Ocular adnexal IgG4-related disease: comparative analysis with mucosa-associated lymphoid tissue lymphoma and other chronic inflammatory conditions

Histopathology. 2012 Jan;60(2):296-312. doi: 10.1111/j.1365-2559.2011.04089.x.

Abstract

Aims: Making a differential diagnosis of IgG4-related disease from mucosa-associated lymphoid tissue (MALT) lymphoma or any other chronic inflammation is often challenging. Moreover, the association with secondary lymphoma of ocular adnexal IgG4-related disease needs to be elucidated.

Methods and results: We investigated 14 cases of IgG4-related disease, nine MALT lymphomas and 12 other chronic inflammations involving the lacrimal gland and orbit. Bilateral involvement was frequent in IgG4-related diseases. The number of IgG4-positive cells and the ratio of IgG4/IgG-positive cells were higher in patients with IgG4-related disease than in those with MALT lymphoma (P = 0.016; P < 0.001) and other types of inflammation (P < 0.001; P < 0.001). Monoclonal B cell proliferation was suspected in two cases (14.3%) of IgG4-related disease. One of these patients also displayed monomorphous features suggesting secondary MALT lymphoma. In the other case, κ-chain restriction in IgG4-positive cells was observed, raising the possibility of IgG4-producing MALT lymphoma. Trisomy 3, trisomy 18 or MALT1 translocation was observed in none of the IgG4-related cases. Regulatory T-cell infiltration was higher in cases of IgG4-related disease than in MALT lymphomas (P < 0.001) and other types of inflammation (P = 0.006).

Conclusions: Some genetically and morphologically complicated cases of ocular adnexal IgG4-related disease emphasize the need for in-depth studies to differentiate this disease from MALT lymphoma, and to exclude secondary lymphoma.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adnexal Diseases / diagnosis
  • Adnexal Diseases / metabolism
  • Adnexal Diseases / pathology*
  • Adult
  • Aged
  • Diagnosis, Differential
  • Eyelid Diseases / diagnosis
  • Eyelid Diseases / metabolism
  • Eyelid Diseases / pathology*
  • Female
  • Forkhead Transcription Factors / metabolism
  • Humans
  • Immunoglobulin G / metabolism*
  • Immunophenotyping
  • Lacrimal Apparatus Diseases / diagnosis
  • Lacrimal Apparatus Diseases / metabolism
  • Lacrimal Apparatus Diseases / pathology*
  • Lymphoma, B-Cell, Marginal Zone / diagnosis
  • Lymphoma, B-Cell, Marginal Zone / metabolism
  • Lymphoma, B-Cell, Marginal Zone / pathology*
  • Male
  • Middle Aged
  • Orbital Diseases / diagnosis
  • Orbital Diseases / metabolism
  • Orbital Diseases / pathology*
  • Retrospective Studies
  • T-Lymphocytes, Regulatory / metabolism
  • T-Lymphocytes, Regulatory / pathology

Substances

  • FOXP3 protein, human
  • Forkhead Transcription Factors
  • Immunoglobulin G