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16 September 2022 : Case report  Italy

[In Press] A 60-Year-Old Woman with Primary Biliary Cholangitis and Crohn’s Ileitis Following the Suspension of Ursodeoxycholic Acid

Unusual clinical course, Challenging differential diagnosis, Unusual or unexpected effect of treatment, Diagnostic / therapeutic accidents, Adverse events of drug therapy , Educational Purpose (only if useful for a systematic review or synthesis)

Mario Romeo
  • A Study design/planning
  • B Data collection/entry
  • D Data interpretation
  • E Preparation of manuscript
  • F Literature analysis/search
" data-author="1068663">1ABDEF
, Marina Cipullo
  • A Study design/planning
  • B Data collection/entry
  • E Preparation of manuscript
  • F Literature analysis/search
" data-author="1068664">1ABEF
, Giorgia Iadanza
  • A Study design/planning
  • E Preparation of manuscript
" data-author="1068665">1AE
, Simone Olivieri
  • B Data collection/entry
  • E Preparation of manuscript
  • F Literature analysis/search
" data-author="1068666">1BEF
, Antonietta Gerarda Gravina
  • B Data collection/entry
  • D Data interpretation
" data-author="1075089">1BD
, Raffaele Pellegrino
  • B Data collection/entry
  • D Data interpretation
" data-author="1075090">1BD
, Iacopo Panarese
  • B Data collection/entry
  • D Data interpretation
" data-author="1075091">2BD
, Marcello Dallio
  • A Study design/planning
  • C Data analysis/statistics
  • E Preparation of manuscript
  • F Literature analysis/search
" data-author="1075092">1ACEF
, Alessandro Federico ORCID logo
  • A Study design/planning
  • B Data collection/entry
  • D Data interpretation
" data-author="1075093">1ABD

DOI: 10.12659/AJCR.936387

Am J Case Rep In Press; DOI: 10.12659/AJCR.936387  

Available online: 2022-09-16, In Press, Corrected Proof

Publication in the "In-Press" formula aims at speeding up the public availability of the pending manuscript while waiting for the final publication. The assigned DOI number is active and citable. The availability of the article in the Medline, PubMed and PMC databases as well as Web of Science will be obtained after the final publication according to the journal schedule

Abstract

BACKGROUND
There is a recognized association between inflammatory bowel disease (IBD) and hepatobiliary autoimmune disease, particularly primary sclerosing cholangitis (PSC). There have been fewer reported cases of IBD and primary biliary cholangitis (PBC), which is treated with ursodeoxycholic acid (UDCA). This report presents the case of a 60-year-old woman with PBC who was diagnosed with Crohn’s ileitis after suspension of UDCA treatment.
CASE REPORT
A 66-year-old female patient with PBC was admitted to our department for irrepressible chronic diarrhea and recurrent abdominal pain. PBC was diagnosed on the basis of serological data: chronic (>6 months) increase in alkaline phosphatase (ALP) associated with positivity for specific anti-nuclear antibodies (sp100 and gp210), without requiring a liver biopsy and a magnetic resonance cholangiopancreatography to rule out PSC. Given the intolerance and non-responsiveness according to the Toronto criteria (ALP <1.67 times the normal limit after 2 years) to UDCA at 15 mg/kg/day, an oral monotherapy treatment using obeticholic acid at 5 mg/day was prescribed. The patient complained of abdominal pain and upper gastrointestinal symptoms. The endoscopic/histologic and radiologic examinations supported the diagnosis of Crohn’s ileitis. Given the potential benefits to PBC patients of what is described as off-label therapy, budesonide at a dosage of 9 mg/day p.o. was also administered. One month after discharge, an improvement was observed both in the cholestasis indices and in gastrointestinal symptoms.
CONCLUSIONS
This report presents a case of PBC in which the patient was diagnosed with Crohn’s ileitis after cessation of treatment with UDCA, and highlights the importance of recognizing the association between autoimmune hepatobiliary disease and IBD.

Keywords: Crohn Disease; Inflammatory Bowel Diseases; Liver Cirrhosis, Biliary

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923