Primary sclerosing cholangitis (PSC) is a liver disease of idiopathic origin, displaying a diverse and varied nature, which leads to cholestasis. It is characterized by continuous, advancing inflammation and fibrosis in the bile ducts. PSC is closely linked with inflammatory bowel disease and poses a risk for colon, bile duct, and gallbladder cancer. Unfortunately, there is currently no effective medical treatment available for this condition. In some cases, the disease may progress to end-stage liver failure, making liver transplantation a possible necessity for affected individuals. PSC association with autoimmune hepatitis (AIH) is very rare. This is a case of PSC that is overlapped with AIH. Screening colonoscopy showed colitis, and a biopsy was consistent with ulcerative colitis without any colitis symptoms, emphasizing the need for ruling out any other associated conditions, which respond well to the effective treatment to avoid morbidity and mortality in PSC.
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