Objectives: Data regarding pediatric PSC natural history are limited. We describe a large pediatric PSC cohort with longitudinal follow-up. Methods: Records review of pediatric PSC patients diagnosed between 1984 and 2014. Results: N = 120 (63% M) aged 1-21y (median 14y) at diagnosis. 27% (31/113) had ASC (autoimmune sclerosing cholangitis), 24% had exclusive small duct PSC, METAVIR stage was F3-F4 in 41%. 81% of PSC patients had IBD; most had ulcerative/indeterminate colitis (72/97), typically pancolitis (40/72). PSC-IBD was more common than ASC-IBD (85% vs. 68%, P = 0.03). Median follow-up was 3.7y (IQR 1.5, 6.9). Median GGT decreased from baseline of 221 U/L (IQR 110, 425) to 104 U/L by 1y post-diagnosis ([IQR 18,229], P
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