Sarcoidosis appearing during anti-tumor necrosis factor alpha therapy: a new "class effect" paradoxical phenomenon. Two case reports and literature review

Semin Arthritis Rheum. 2010 Feb;39(4):313-9. doi: 10.1016/j.semarthrit.2008.11.003. Epub 2009 Jan 15.

Abstract

Objectives: To report 2 cases of sarcoidosis that developed during treatment with tumor necrosis factor alpha (TNFalpha) antagonists, infliximab and adalimumab, used for inflammatory rheumatic disease and to review previously reported cases.

Methods: We describe 2 patients, the first with psoriatic arthritis, the second with rheumatoid arthritis, who developed noncaseating granulomas of the lungs consistent with sarcoidosis while being treated with anti-TNFalpha drugs. A retrospective review of the literature was performed using the PubMed database.

Results: In our patients sarcoidosis developed after 2 years of continuous treatment with infliximab and adalimumab. Both patients presented with low-grade fever, chest pain, and dyspnea. The diagnosis of sarcoidosis was established by the typical well-formed noncaseating granulomas on transbronchial biopsy, after excluding all other granulomatous conditions. Following withdrawal of anti-TNFalpha agents and a brief course of steroids, the clinical picture resolved. Thirteen additional cases of sarcoidosis that developed after anti-TNFalpha treatment have been reported, and in 9 of these the causative agent was etanercept.

Conclusions: The development of sarcoidosis during treatment with TNFalpha antagonists represents a rare and paradoxical adverse event. The occurrence of sarcoidosis with all 3 available agents suggests a new "class effect" probably linked to a cytokine disequilibrium in patients receiving anti-TNFalpha treatment.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Adalimumab
  • Adult
  • Antibodies, Monoclonal / adverse effects*
  • Antibodies, Monoclonal, Humanized
  • Antirheumatic Agents / adverse effects
  • Arthritis, Psoriatic / therapy*
  • Arthritis, Rheumatoid / therapy*
  • Female
  • Humans
  • Infliximab
  • Male
  • Middle Aged
  • Sarcoidosis / chemically induced*
  • Sarcoidosis / diagnosis
  • Treatment Outcome
  • Tumor Necrosis Factor-alpha / antagonists & inhibitors

Substances

  • Antibodies, Monoclonal
  • Antibodies, Monoclonal, Humanized
  • Antirheumatic Agents
  • Tumor Necrosis Factor-alpha
  • Infliximab
  • Adalimumab