Fulminant Giant Cell Myocarditis Following Vaccination Against Herpes Zoster

We report a 51-year-old woman with a history of breast cancer who developed dyspnea the day after receiving the recombinant zoster vaccine (Shingrix®) without preceding infection and with neither a history nor clinical signs of allergy. She developed progressive respiratory distress 1 week later. On admission, vital signs were stable, but troponin-I was markedly elevated. Electrocardiography showed a wide QRS complex (Figure A), chest radiography revealed cardiomegaly and pleural effusion (Figure B), echocardiography showed reduced ejection fraction (20%) (Figure C), but coronary angiography showed no stenosis (Figure D). Cardiac magnetic resonance imaging demonstrated late gadolinium enhancement and localized T2 hyperintensity, fulfilling the Lake Louise Criteria for myocarditis (Figure E). Endomyocardial biopsy revealed lymphocytic and eosinophilic infiltration with focal necrosis (Figure F,G). Multinucleated giant cells positive for CD68 were observed (Figure G,H), diagnostic of giant cell myocarditis.

Figure.

(A) Electrocardiography showing wide QRS complex, (B) cardiomegaly and pleural effusion on chest X-ray, (C) reduced ejection fraction (20%) on echocardiogram and (D) no stenosis on coronary angiogram. (E) Cardiac magnetic resonance imaging revealed late gadolinium enhancement and localized T2 hyperintensity. (F–H) Endomyocardial biopsy showing lymphocytic and eosinophilic infiltration with focal necrosis (F,G), and multinucleated giant cells positive for CD68 (H).

Despite dobutamine and intra-aortic balloon pump support, by hospital day 4, her condition progressed to treatment-refractory cardiogenic shock with recurrent creatine kinase elevation, hyperlactatemia, and worsening pulmonary congestion, necessitating venoarterial extracorporeal membrane oxygenation and corticosteroid pulse therapy. Cardiac function improved, and she was discharged on hospital day 54; however, she subsequently experienced out-of-hospital cardiac arrest due to ventricular tachycardia. This event occurred 85 days after the initial admission, despite ongoing oral prednisolone therapy (5 mg/day). On readmission, she received repeat steroid pulse therapy with tacrolimus, underwent implantable cardioverter-defibrillator implantation, and was discharged ambulatory.

Combination therapy with an additional immunosuppressive agent was not considered during the first hospitalization because of the marked eosinophilic infiltration observed on biopsy, but tacrolimus was initiated after the diagnosis of giant cell myocarditis was established on re-evaluation following recurrence.

The pathogenesis of giant cell myocarditis has been suggested to involve autoimmune mechanisms, with possible associations with myositis or muscle injury.¹ This case highlights a possible novel association between recombinant zoster vaccination and fulminant giant cell myocarditis, which, to our knowledge, has not previously been reported and warrants further investigation.

Reference

• 1.   Bang V, Ganatra S, Shah SP, Dani SS, Neilan TG, Thavendiranathan P, et al. Management of patients with giant cell myocarditis: JACC Review Topic of the Week. J Am Coll Cardiol 2021; 77: 1122–1134, doi:10.1016/j.jacc.2020.11.074.