Clinicopathologic features of orbital immunoglobulin G4-related disease (IgG4-RD): a case series and literature review

Graefes Arch Clin Exp Ophthalmol. 2015 May;253(5):803-9. doi: 10.1007/s00417-014-2905-y. Epub 2015 Jan 10.

Abstract

Background: Involvement of orbital structures by immunoglobulin G4-related disease (IgG4-RD) is not uncommon. We conducted this study to evaluate the clinicopathologic features of orbital IgG4-RD.

Material/methods: This was a retrospective, clinicopathologic study. Clinical records, light microscopic features, results of immunostaining with IgG & IgG4 and laboratory findings were reviewed in 16 patients diagnosed with orbital IgG4-RD.

Results: Eleven patients had a bilateral disease, and the lacrimal gland was involved in 14. Dense sclerosis, plasma cell aggregates and dense lymphoplasmacytic infiltrate were seen in all patients. Serum IgG4 titre was elevated in 12 patients. Nine patients responded completely to glucocorticoid treatment. Five patients had a relapse on discontinuation of treatment.

Conclusion: Orbital IgG4-RD is a distinct clinicopathologic entity requiring increased awareness and needs to be differentiated from other orbital lymphoproliferative lesions.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Adult
  • Aged
  • Child
  • Female
  • Glucocorticoids / therapeutic use
  • Humans
  • Immunoglobulin G / blood*
  • Immunosuppressive Agents / therapeutic use
  • Infusions, Intravenous
  • Lacrimal Apparatus Diseases / drug therapy
  • Lacrimal Apparatus Diseases / immunology
  • Lacrimal Apparatus Diseases / pathology*
  • Male
  • Methylprednisolone / therapeutic use
  • Middle Aged
  • Orbital Myositis / drug therapy
  • Orbital Myositis / immunology
  • Orbital Myositis / pathology*
  • Paraproteinemias / drug therapy
  • Paraproteinemias / immunology
  • Paraproteinemias / pathology*
  • Plasma Cells / pathology*
  • Retrospective Studies
  • Tomography, X-Ray Computed

Substances

  • Glucocorticoids
  • Immunoglobulin G
  • Immunosuppressive Agents
  • Methylprednisolone