Unusual instance of multiply triggered immune disorders, notably witnessed in a case study with myocarditis involvement

Unusual instance of multiply triggered immune disorders, notably witnessed in a case study with myocarditis involvement

A unique case of Multiple Autoimmune Syndrome (MAS) has been reported, involving a 46-year-old female patient with co-existing rheumatoid arthritis (RA) and secondary Sjögren's syndrome (GSJ), and an additional complication of myocarditis. This triad, consisting of an inflammatory heart condition, RA, and Sjögren's syndrome, is seldom documented in the medical literature.

The patient presented with a markedly elevated troponin level of 1090 ng/ml, indicating myocardial damage, and MRI-confirmed myocarditis, likely of autoimmune origin. Myocarditis can occur secondary to autoimmune disease activity, but its occurrence in RA and Sjögren's syndromes is uncommon.

In patients with myocarditis, higher T1, T2, and extracellular volume fraction (ECV) values, along with a modest prevalence of late gadolinium enhancement (LGE), have been observed. Interstitial and granulomatous forms of myocarditis have been the most frequently described in the course of RA, with a greater specificity for the granulomatous form.

The co-occurrence of at least three autoimmune diseases in a single patient is defined as MAS. While RA and Sjögren's syndrome commonly coexist due to their shared systemic autoimmune nature, myocarditis is not a frequent associated condition in these patients. Large-scale epidemiological studies do not provide specific incidence or prevalence data on this particular triad, reflecting its rarity.

In the management of acute myocarditis, guideline-based therapy includes supportive therapy for left ventricular dysfunction. The use of angiotensin-converting enzyme (ACE) inhibitors and beta-blockers not only mitigate heart failure but also provide significant anti-inflammatory effects. Myocardial biopsy can be useful for distinguishing between autoimmune myocarditis and viral myocarditis, but it is less informative when inflammatory infiltrates are present without identifiable pathogens.

In this case, the use of leucovorin (or folinic acid) at a dose of 100 mg/m2 every 6 hours for 24 hours was recommended to mitigate potential toxicity. MTX (methotrexate), a commonly used treatment for RA and Sjögren's syndrome, can contribute to cardiotoxicity, but this is mainly due to polypharmacy, overdosage, long pharmacotherapy with conventional disease-modifying antirheumatic drugs (DMARD), renal impairment, folate deficiency, and biologic agents such as Tumor Necrosis Factor (TNF)-α blockers and IL-6 receptor Inhibitors. Cardiac MRI is considered the test of choice for monitoring cardiotoxicity in patients undergoing long-term treatment with DMARDs.

This case highlights the importance of further research to better guide management strategies in such complex clinical scenarios. The rarity of the triad of myocarditis, RA, and Sjögren's syndrome necessitates a deeper understanding of its pathogenesis and treatment options to improve outcomes for affected patients.

1. Science continues to uncover unusual instances of Multiple Autoimmune Syndrome (MAS), as seen in a 46-year-old woman with concurrent rheumatoid arthritis (RA) and secondary Sjögren's syndrome (GSJ), accompanied by an unexpected myocarditis complication.
2. This medical-conditions triad – inflammatory heart condition, RA, and Sjögren's syndrome – is rarely documented in the medical literature.
3. The patient exhibited an exceptionally high troponin level of 1090 ng/ml, reflecting myocardial damage, and MRI-verified myocarditis, probably of autoimmune origin.
4. Myocarditis can develop secondary to autoimmune disease activity, but its occurrence in RA and Sjögren's syndromes is Infrequent.
5. In patients experiencing myocarditis, higher T1, T2, and extracellular volume fraction (ECV) values, coupled with a moderate prevalence of late gadolinium enhancement (LGE), have been observed.
6. To this day, the interstitial and granulomatous forms of myocarditis have been the most frequently described in the course of RA.
7. The specificity for the granulomatous form of myocarditis in RA is greater compared to other forms.
8. MAS, defined as the co-occurrence of at least three autoimmune diseases in a single patient, is not common in RA and Sjögren's syndrome patients due to their shared systemic nature.
9. Large-scale epidemiological studies still lack specific incidence or prevalence data on this unique MAS triad due to its rarity.
10. In the management of acute myocarditis, guidelines recommend supportive therapy for left ventricular dysfunction.
11. The use of angiotensin-converting enzyme (ACE) inhibitors and beta-blockers not only control heart failure symptoms but also offer significant anti-inflammatory effects.
12. A myocardial biopsy may help distinguish between autoimmune myocarditis and viral myocarditis, but it offers less insights in situations where inflammatory infiltrates are present without identifiable pathogens.
13. In this case, the recommendation of leucovorin (or folinic acid) at a dose of 100 mg/m2 every 6 hours for 24 hours aims to minimize potential toxicity.
14. MTX (methotrexate), a commonly prescribed RA and Sjögren's syndrome treatment, can contribute to cardiotoxicity.
15. Cardiac MRI is considered the primary test for monitoring cardiotoxicity in patients undergoing long-term treatment with disease-modifying antirheumatic drugs (DMARDs).
16. This case emphasizes the need for more research to develop better management strategies in complex clinical situations.
17. The uncommon nature of the myocarditis, RA, and Sjögren's syndrome triad requires further understanding of its pathogenesis and treatment alternatives to enhance patient outcomes.
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