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Original article
Polymyalgia rheumatica-like syndrome from checkpoint inhibitor therapy: case series and systematic review of the literature
  1. Cassandra Calabrese1,
  2. Laura C Cappelli2,
  3. Marie Kostine3,
  4. Elizabeth Kirchner1,
  5. Tawnie Braaten2 and
  6. Leonard Calabrese1
  1. 1Rheumatic & Immunologic Disease, Cleveland Clinic, Cleveland, Ohio, USA
  2. 2Division of Rheumatology, Johns Hopkins School of Medicine, Baltimore, Maryland, USA
  3. 3Rheumatology Department, Centre Hospitalier Universitaire de Bordeaux, Bordeaux, France
  1. Correspondence to Dr Leonard Calabrese; calabrl{at}ccf.org

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.

  • polymyalgia rheumatica
  • inflammation
  • autoimmune diseases

This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/.

https://doi.org/10.1136/rmdopen-2019-000906

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    Footnotes

    • Contributors All authors contributed planning, data, analysis and drafting of this manuscript.

    • Funding A portion of the research reported in this publication was supported by the National Institute of Arthritis and Musculoskeletal and Skin Diseases of the National Institutes of Health under award number T32AR048522. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.

    • Competing interests CC speaks for Regeneron/Sanofi. LCC received a research grant from Bristol-Myers Squibb and consults for Regeneron/Sanofi. MK consults for Bristol-Myers-Squibb. TB is a fellow supported by T32 grant. EK consults for Celgene, Horizon, Novartis, Regeneron and speaks for Merck and Sanofi. LC consults for Bristol-Myers-Squibb, Genentech and Astra-Zeneca.

    • Patient consent for publication Not required.

    • Ethics approval This study obtained ethics approval through the ethics committees at the Cleveland Clinic (IRB# 17-575), Johns Hopkins University (# CIR00041091) and Centre Hospitalier Universitaire (local ethics committee #CE-GR-2017/007).

    • Provenance and peer review Not commissioned; externally peer reviewed.

    • Data sharing statement All data relevant to the study are included in the article or uploaded as supplementary information.