Top

Published in:

01-12-2020 | Colchicine | Case Report

Failure of anti Interleukin-1 β monoclonal antibody in the treatment of recurrent pericarditis in two children

Authors: Sara Signa, Matteo D’Alessandro, Rita Consolini, Angela Miniaci, Marta Bustaffa, Chiara Longo, Maria A. Tosca, Martina Bizzi, Roberta Caorsi, Leonardo Oliveira Mendonça, Andrea Pession, Angelo Ravelli, Marco Gattorno

Published in: Pediatric Rheumatology | Issue 1/2020

Abstract

Background

Recurrent pericarditis (RP) is a complication (15–30%) of acute pericarditis with an unknown etiology. Treatment regimen consists of a combination of non-steroidal anti-inflammatory drugs (NSAIDs) and colchicine, with the addition of corticosteroids in resistant or intolerant cases. In the last decade anakinra was shown as an effective treatment in patients with colchicine resistant and steroid-dependent RP, initially in anecdotal reports in children and more recently in a randomized trial. Canakinumab is a monoclonal antibody selectively blocking IL-1β and its use is only anecdotally reported to treat pericarditis. We report two pediatric patients with refractory recurrent pericarditis, who presented an optimal response to anakinra treatment but prompt relapse after switch to canakinumab.

Case presentation

The first patient is a girl with Recurrent Pericarditis started in April 2015, after heart surgery. NSAIDs and oral steroids were started, with prompt relapse after steroid suspension. The child showed a steroid-dependent RP; anakinra was therefore started with excellent response, but discontinued after 2 weeks for local reactions. In July 2016 therapy with canakinumab was started. She experienced four relapses during canakinumab therapy despite dosage increase and steroid treatment. In January 2018 a procedure of desensitization from anakinra was performed, successfully. Anakinra as monotherapy is currently ongoing, without any sign of flare.

The second patient is a girl with an idiopathic RP, who showed an initial benefit from NSAIDs and colchicine. However, 10 days after the first episode a relapse occurred and therapy with anakinra was established. Two months later, while being in complete remission, anakinra was replaced with canakinumab due to patient’s poor compliance to daily injections. She experienced a relapse requiring steroids 10 days after the first canakinumab injection. Anakinra was subsequently re-started with complete remission, persisting after 24 months follow-up.

Conclusions

We describe two cases of failure of the treatment with anti-IL-1β monoclonal antibodies in steroid- dependent idiopathic RP. This anecdotal and preliminary observation suggests a different efficacy of the two IL-1 blockers in the management of RP and support a possible pivotal role of IL-1α in the pathogenesis of this condition.

Imazio M, Gaita F. Acute and recurrent pericarditis. Cardiol Clin. 2017;35(4):505–13. https://doi.org/10.1016/j.ccl.2017.07.004.CrossRefPubMed

Brucato A, Emmi G, Cantarini L, et al. Management of idiopathic recurrent pericarditis in adults and in children: a role for IL-1 receptor antagonism. Intern Emerg Med. 2018;13(4):475–89. https://doi.org/10.1007/s11739-018-1842-x.CrossRefPubMed

Picco P, Brisca G, Traverso F, Loy A, Gattorno M, Martini A. Successful treatment of idiopathic recurrent pericarditis in children with interleukin-1β receptor antagonist (anakinra): an unrecognized autoinflammatory disease? Arthritis Rheum. 2009;60(1):264–8. https://doi.org/10.1002/art.24174.CrossRefPubMed

Galluzzo A, Imazio M. Advances in medical therapy for pericardial diseases. Expert Rev Cardiovasc Ther. 2018;16(9):635–43. https://doi.org/10.1080/14779072.2018.1510315.CrossRefPubMed

Brucato A, Imazio M, Gattorno M, et al. Effect of Anakinra on recurrent pericarditis among patients with colchicine resistance and corticosteroid dependence. JAMA. 2016;316(18):1906. https://doi.org/10.1001/jama.2016.15826.CrossRefPubMed

Imazio M, Andreis A, De Ferrari GM, et al. Anakinra for corticosteroid-dependent and colchicine-resistant pericarditis: the IRAP (international registry of Anakinra for pericarditis) study. Eur J Prev Cardiol. 2019. https://doi.org/10.1177/2047487319879534.

Kougkas N, Fanouriakis A, Papalopoulos I, et al. Canakinumab for recurrent rheumatic disease associated-pericarditis: a case series with long-term follow-up. Rheumatology. 2018;57(8):1494–5. https://doi.org/10.1093/rheumatology/key077.CrossRefPubMed

Epçaçan S, Sahin S, Kasapcopur O. Anaphylactic reaction to anakinra in a child with steroid-dependent idiopathic recurrent pericarditis and successful management with canakinumab. Cardiol Young. 2019;29(4):549–51. https://doi.org/10.1017/S1047951119000672.CrossRefPubMed

Mendonca LO, Malle L, Donovan FX, et al. Deficiency of Interleukin-1 receptor antagonist (DIRA): report of the first Indian patient and a novel deletion affecting IL1RN. J Clin Immunol. 2017;37(5):445–51. https://doi.org/10.1007/s10875-017-0399-1.CrossRefPubMed

10.

Yilmaz TM, Bahçecioğlu SN. Successful rapid subcutaneous desensitization to anakinra in a case with a severe immediate-type hypersensitivity reaction. Eur Ann Allergy Clin Immunol. 2018;50(2):94–6. https://doi.org/10.23822/EurAnnACI.1764-1489.30.CrossRefPubMed

11.

Di Paolo NC, Shayakhmetov DM. Interleukin 1α and the inflammatory process. Nat Immunol. 2016;17(8):906–13. https://doi.org/10.1038/ni.3503.CrossRefPubMedPubMedCentral

12.

Finetti M, Insalaco A, Cantarini L, et al. Long-term efficacy of interleukin-1 receptor antagonist (Anakinra) in corticosteroid-dependent and colchicine-resistant recurrent pericarditis. J Pediatr. 2014;164(6):1425–1431.e1. https://doi.org/10.1016/j.jpeds.2014.01.065.CrossRefPubMed

Title: Failure of anti Interleukin-1 β monoclonal antibody in the treatment of recurrent pericarditis in two children
Authors: Sara Signa
Matteo D’Alessandro
Rita Consolini
Angela Miniaci
Marta Bustaffa
Chiara Longo
Maria A. Tosca
Martina Bizzi
Roberta Caorsi
Leonardo Oliveira Mendonça
Andrea Pession
Angelo Ravelli
Marco Gattorno
Publication date: 01-12-2020
Publisher: BioMed Central
Keywords: Colchicine
Anakinra
Published in: Pediatric Rheumatology / Issue 1/2020
Electronic ISSN: 1546-0096
DOI: https://doi.org/10.1186/s12969-020-00438-5

At a glance: The STEP trials

Springer Medicine

Failure of anti Interleukin-1 β monoclonal antibody in the treatment of recurrent pericarditis in two children

Abstract

Background

Case presentation

Conclusions

At a glance: The STEP trials

Springer Medicine

Abstract

Background

Case presentation

Conclusions

Please log in to get access to this content

Other articles of this Issue 1/2020

The use of S100 proteins testing in juvenile idiopathic arthritis and autoinflammatory diseases in a pediatric clinical setting: a retrospective analysis

C-type natriuretic peptide stimulates osteoblastic proliferation and collagen-X expression but suppresses fibroblast growth factor-23 expression in vitro

Children’s fear of needle injections: a qualitative study of training sessions for children with rheumatic diseases before home administration

Spectrum of paediatric rheumatic disorders at a tertiary hospital in Tanzania

Image guided sacroiliac joint corticosteroid injections in children: an 18-year single-center retrospective study

Joint injection practice variations in pediatric rheumatology – a global survey and call for action