Pancolitis was numerically more common in the PSC-IBD group, but the difference was not statistically significant. Backwash ileitis and relative rectal sparing were present in a significantly greater proportion of PSC-IBD patients. Patient and disease characteristics at study entry are shown in Table 1. The international normalized ratio was increased in 2 patients. Median laboratory values at the time of colonoscopy included albumin level of 43 g/L (range, 28–49 g/L), direct bilirubin level of 6.5 μmol/L (range, 0–45 μmol/L), alanine aminotransferase level of 48 U/L (range, 16–454 U/L), aspartate aminotransferase level of 44 U/L (range, 19–510 U/L), alkaline phosphatase level of 0.8 times upper limit of normal (ULN) (range, 0.3–4.2 times ULN), and γ-glutamyltransferase 0.8 times ULN (range, 0.2–9.2 times ULN). Three had previously undergone a liver transplant (of whom 1 developed recurrent PSC before study inclusion), 1 was listed for transplant, and 6 had portal hypertension. Eleven (30%) PSC patients had autoimmune hepatitis overlap and 5 (14%) had small-duct PSC (abnormal liver biopsy with normal cholangiogram). Spearman correlations between endoscopic scores and both PUCAI and FC were determined and compared between PSC-IBD and UC/IBD-U patients.Įighty-seven children were included, 37 with PSC-IBD and 50 with UC/IBD-U without liver disease. Scatterplots and boxplots were used to illustrate the relationship between extent UCEIS and PUCAI and FC in both study groups. Results were expressed as odds ratios (ORs) with 95% CIs. Logistic regression was repeated to determine the odds of endoscopic disease in PSC-IBD patients in biochemical remission (FC, <100 μg/g) relative to UC/IBD-U patients in biochemical remission. In addition, endoscopic scores were compared between PSC-IBD and UC/IBD-U patients in clinical remission using the Mann–Whitney U test. To test the hypothesis that symptoms underestimate endoscopic activity in PSC-IBD, the odds of active endoscopic disease were determined in PSC-IBD patients in clinical remission (PUCAI < 10), relative to UC/IBD-U patients in clinical remission, using univariate and multivariable logistic regression, adjusting for sex, age at IBD diagnosis, and IBD duration, with variables selected a priori based on clinical relevance. At our institution, biopsy specimens are taken routinely from each colonic segment during colonoscopy and pathologists score histologic IBD activity (based on acute inflammation) according to a systematic, 3-tiered grading systemīaseline characteristics were summarized using medians with interquartile ranges (IQRs) and the Mann–Whitney U test for continuous variables, and using frequencies with proportions and the chi-square test (or the Fisher exact test as appropriate) for categoric variables. The colonic segment with the highest UCEIS score was considered the worst endoscopic lesion. MH was defined as an extent UCEIS score of 0 (ie, normal macroscopic findings in all colonic segments) and active endoscopic disease was defined as a UCEIS score of 1 or higher. A global rating of endoscopic lesion severity, rated as none, mild, moderate, or severe based on the overall impression of the entire colon, also was assigned. The extent UCEIS was derived by summing the usual UCEIS score applied to the 4 colonic segments (rectum, descending colon/sigmoid, transverse colon, and ascending colon) and dividing by 4 to maintain the tool’s original range. In addition, given the predilection of PSC-IBD for extensive, often right-sided, colonic involvement, a modified version of the UCEIS, termed extent UCEIS, which incorporated disease extent, was used as well. Both are conventionally applied to the worst affected region of the rectosigmoid colon. The Mayo Endoscopic Subscore ranges from 0 to 3 and the UCEIS ranges from 0 to 8. Analogous analyses were performed for histologic activity. The ability of fecal calprotectin to identify mucosal healing in patients with PSC-IBD was assessed using receiver operating characteristic curve analyses. Correlations between activity markers were compared between groups. Among patients in clinical remission, endoscopic scores and the odds of active endoscopic disease (based on a UCEIS score ≥1) were compared between patients with and without PSC in univariate and multivariable analyses. Colonoscopies were scored using the Mayo endoscopic subscore and the UC Endoscopic Index of Severity (UCEIS) scores, and histologic activity was graded. We collected pediatric ulcerative colitis activity index (PUCAI) scores (to measure symptoms) and fecal levels of calprotectin from 37 children with PSC-IBD and 50 children with only IBD (controls UC or IBD-unclassified). We performed a prospective study of children (age, <18 y) with colonic IBD with and without PSC who underwent colonoscopy from February 1, 2016, through March 31, 2017, at the Hospital for Sick Children in Toronto, Canada.
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