Pediatric Gastroenterology, Hepatology & Nutrition

The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition (대한소아소화기영양학회)
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Urosodeoxycholic Acid Therapy in a Child with Trimethoprim- Sulfamethoxazole-induced Vanishing Bile Duct Syndrome

  • Cho, Hyun Jeong (Department of Pediatrics, Chungnam National University School of Medicine) ;
  • Jwa, Hye Jeong (Department of Pediatrics, Chungnam National University School of Medicine) ;
  • Kim, Kyu Seon (Department of Pediatrics, Chungnam National University School of Medicine) ;
  • Gang, Dae Yong (Department of Pathology, Chungnam National University School of Medicine) ;
  • Kim, Jae Young (Department of Pediatrics, Chungnam National University School of Medicine)
  • Received : 2013.10.05
  • Accepted : 2013.10.28
  • Published : 2013.12.30
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Abstract

We present a case of a 7-year-old boy who had cholestasis after trimethoprim-sulfamethoxazole combination therapy. Liver biopsy was performed 36 days after the onset of jaundice because of no evidence of improving cholestasis. Liver histology revealed portal inflammation, bile plug, and biliary stasis around the central vein with the loss of the interlobular bile ducts. Immunohistochemical stains for cytokeratin 7 and 19 were negative. These findings were consistent with those of vanishing bile duct syndrome (VBDS). Chlestasis was progressively improved with dose increment of urosodeoxycholic acid from conventional to high dose. This is the first case report of trimethoprime-sulfamethoxazole associated VBDS in Korean children. The case suggests that differential diagnosis of VBDS should be considered in case of progressive cholestatic hepatitis with elevation of alkaline phosphatase and gamma-glutamyl transpeptidase after or during taking medicine to treat nonhepatobiliary diseases illness.

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References

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