Sarcoidosis-like granulomatosis of the hypopharynx as a complication of anti-TNF therapy

Am J Otolaryngol. 2013 May-Jun;34(3):268-72. doi: 10.1016/j.amjoto.2012.12.016. Epub 2013 Jan 26.

Abstract

Introduction: Sarcoidosis is a multisystem granulomatous disease of unknown etiology, occasionally presenting with signs and symptoms that occur within the head and neck. Recently, granulomatous reactions and cases of sarcoidosis have been reported in patients treated with anti-TNF agents.

Methods: This report describes a 56-year-old man who developed sarcoidosis in the hypopharynx during adalimumab therapy for psoriatic arthritis. A retrospective review of the literature was performed using the PubMed database.

Results: In our patient, a chronic granulomatous reaction consistent with sarcoidosis developed after 2 years of continuous treatment with adalimumab. The diagnosis of sarcoidosis was established by the typical well-formed non caseating granulomas on biopsy, after excluding all other granulomatous conditions. Following withdrawal of anti-TNF agents and a course of steroids, the clinical picture resolved.

Conclusions: The development of sarcoidosis during treatment with TNF-a antagonists represents a rare and paradoxical adverse event. To our knowledge this is the first case of sarcoidosis of the hypopharynx reported in the literature.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adalimumab
  • Anti-Inflammatory Agents / adverse effects
  • Antibodies, Monoclonal, Humanized / adverse effects*
  • Antibodies, Monoclonal, Humanized / therapeutic use
  • Arthritis, Psoriatic / drug therapy
  • Humans
  • Hypopharynx*
  • Male
  • Middle Aged
  • Oropharynx / diagnostic imaging
  • Oropharynx / pathology
  • Pharyngeal Diseases / chemically induced*
  • Sarcoidosis / chemically induced*
  • Tomography, X-Ray Computed
  • Tongue / pathology
  • Tumor Necrosis Factor-alpha / antagonists & inhibitors*
  • Ulcer / complications

Substances

  • Anti-Inflammatory Agents
  • Antibodies, Monoclonal, Humanized
  • Tumor Necrosis Factor-alpha
  • Adalimumab