Introduction: Ulcerative colitis is a chronic intestinal inflammation, part of inflammatory bowel disease, which also includes Crohn’s disease. Both have extraintestinal manifestations, but those that tend to occur more commonly with ulcerative colitis include chronic active hepatitis, pyoderma gangrenosum and ankylosing spondylitis. Many individuals present with overlapping non-diagnostic features of more than one of these conditions that is referred to in the literature as autoimmune overlap syndrome. Sclerosing cholangitis associated with IBD is often referred to as overlap syndrome.
Material and methods: We present the case of a 15-year-old female, with an association between ulcerative colitis, primitive sclerosing cholalangitis and autoimmune hepatitis. She was admitted for: diarrheic bloody stools, abdominal pain, diminished appetite, headache and aphthous stomatitis. Blood sample analysis revealed: hypochromic anemia, iron deficiency, high levels of transaminase, abnormal protein electrophoresis, positive anti-neutrophil cytoplasmic antibodies and anti–smooth muscle antibodies, high level of faecal calprotectin, modified biliary tract on imaging of digestive system and suggestive modifications of colic mucosa for ulcerative colitis. We administered treatment with Arginine Chloride 5%, Sorbitol 10%, Aspartic acid, Vitamin B6, Ursodeoxycholic acid, 5-aminosalicylic acid.
Results: With the administered therapy the evolution was good, macroscopic blood disappeared from stools, and tests for blood trace in stool were also negative.
Conclusions: The patient had simultaneous onset of diarrhea with bloody stools and extraintestinal manifestations. Immunological markers didn’t fully match any of the associated diseases, so we concluded that there was an overlap syndrome. Budesonide was effective on both hepatic and intestinal disease.
Ulcerative Colitis associated with Sclerosing Cholangitis and Autoimmune Hepatitis
DOI: 10.2478/amma-2013-0028
Keywords: budesonide, 5-aminosalicylic acid, ursodeoxycholic acid, ulcerative colitis, overlap syndrome, calprotectin
Full text: PDF