Supraglottic immunoglobulin-G4 related plasma cell granuloma: case report and literature review

Head Neck. 2014 Jun;36(6):E57-9. doi: 10.1002/hed.23519. Epub 2014 Jan 22.

Abstract

Background: Granuloma of the upper aerodigestive tract is a rare presentation of immunoglobulin-G4 (IgG4)-related disease. Since the disease process was defined in 2003, only 2 cases affecting the laryngopharynx have been reported in the literature.

Methods and results: A 62-year-old white man presented with persistent productive cough of brown sputum, globus symptoms, dysphagia, odynophagia, dysphonia, otalgia, and general malaise over a period of 2 months. Investigations revealed IgG4-positive plasma cell granuloma of the supraglottic region. This was successfully treated with oral corticosteroids.

Conclusion: Because IgG4 testing is not performed routinely, management recommendations have been poorly defined. We reviewed the literature and discuss herein the clinical characteristics, pathology, diagnosis, and management. The authors theorize that IgG4-related disease involving the larynx and pharynx may be more common than suggested by the paucity of reported cases.

Keywords: inflammatory myofibroblastic tumor; inflammatory pseudotumor; laryngeal diseases; plasma cell granuloma; serum immunoglobulin (Ig)G4 concentrations.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Administration, Oral
  • Biomarkers / blood
  • Diagnosis, Differential
  • Glottis / pathology
  • Glucocorticoids / therapeutic use
  • Granuloma, Plasma Cell / blood
  • Granuloma, Plasma Cell / diagnosis*
  • Granuloma, Plasma Cell / drug therapy
  • Humans
  • Immunoglobulin G / blood*
  • Male
  • Middle Aged
  • Treatment Outcome

Substances

  • Biomarkers
  • Glucocorticoids
  • Immunoglobulin G