Successful response of primary sclerosing cholangitis and associated ulcerative colitis to oral vancomycin may depend on
- PDF / 1,333,822 Bytes
- 6 Pages / 595.276 x 790.866 pts Page_size
- 24 Downloads / 157 Views
CASE REPORT
Successful response of primary sclerosing cholangitis and associated ulcerative colitis to oral vancomycin may depend on brand and personalized dose: report in an adolescent Cynthia W. Buness1 · Kevin M. Johnson2 · Ahmad Hassan Ali3 · Leina Alrabadi4 · Keith D. Lindor5 · Tamir Miloh6 · Kenneth L. Cox7,8 Received: 6 October 2020 / Accepted: 5 November 2020 © Japanese Society of Gastroenterology 2020
Abstract Primary sclerosing cholangitis (PSC) is a rare, progressive liver disease characterized by cholestasis and bile duct fibrosis that has no accepted therapy known to delay or arrest its progression. We report a 23-year-old female patient who at age 14 was diagnosed with moderate pancolonic ulcerative colitis (UC) and at age 15 with small-duct PSC unresponsive to conventional therapy. The patient began single drug therapy with the antibiotic oral vancomycin (OVT) and achieved normalization of liver enzymes and resolution of UC symptoms with colonic mucosal healing. These improvements have persisted over 8 years. There has been no colon dysplasia, liver fibrosis or failure, bile duct stricture, or cancer. Of note, the patient’s response was dependent on the brand of oral vancomycin capsule, as well as dose. This raised the questions of possible differences in bioequivalence of different commercial versions of the drug and whether this factor might play into the variability of efficacy seen in published trials. Evidence suggests that oral vancomycin both alters the intestinal microbiome and has immunomodulatory effects. Its striking effectiveness in this and other patients supports further investigation in randomized trials, with careful attention to its bioavailability profile in the gut. Keywords Primary sclerosing cholangitis · Ulcerative colitis · Vancomycin · Microbiome · Bioequivalence
Introduction * Kenneth L. Cox [email protected] 1
National Patient Advocate Foundation, Arizona State University, Phoenix, AZ, USA
2
Department of Radiology and Biomedical Imaging, Yale School of Medicine, New Haven, CT, USA
3
Division of Gastroenterology and Hepatology, Mayo Clinic, Rochester, MN, USA
4
Division of Pediatric Gastroenterology, Hepatology and Nutrition, Stanford University, Palo Alto, CA, USA
5
Division of Gastroenterology and Hepatology, Mayo Clinic, Scottsdale, AZ, USA
6
Miami Transplant Institute, 1801 NW 9th Avenue, Miami, FL, USA
7
Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Stanford University, Palo Alto, CA, USA
8
Emeritus Professor of Pediatrics, Stanford University, Palo Alto, CA, USA
Primary sclerosing cholangitis (PSC) is a rare progressive disease of the bile ducts closely linked to inflammatory bowel disease (IBD), especially ulcerative colitis (UC). It is associated with an increased risk for cirrhosis and cholangiocarcinoma [1–3]. The etiology is poorly understood; one theory is that an abnormal gut microbiome activates innate immunity within the liver, resulting in bile duct-targeted inflammation and biliary fibrosis [4].
Data Loading...
