Case Report
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World J Gastroenterol. Jan 28, 2013; 19(4): 590-593
Published online Jan 28, 2013. doi: 10.3748/wjg.v19.i4.590
Steroid-refractory ulcerative colitis and associated primary sclerosing cholangitis treated with infliximab
Ileana Duca, Patricia Ramírez de la Piscina, Silvia Estrada, Rosario Calderón, Katerina Spicakova, Leire Urtasun, Carlos Marra-López, Salvador Zabaleta, Raquel Bengoa, María Asunción Marcaide, Francisco García-Campos
Ileana Duca, Patricia Ramírez de la Piscina, Silvia Estrada, Rosario Calderón, Katerina Spicakova, Leire Urtasun, Carlos Marra-López, Salvador Zabaleta, Raquel Bengoa, María Asunción Marcaide, Francisco García-Campos, Service of Gastroenterology, Hospital Universitario de Álava-Sede Txagorritxu, Universidad del País Vasco, E-01009 Vitoria-Gasteiz, Spain
Author contributions: Duca I and Ramírez de la Piscina P reviewed the literature, design the study, collected information, provided care to the patient, and wrote the manuscript; Estrada S, Calderón R, Spicakova K, Urtasun L, Zabaleta S, Marra-López C, Bengoa R, Marcaide MA and García-Campos F were involved in the different stages of the diagnosis, care, and management of the patient, provided constructive criticisms, revised and approved the final draft; García-Campos F also supervised the entire process from drafting the paper to the final manuscript submitted to the journal.
Correspondence to: Patrícia Ramírez de la Piscina, MD, Service of Gastroenterology, Hospital Universitario de Álava-Sede Txagorritxu, Universidad del País Vasco, C/ José Achótegui s/n, E-01009 Vitoria-Gasteiz, Spain. patricia.ramirezdelapiscinaurraca@osakidetza.net
Telephone: +34-945-007301 Fax: +34-945-007359
Received: October 18, 2012
Revised: December 9, 2012
Accepted: December 15, 2012
Published online: January 28, 2013
Abstract

Primary sclerosing cholangitis is an infrequent extraintestinal manifestation of ulcerative colitis. Damage to bile ducts is irreversible and medical therapies to prevent progression of the disease are usually ineffective. We describe a patient with long-standing ulcerative colitis, which was refractory to corticosteroid therapy who developed primary sclerosing cholangitis (biochemical stage II/IV) in the course of his pancolitis. Treatment with infliximab (5 mg/kg as an induction dose followed by maintenance doses every two months) was indicated because of steroid-dependent disease associated to primary sclerosing cholangitis as well as sacroiliitis and uveitis and previous episode of severe azathioprine-related hepatic toxicity. At present, after two years of follow-up, the patient is asymptomatic with normal liver tests and complete resumption of daily life activities. This case draws attention to the usefulness of anti-tumor necrosis factor-alpha therapy for the management of primary sclerosing cholangitis as extraintestinal manifestation of inflammatory bowel disease.

Keywords: Ulcerative colitis, Infliximab, Monoclonal antibodies, Sclerosing cholangitis, Bile duct diseases, Tumor necrosis factor-alpha