Polymyalgia rheumatica-like syndrome from checkpoint inhibitor therapy: case series and systematic review of the literature

RMD Open. 2019 Apr 25;5(1):e000906. doi: 10.1136/rmdopen-2019-000906. eCollection 2019.

Abstract

Objective: To assess whether the polymyalgia rheumatica (PMR)-like syndrome reported as an immune related adverse event (irAE) from checkpoint inhibitor therapy is consistent with the 2012 European League Against Rheumatism (EULAR)/American College of Rheumatology (ACR) provisional criteria for PMR.

Methods: The cases were derived from two sources. Group 1 represents reported cases from three contributing centres. Group 2 was derived from a systematic review of the literature searching for all cases reported as PMR or PMR-like illness associated with checkpoint inhibitor therapy. Cases were assessed for the quality of reporting and then analysed to determine whether they fulfilled the 2012 EULAR/ACR provisional criteria for PMR.

Results: A total of 49 patients were included for analysis. Among the entire group, 37 (75%) were designated 'complete' indicating that they had sufficient data to reliably apply the 2012 EULAR/ACR criteria. 28 (75%) cases fulfilled complete criteria for PMR. A number of cases also demonstrated some clinical features unusual for idiopathic PMR.

Conclusion: This study suggests a high proportion of reported cases of checkpoint inhibitor-related PMR fulfil preliminary criteria for PMR, yet in one quarter clinical details were incomplete making verification problematic. Furthermore, in the absence of a gold standard for the diagnosis of PMR, the relationship of checkpoint inhibitor-related PMR to the idiopathic form remains unclear.

Keywords: autoimmune diseases; inflammation; polymyalgia rheumatica.

Publication types

  • Case Reports
  • Meta-Analysis
  • Research Support, N.I.H., Extramural
  • Systematic Review

MeSH terms

  • Antirheumatic Agents / pharmacology
  • Antirheumatic Agents / therapeutic use*
  • Biomarkers*
  • Humans
  • Molecular Targeted Therapy* / methods
  • Polymyalgia Rheumatica / drug therapy*
  • Polymyalgia Rheumatica / etiology
  • Treatment Outcome

Substances

  • Antirheumatic Agents
  • Biomarkers