IgG4-related disease in autoimmune lymphoproliferative syndrome

Clin Immunol. 2017 Jul:180:97-99. doi: 10.1016/j.clim.2017.05.003. Epub 2017 May 3.

Abstract

A patient with autoimmune lymphoproliferative disorder (ALPS) developed IgG4-related disease. In retrospect, he had high levels of serum IgG4 for several years prior to presenting with IgG4-related pancreatitis. These high IgG4 levels were masked by hypergammaglobulinemia, a common feature of ALPS. We next screened 18 ALPS patients; four of them displayed increased levels of IgG4. Hence, IgG4-related disease should be considered in ALPS patients, especially in those manifesting lymphocytic organ infiltration or excessive hypergammaglobulinaemia. Screening of IgG4-related disease patients for ALPS-associated mutations would provide further information on whether this disease could be a late-onset atypical presentation of ALPS.

Keywords: ALPS; Hypergammaglobulinemia; IgG4; IgG4-related disease; Pancreatitis.

Publication types

  • Case Reports
  • Letter

MeSH terms

  • Adult
  • Autoimmune Lymphoproliferative Syndrome / blood
  • Autoimmune Lymphoproliferative Syndrome / immunology*
  • Autoimmune Lymphoproliferative Syndrome / pathology
  • Humans
  • Hypergammaglobulinemia / immunology
  • Immunoglobulin G / blood
  • Immunoglobulin G / immunology*
  • Lymph Nodes / pathology
  • Lymphoproliferative Disorders / immunology
  • Male
  • Pancreas / pathology

Substances

  • Immunoglobulin G