Treatment of hyper-IgG4 disease with sequential corticosteroids and tamoxifen - case report and review of the literature

Clin Nephrol. 2009 Nov;72(5):414-7. doi: 10.5414/cnp72414.

Abstract

We report a patient with multifocal fibrosclerosis presenting as sialadenitis, hepatic fibrosis, and retroperitoneal fibrosis with renal failure. His medical management consisted of prednisone (4 months at 40 mg daily, then tapered down to 5 mg daily for another 14 months) and 18 months of tamoxifen. He responded clinically and radiographically to this regimen, and remains in clinical remission 10 months after discontinuing medical therapy. Subsequent histologic examination of submandibular gland tissue revealed strong staining for IgG4-positive plasma cells. To our knowledge, this is the first case of confirmed multifocal hyper-IgG4 disease to be successfully treated with sequential corticosteroids and tamoxifen.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Fibrosis
  • Glucocorticoids / administration & dosage*
  • Humans
  • Hypergammaglobulinemia / complications
  • Hypergammaglobulinemia / diagnosis
  • Hypergammaglobulinemia / drug therapy*
  • Immunoglobulin G / analysis*
  • Male
  • Middle Aged
  • Prednisone / administration & dosage*
  • Renal Insufficiency / complications
  • Renal Insufficiency / immunology
  • Retroperitoneal Fibrosis / complications
  • Retroperitoneal Fibrosis / immunology
  • Retroperitoneal Fibrosis / pathology
  • Sialadenitis / complications
  • Sialadenitis / immunology
  • Sialadenitis / pathology
  • Tamoxifen / administration & dosage*

Substances

  • Glucocorticoids
  • Immunoglobulin G
  • Tamoxifen
  • Prednisone