Improvement of refractory pruritus after lipoprotein-apheresis in arthrogryposis-renal failure-cholestasis syndrome

J Clin Apher. 2018 Jun;33(3):401-403. doi: 10.1002/jca.21595. Epub 2017 Oct 24.

Abstract

Accumulation of bile acids can lead to invalidating pruritus in cholestatic patients. Few reports exist on the influence of lipoprotein-apheresis (LA) on plasma level of total bile acids (tBA). We report of significant decrease in tBA levels and drastic improvement of pruritus in a 5-year-old girl with arthrogryposis-renal failure-cholestasis syndrome. We present LA as a suitable rescue treatment option in therapy-refractory cholestasis-associated pruritus, at least as bridge until a long-term solution such as entero-biliary anastomosis or transplantation is possible.

Keywords: LDL-apheresis; arthrogryposis-renal failure-cholestasis syndrome; bile acids; cholestasis; lipoprotein-apheresis; pruritus.

Publication types

  • Case Reports

MeSH terms

  • Arthrogryposis
  • Bile Acids and Salts / blood*
  • Blood Component Removal / methods*
  • Child, Preschool
  • Cholestasis
  • Female
  • Humans
  • Lipoproteins / isolation & purification*
  • Pruritus / therapy*
  • Renal Insufficiency
  • Salvage Therapy / methods
  • Syndrome

Substances

  • Bile Acids and Salts
  • Lipoproteins