Cargando…

Myocarditis as the Initial Presentation of a Refractory Adult-Onset Still’s Disease

We present a case of adult-onset Still’s disease, a rare disease that presented with a rare complication, myocarditis. After initial therapy with prednisolone 1 mg/kg/day, the patient experienced dyspnea due to severe pleural effusion and palpitations on account of new-onset supraventricular tachyca...

Descripción completa

Detalles Bibliográficos
Autores principales: Pina Gonçalves, Nuno, Lourenço, Maria Helena, Albuquerque, Francisco, Gonçalves, Maria J, Falcão, Sandra
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Cureus 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9623201/
https://www.ncbi.nlm.nih.gov/pubmed/36337832
http://dx.doi.org/10.7759/cureus.29821
_version_ 1784821943961124864
author Pina Gonçalves, Nuno
Lourenço, Maria Helena
Albuquerque, Francisco
Gonçalves, Maria J
Falcão, Sandra
author_facet Pina Gonçalves, Nuno
Lourenço, Maria Helena
Albuquerque, Francisco
Gonçalves, Maria J
Falcão, Sandra
author_sort Pina Gonçalves, Nuno
collection PubMed
description We present a case of adult-onset Still’s disease, a rare disease that presented with a rare complication, myocarditis. After initial therapy with prednisolone 1 mg/kg/day, the patient experienced dyspnea due to severe pleural effusion and palpitations on account of new-onset supraventricular tachycardia. Therapy with three consecutive pulses of 1000 mg of methylprednisolone and anakinra 100 mg/day proved to be effective, with a progressive resolution of all symptoms. However, at three months follow-up, oligoarthritis recurred along with high serum ferritin. Secondary failure to anakinra was assumed, and a switch to tocilizumab 162 mg was made. Nevertheless, arthralgia of the wrists and knees as well as high serum ferritin still persisted after two months of therapy. Chronic disease was assumed, and the patient switched to canakinumab 4 mg/kg/dose q4week, and a complete resolution of symptoms occurred with normalization of inflammation markers. Follow-up cardiac magnetic resonance showed a complete resolution of heart involvement.
format Online
Article
Text
id pubmed-9623201
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Cureus
record_format MEDLINE/PubMed
spelling pubmed-96232012022-11-04 Myocarditis as the Initial Presentation of a Refractory Adult-Onset Still’s Disease Pina Gonçalves, Nuno Lourenço, Maria Helena Albuquerque, Francisco Gonçalves, Maria J Falcão, Sandra Cureus Cardiology We present a case of adult-onset Still’s disease, a rare disease that presented with a rare complication, myocarditis. After initial therapy with prednisolone 1 mg/kg/day, the patient experienced dyspnea due to severe pleural effusion and palpitations on account of new-onset supraventricular tachycardia. Therapy with three consecutive pulses of 1000 mg of methylprednisolone and anakinra 100 mg/day proved to be effective, with a progressive resolution of all symptoms. However, at three months follow-up, oligoarthritis recurred along with high serum ferritin. Secondary failure to anakinra was assumed, and a switch to tocilizumab 162 mg was made. Nevertheless, arthralgia of the wrists and knees as well as high serum ferritin still persisted after two months of therapy. Chronic disease was assumed, and the patient switched to canakinumab 4 mg/kg/dose q4week, and a complete resolution of symptoms occurred with normalization of inflammation markers. Follow-up cardiac magnetic resonance showed a complete resolution of heart involvement. Cureus 2022-10-01 /pmc/articles/PMC9623201/ /pubmed/36337832 http://dx.doi.org/10.7759/cureus.29821 Text en Copyright © 2022, Pina Gonçalves et al. https://creativecommons.org/licenses/by/3.0/This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Cardiology
Pina Gonçalves, Nuno
Lourenço, Maria Helena
Albuquerque, Francisco
Gonçalves, Maria J
Falcão, Sandra
Myocarditis as the Initial Presentation of a Refractory Adult-Onset Still’s Disease
title Myocarditis as the Initial Presentation of a Refractory Adult-Onset Still’s Disease
title_full Myocarditis as the Initial Presentation of a Refractory Adult-Onset Still’s Disease
title_fullStr Myocarditis as the Initial Presentation of a Refractory Adult-Onset Still’s Disease
title_full_unstemmed Myocarditis as the Initial Presentation of a Refractory Adult-Onset Still’s Disease
title_short Myocarditis as the Initial Presentation of a Refractory Adult-Onset Still’s Disease
title_sort myocarditis as the initial presentation of a refractory adult-onset still’s disease
topic Cardiology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9623201/
https://www.ncbi.nlm.nih.gov/pubmed/36337832
http://dx.doi.org/10.7759/cureus.29821
work_keys_str_mv AT pinagoncalvesnuno myocarditisastheinitialpresentationofarefractoryadultonsetstillsdisease
AT lourencomariahelena myocarditisastheinitialpresentationofarefractoryadultonsetstillsdisease
AT albuquerquefrancisco myocarditisastheinitialpresentationofarefractoryadultonsetstillsdisease
AT goncalvesmariaj myocarditisastheinitialpresentationofarefractoryadultonsetstillsdisease
AT falcaosandra myocarditisastheinitialpresentationofarefractoryadultonsetstillsdisease