Unusual Polyclonal IgM in an IgG Deficient Patient with Autoimmune Lymphoproliferative Syndrome: A Case Study and Literature Review

Ann Clin Lab Sci. 2022 Nov;52(6):996-1001.

Abstract

Autoimmune lymphoproliferative syndrome (ALPS) is a rare genetic disease caused germline mutation of FAS gene, gene encoding Fas ligand or Caspase 10 gene. However, in 20% of all ALPS patients, genetic defect is unknown. We presented a case of a 20-year-old male with a history of autoimmune lymphoproliferative syndrome (ALPS; confirmed by genetic study) who came to our medical center with a concern for malignancy. Although no malignancy was detected, his lack of IgA, very low level of IgG (requiring therapy with intravenous IgG) and highly elevated polyclonal IgM (hyperimmunoglobulin M syndrome) were unusual findings because ALPS patients with hypergammaglobulinemia usually demonstrate elevated IgA or IgG.

Keywords: Autoimmune lymphoproliferative syndrome; Casease 10; FAS gene; Fas-ligand; Hyperimmunoglobulin M syndrome.

Publication types

  • Review
  • Case Reports

MeSH terms

  • Adult
  • Apoptosis
  • Autoimmune Lymphoproliferative Syndrome* / complications
  • Autoimmune Lymphoproliferative Syndrome* / genetics
  • Humans
  • Immunoglobulin A / genetics
  • Immunoglobulin G
  • Immunoglobulin M
  • Male
  • Mutation
  • Young Adult
  • fas Receptor / genetics

Substances

  • fas Receptor
  • Immunoglobulin A
  • Immunoglobulin G
  • Immunoglobulin M