What is the clinical evidence from this study?

Study design: Case Report. Population: Multiple Autoimmune Syndrome with Acute Myocarditis (n=1). Intervention: Supportive heart failure therapy (ramipril and bisoprolol). Primary outcome: Clinical and echocardiographic improvement.

March 18, 2025Open Access

An atypical multiple autoimmune syndrome: A case report including myocarditis

ITIkram TahaniMohamed I University FAFadoua AchourMohamed I University NONoha El OuafiMohamed I University

Key Result

A 46-year-old female with rheumatoid arthritis and secondary Sjögren's syndrome developed MRI-confirmed acute myocarditis, representing a rare and atypical presentation of multiple autoimmune syndrome.

Study Design

Type

Case Report (n=1)

Multicenter

Structured PICO

Population

A 46-year-old female with rheumatoid arthritis and secondary Sjögren's syndrome who developed acute myocarditis.

Intervention

Initiation of ramipril 5 mg and bisoprolol 2.5 mg, alongside continuation of methotrexate 20 mg weekly and hydrocortisone 20 mg daily.

Outcome

Clinical progression and improvement in left ventricular systolic function.

This case highlights the rare occurrence of acute myocarditis as part of a multiple autoimmune syndrome involving rheumatoid arthritis and Sjögren's syndrome, which responded well to standard heart failure therapy.

Limitations

Endomyocardial biopsy was not performed to definitively confirm the autoimmune etiology of the myocarditis.

Abstract

The co-occurrence of autoimmune diseases has been epidemiologically studied and has contributed to our understanding of autoimmunity. The underlying mechanisms of this syndrome remain elusive; however, its prevalence may be higher than currently documented. The co-occurrence of at least 3 autoimmune diseases in a single patient has been defined as multiple autoimmune syndrome (MAS). This syndrome can be classified into 3 categories based on the prevalence of their associations with one another: type 1, type 2, and type 3. Myocarditis is classified as type 3, and typically does not present with connective tissue involvement. This report presents the case of a young female with known autoimmune disorders: rheumatoid arthritis (RA)and secondary Sjögren's syndrome (GSJ). The patient presented with MRI-confirmed myocarditis, likely of autoimmune origin, combining the 3 conditions in an atypical presentation. An additional noteworthy aspect of this case is the uncommon occurrence of myocarditis in rheumatoid arthritis, and even more so in Sjögren's syndromes.

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Cite This Study

Tahani et al. (Tue,) conducted a case report in Multiple Autoimmune Syndrome with Acute Myocarditis (n=1). Supportive heart failure therapy (ramipril and bisoprolol) was evaluated on Clinical and echocardiographic improvement. A 46-year-old female with rheumatoid arthritis and secondary Sjögren's syndrome developed MRI-confirmed acute myocarditis, representing a rare and atypical presentation of multiple autoimmune syndrome.

synapsesocial.com/papers/6a207b461f3770407f0999e5 https://doi.org/https://doi.org/10.1016/j.radcr.2025.02.067

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