Febuxostat for the Prevention of Recurrent 2,8-dihydroxyadenine Nephropathy due to Adenine Phosphoribosyltransferase Deficiency Following Kidney Transplantation

Intern Med. 2017;56(11):1387-1391. doi: 10.2169/internalmedicine.56.8142. Epub 2017 Jun 1.

Abstract

Adenine phosphoribosyltransferase (APRT) deficiency is a rare autosomal recessive disorder that results in irreversible renal damage due to 2,8-dihydroxyadenine (DHA) nephropathy. A 28-year-old man underwent living-related kidney transplantation for chronic kidney disease of unknown etiology. Numerous spherical brownish crystals observed in his urinary sediment on postoperative day 3 and were observed within the tubular lumen of renal allograft biopsy specimens on postoperative day 7. After a genetic diagnosis, febuxostat treatment was started on postoperative day 7, with the dosage gradually increased to 80 mg/day until complete the disappearance of 2,8-DHA crystals. Febuxostat prevented secondary 2,8-DHA nephropathy after kidney transplantation.

Keywords: 2,8-dihydroxyadenine nephropathy; adenine phosphoribosyltransferase deficiency; febuxostat; kidney transplantation; nephrolithiasis; xanthine dehydrogenase inhibitor.

Publication types

  • Case Reports

MeSH terms

  • Adenine / analogs & derivatives
  • Adenine / metabolism
  • Adenine Phosphoribosyltransferase / deficiency*
  • Adult
  • Febuxostat / administration & dosage*
  • Gout Suppressants / administration & dosage*
  • Humans
  • Kidney Transplantation / methods*
  • Male
  • Metabolism, Inborn Errors / prevention & control*
  • Urolithiasis / prevention & control*

Substances

  • Gout Suppressants
  • Febuxostat
  • 2,8-dihydroxyadenine
  • Adenine Phosphoribosyltransferase
  • Adenine

Supplementary concepts

  • Adenine phosphoribosyltransferase deficiency