysplasia, traditional serrated adenoma [TSA]-like dysplasia, and serrated dysplasia, not otherwise specified [NOS]. Despite the lack of high-grade morphological features, crypt cell, hypermucinous, and goblet cell-deficient dysplasias often show molecular features characteristic of advanced neoplasia [i.e. aneuploidy and KRAS mutations] and are more frequently associated with advanced neoplasia than conventional dysplasia on follow-up. We aimed to characterise clinicopathological features of dysplasia found in PSC-IBD patients.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A cohort of 173 PSC-IBD patients were analysed. All dysplastic lesions were subtyped as either conventional or non-conventional dysplasia. The clinicopathological features of PSC-IBD patients with neoplasia were also compared with those of non-PSC IBD patients with neoplasia.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>There were 109 [63%] men and 64 [37%] women, with a mean age of 26 years at IBD diagnosis and a long history of IBD [mean duration: 14 years]. Ulcerative colitis was the most common IBD subtype [80%], and the majority of patients [92%] had a history of pancolitis. A total of 153 dysplastic lesions were detected in 54 [31%] patients, 35 [65%] of whom had multifocal dysplasia. One additional patient presented with colorectal cancer [CRC] without a history of dysplasia. Dysplasia was often non-conventional [n = 93; 61%], endoscopically\/grossly invisible [n = 101; 66%], and right\/proximal-sided [n = 90; 59%]. All seven non-conventional subtypes were identified, including 46 [30%] crypt cell dysplasia, 23 [15%] hypermucinous dysplasia, 12 [8%] goblet cell-deficient dysplasia, seven [5%] DPD, three [2%] TSA-like dysplasia, one [1%] SSL-like dysplasia, and one [1%] serrated dysplasia NOS. Follow-up information was available for 86 lesions, of which 32 [37%] were associated with subsequent detection of advanced neoplasia [high-grade dysplasia or CRC] within a mean follow-up time of 55 months. PSC-IBD patients with neoplasia were more likely to have pancolitis [98%, p = 0.039] and a longer IBD duration [mean: 17 years, p = 0.021] than those without neoplasia [89% and 12 years, respectively]. When compared with a cohort of non-PSC IBD patients with neoplasia, the PSC-IBD group with neoplasia was more often associated with non-conventional [61%, p &amp;lt;0.001], invisible [66%, p &amp;lt;0.001], and right\/proximal-sided [59%, p = 0.045] dysplasias [vs 25%, 21%, and 47%, respectively, for the non-PSC IBD group]. The rate of advanced neoplasia was nearly 2-fold higher in the PSC-IBD group [37%] compared with the non-PSC IBD group [22%] [p = 0.035].<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Nearly a third of PSC-IBD patients developed dysplasia, which is often associated with non-conventional dysplastic features, invisible endoscopic\/gross appearance, right\/proximal-sided colon, multifocality, and advanced neoplasia on follow-up. These findings underscore the importance of recognising these non-conventional subtypes by practising pathologists and the need for careful and frequent endoscopic surveillance, with random biopsies, in PSC-IBD patients.<\/jats:p><\/jats:sec>","DOI":"10.1093\/ecco-jcc\/jjac090","type":"journal-article","created":{"date-parts":[[2022,6,30]],"date-time":"2022-06-30T18:06:02Z","timestamp":1656612362000},"page":"1825-1834","source":"Crossref","is-referenced-by-count":49,"title":["Increased Risk of Non-conventional and Invisible Dysplasias in Patients with Primary Sclerosing Cholangitis and Inflammatory Bowel Disease"],"prefix":"10.1093","volume":"16","author":[{"given":"Ruth","family":"Zhang","sequence":"first","affiliation":[{"name":"University of California at San Francisco, Department of Pathology , San Francisco, CA , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gregory Y","family":"Lauwers","sequence":"additional","affiliation":[{"name":"H. Lee Moffitt Cancer Center and Research Institute, Department of Pathology , Tampa, FL , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6892-5881","authenticated-orcid":false,"given":"Won-Tak","family":"Choi","sequence":"additional","affiliation":[{"name":"University of California at San Francisco, Department of Pathology , San Francisco, CA , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2022,6,30]]},"reference":[{"key":"2022120518042377500_CIT0001","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1136\/gut.2004.046615","article-title":"PSC-IBD: a unique form of inflammatory bowel disease associated with primary sclerosing cholangitis","volume":"54","author":"Loftus","year":"2005","journal-title":"Gut"},{"key":"2022120518042377500_CIT0002","doi-asserted-by":"crossref","first-page":"774","DOI":"10.1093\/ibd\/izz209","article-title":"Unique phenotypic characteristics and clinical course in patients with ulcerative colitis and primary sclerosing cholangitis: a multicenter US experience","volume":"26","author":"Wang","year":"2020","journal-title":"Inflamm Bowel Dis"},{"key":"2022120518042377500_CIT0003","doi-asserted-by":"crossref","first-page":"1494","DOI":"10.1097\/DCR.0000000000001496","article-title":"Distinct disease phenotype of ulcerative colitis in patients with coincident primary sclerosing cholangitis: evidence from a large retrospective study with matched cohorts","volume":"62","author":"Cordes","year":"2019","journal-title":"Dis Colon Rectum"},{"key":"2022120518042377500_CIT0004","doi-asserted-by":"crossref","first-page":"1956","DOI":"10.3748\/wjg.v21.i6.1956","article-title":"Distinctive inflammatory bowel disease phenotype in primary sclerosing cholangitis","volume":"21","author":"de Vries","year":"2015","journal-title":"World J Gastroenterol"},{"key":"2022120518042377500_CIT0005","doi-asserted-by":"crossref","first-page":"1004","DOI":"10.1097\/MIB.0b013e3182802893","article-title":"Inflammatory bowel disease in primary sclerosing cholangitis: a robust yet changing relationship","volume":"19","author":"Sinakos","year":"2013","journal-title":"Inflamm Bowel Dis"},{"key":"2022120518042377500_CIT0006","doi-asserted-by":"crossref","first-page":"2270","DOI":"10.1002\/ibd.22938","article-title":"Primary sclerosing cholangitis is associated with a distinct phenotype of inflammatory bowel disease","volume":"18","author":"Boonstra","year":"2012","journal-title":"Inflamm Bowel Dis"},{"key":"2022120518042377500_CIT0007","doi-asserted-by":"crossref","first-page":"1975","DOI":"10.1053\/j.gastro.2017.02.038","article-title":"International PSC Study Group. 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