The overlap syndrome between primary biliary cirrhosis and primary sclerosing cholangitis

Digestive and Liver Disease : Official Journal of the Italian Society of Gastroenterology and the Italian Association for the Study of the Liver
Annarosa FloreaniPietro Invernizzi

Abstract

The overlap syndrome between primary biliary cirrhosis and primary sclerosing cholangitis is an extremely rare condition that has been reported in only six published cases so far. Here we report two cases showing the clinical manifestations of both primary biliary cirrhosis and primary sclerosing cholangitis. In one case the overlap condition was associated with psoriatric arthritis, and the patient successfully underwent dual treatment with ursodeoxycholic acid and the anti-tumour necrosis factor-alpha agent adalimumab. In the second case, the predominant condition was, initially, an antimitochondrial antibody-negative primary biliary cirrhosis with progressive course towards end-stage liver disease; the patient then developed either antimitochondrial antibody positivity or changes in the biliary tree compatible with primary sclerosing cholangitis. These two cases add information on a controversial issue in the literature, and indicate the importance of recognizing a possible overlap syndrome to optimize treatment.

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Citations

Mar 22, 2016·Cellular & Molecular Immunology·Zongwen ShuaiM Eric Gershwin
Oct 11, 2017·Clinical Reviews in Allergy & Immunology·Benedetta Terziroli Beretta-PiccoliCarlo Mainetti
Jul 25, 2018·ACG Case Reports Journal·Sridhar SundaramAkash Shukla
Jun 6, 2019·Archivum Immunologiae Et Therapiae Experimentalis·George N Dalekos, Nikolaos K Gatselis
May 18, 2020·Journal of Clinical Medicine·Benjamin L FreedmanAlan Bonder
Mar 17, 2018·BioMed Research International·Marco FioreGiovanni Damiani
Dec 14, 2016·World Journal of Hepatology·Ying-Qiu Huang
Oct 22, 2020·Journal of Clinical and Translational Hepatology·Sheena Mago, George Y Wu

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