Multicentric carpal-tarsal osteolysis with nephropathy treated successfully with cyclosporine A: a case report and literature review

Am J Kidney Dis. 2007 Oct;50(4):649-54. doi: 10.1053/j.ajkd.2007.06.014.

Abstract

Multicentric carpal-tarsal osteolysis is a rare skeletal disorder characterized by osteolysis of the metacarpal, carpal, and tarsal bones and leading to crippling joint deformities. Progressive nephropathy occurs in more than half the cases. All previously reported series with renal biopsies showed only end-stage renal disease on histological examination because of the late presentation to nephrologists. Accurate diagnosis of the underlying renal pathological state therefore has not been possible. We report the first case in which early and sequential renal biopsies were performed. These show the renal lesion to be focal and segmental glomerulosclerosis, which was treated successfully with cyclosporine A.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Carpal Bones / pathology*
  • Child
  • Cyclosporine / therapeutic use*
  • Hajdu-Cheney Syndrome / complications
  • Hajdu-Cheney Syndrome / diagnosis
  • Hajdu-Cheney Syndrome / drug therapy*
  • Humans
  • Kidney Diseases / complications
  • Kidney Diseases / diagnosis
  • Kidney Diseases / drug therapy*
  • Male
  • Tarsal Bones / pathology*

Substances

  • Cyclosporine