A Case of Rapidly Progressing Granulomatous Myocarditis

Cao, Jessica; Schwartz, Yosef; Lomasney, Jon W.; Narang, Akhil; Okwuosa, Ike S.; Avery, Ryan; Passman, Rod; Cooper, Leslie T.; Vorovich, Esther

doi:10.1161/circheartfailure.120.007800

Cited by 3 publications

(4 citation statements)

References 4 publications

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“…Certain novel techniques, such as T 1 and T 2 mapping, assessment of myocardial strain, or hybrid CMR/FDG-PET imaging could not be used here. In theory, simultaneous PET could help as FDG uptake outside the heart would strongly favour CS, although, by some experts, 2 , 39 not fully excluding GCM. Importantly, due to its matched design, our work does not exclude differences on CMR between GCM and all-comers with CS.…”

Section: Discussionmentioning

confidence: 99%

Cardiac magnetic resonance in giant cell myocarditis: a matched comparison with cardiac sarcoidosis

Pöyhönen

Nordenswan

Lehtonen

et al. 2023

European Heart Journal - Cardiovascular Imaging

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Aims Giant cell myocarditis (GCM) is an inflammatory cardiomyopathy akin to cardiac sarcoidosis (CS). We decided to study the findings of GCM on cardiac magnetic resonance (CMR) imaging and to compare GCM with CS. Methods and results CMR studies of 18 GCM patients were analyzed and compared with 18 CS controls matched for age, sex, left ventricular (LV) ejection fraction and presenting cardiac manifestations. The analysts were blinded to clinical data. On admission, the duration of symptoms (median) was 0.2 months in GCM vs. 2.4 months in CS (P = 0.002), cardiac troponin T was elevated (>50 ng/L) in 16/17 patients with GCM and in 2/16 with CS (P < 0.001), their respective median plasma B-type natriuretic propeptides measuring 4488 ng/L and 1223 ng/L (P = 0.011). On CMR imaging, LV diastolic volume was smaller in GCM (177 ± 32 mL vs. 211 ± 58 mL, P = 0.014) without other volumetric or wall thickness measurements differing between the groups. Every GCM patient had multifocal late gadolinium enhancement (LGE) in a distribution indistinguishable from CS both longitudinally, circumferentially, and radially across the LV segments. LGE mass averaged 17.4 ± 6.3% of LV mass in GCM vs 25.0 ± 13.4% in CS (P = 0.037). Involvement of insertion points extending across the septum into the right ventricular wall, the “hook sign” of CS, was present in 53% of GCM and 50% of CS. Conclusion In GCM, CMR findings are qualitatively indistinguishable from CS despite myocardial inflammation being clinically more acute and injurious. When matched for LV dysfunction and presenting features, LV size and LGE mass are smaller in GCM.

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Section: Discussionmentioning

confidence: 99%

Cardiac magnetic resonance in giant cell myocarditis: a matched comparison with cardiac sarcoidosis

Pöyhönen

Nordenswan

Lehtonen

et al. 2023

European Heart Journal - Cardiovascular Imaging

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“…Ammirati et al 9 reported a case of an otherwise healthy 31-year-old male who required inotropy, MCS, and extracorporeal membrane oxygenation, who made a full recovery after receiving immunosuppression which included rATG. Cao et al 10 published a case initially thought to be cardiac sarcoidosis that was later re-diagnosed as GCM, who was successfully treated with rATG after heart transplantation. Suarez-Barrientos et al 12 published a case series involving six patients with GCM.…”

Section: Discussionmentioning

confidence: 99%

Two case reports of fulminant giant cell myocarditis treated with rabbit anti-thymocyte globulin

Bartz-Overman,

Li,

Puligandla

et al. 2024

European Heart Journal - Case Reports

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Background Giant cell myocarditis is an inflammatory form of acute heart failure with high rates of cardiac transplantation or death. Standard acute treatment includes multi-drug immunosuppressive regimens. There is a small but growing number of case reports utilizing rabbit anti-thymocyte globulin in severe cases. Case Summary Two cases are presented with similar presentations and clinical courses. Both are middle-aged patients with no significant past medical history, who presented with new acute decompensated heart failure that quickly progressed to cardiogenic shock requiring inotropic and mechanical circulatory support. Both underwent endomyocardial biopsies that diagnosed giant cell myocarditis. Both were treated with a multi-agent immunosuppressive regimen, notably including rabbit anti-thymocyte globulin, with subsequent resolution of shock and recovery of left ventricular ejection fraction. Both remain transplant-free and without ventricular arrythmias at 7 months and 26 months, respectively. Discussion In aggregate, these cases are typical of giant cell myocarditis. They add to growing observational data that upfront rATG may reduce morbidity and mortality in GCM, including potentially preventing the need for complex interventions like orthotopic heart transplantation.

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“…Myocyte damage and necrosis are always present. The presence of poorly formed granulomas is possible (36). Clearly formed granulomas exclude the diagnosis of GCM (35).…”

Section: Histopathological Findings Potential Etiological Factors And...mentioning

confidence: 99%

“…In case of cardiac sarcoidosis, the presence of granulomas and the absence of necrosis are important differential diagnostic criteria. Non-caseating granulomas may be composed of epithelioid cells that can occasionally fuse to form Langhans giant cells (36). However, Cooper LT Jr. and colleagues emphasize that the presence of granulomatous infiltrate in lymph nodes or other organs, or the presence of sarcoidosis, does not exclude the possibility of GCM coexistence (35).…”

Section: Histopathological Findings Potential Etiological Factors And...mentioning

confidence: 99%

Giant cell myocarditis in modern clinical practice

Lasica,

Đukanović,

Zdravković

et al. 2023

Medicinska istraživanja

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Giant cell myocarditis is a rare but extremely severe disease with a frequent need for heart transplantation and a high mortality rate. To this day, the most common reason for the occurrence of this disease has not been precisely determined, but its frequent association with autoimmune diseases speaks in favor of autoimmune etiology. Clinically, it is presented to the greatest extent by symptoms and signs of acute heart failure and ventricular rhythm disturbances, which are registered in as many as half of the patients. Arrhythmias can be accompanied by the occurrence of palpitations, repeated syncope, and even sudden cardiac death. A severe degree of acute heart failure in hemodynamically unstable patents who respond inadequately to the administered therapy for heart failure and refractory heart rhythm disorders and/or conduction disorders that are common in these patients should always raise suspicion of giant cell myocarditis. Given that changes in electrocardiogram, echocardiography and positive biomarker values are not strictly specific for this disease, the diagnosis of giant cell myocarditis is most often established by endomyocardial biopsy, especially in patients with the fulminant form of the disease. Timely endomyocardial biopsy enables not only quick and accurate diagnosis, but also early administration of immunosuppressive therapy, which greatly improves the outcome in these patients. Pathohistological verification of this disease is important so as to rule out cardiac sarcoidosis and other granulomatous infectious and non-infectious diseases. Given that in a large number of patients the disease has a fulminant course, early and rapid diagnosis, application of inotropic stimulation and mechanical circulatory support in hemodynamically unstable patients and adequate modern therapeutic regimen can largely reduce mortality in these patients, which is still high despite modern diagnostics and therapeutic progress of medicine.

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A Case of Rapidly Progressing Granulomatous Myocarditis

Cited by 3 publications

References 4 publications

Cardiac magnetic resonance in giant cell myocarditis: a matched comparison with cardiac sarcoidosis

Cardiac magnetic resonance in giant cell myocarditis: a matched comparison with cardiac sarcoidosis

Two case reports of fulminant giant cell myocarditis treated with rabbit anti-thymocyte globulin

Giant cell myocarditis in modern clinical practice

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