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Ulcerative colitis (UC) with concomitant primary sclerosing cholangitis (PSC) represents a distinct disease entity (PSC-UC). Our results highlight the limitations of endoscopic examination in uncovering microscopic inflammatory lesions, resulting in the erroneous classification of PSC-UC patients as healthy. To mitigate the risk of underdiagnosis, histopathological examination should therefore be an essential component of the diagnostic approach for UC in PSC patients.