Frankel Eitan S, Hajduczok Alexander G, Rajapreyar Indranee N, Brailovsky Yevgeniy
Division of Cardiology, Jefferson Heart Institute, Sidney Kimmel School of Medicine, Thomas Jefferson University Hospital, 925 Chestnut Street, Suite 200, Philadelphia, PA 19107, USA.
Eur Heart J Case Rep. 2022 Sep 5;6(9):ytac362. doi: 10.1093/ehjcr/ytac362. eCollection 2022 Sep.
Giant cell myocarditis (GCM) is a rare but well-known cause of fulminant myocarditis. Despite optimal medical therapy, many patients progress to orthotopic heart transplant (OHT). We present a case of recurrent GCM following OHT, including complex considerations in patient management and infectious sequelae.
A 33-year-old previously healthy male presented with 2 months of worsening shortness of breath. Transthoracic echocardiogram (TTE) demonstrated a left ventricular ejection fraction of 30-35%. After ruling out an ischaemic aetiology, he was discharged on guideline-directed medical therapy and later presented with productive cough, worsening dyspnoea on exertion, and diarrhoea. He was found to have elevated troponins and N-terminal pro-brain natriuretic peptide, lactic acidosis, progression of severe bi-ventricular dysfunction on TTE and right heart catheterization, and low cardiac index (1.0 L/min/m) requiring inotropes. He then required left ventricular assist device as a bridge to OHT. Pathology of the apical core diagnosed GCM as the cause of his fulminant heart failure. He eventually underwent heart transplantation, which was complicated by recurrent GCM. Treatment required intensification of his immunosuppressive regimen, which led to multiple infectious sequelae including norovirus, Shiga-like toxin producing , and disseminated nocardia of the lung and brain. As of the most recent follow-up, the patient is currently clinically stable.
Although recurrent GCM after OHT has been reported in the literature, the prognosis is not well understood and there are no clear guidelines regarding management. This case summarizes clinical considerations, treatment strategies, and adverse effects of recurrent GCM treatment.
巨细胞性心肌炎（GCM）是暴发性心肌炎的一种罕见但已知病因。尽管进行了最佳药物治疗，许多患者仍进展为原位心脏移植（OHT）。我们报告一例OHT后复发性GCM病例，包括患者管理中的复杂考量和感染后遗症。
一名33岁既往健康男性出现2个月的进行性气短。经胸超声心动图（TTE）显示左心室射血分数为30%-35%。排除缺血性病因后，他接受了指南指导的药物治疗并出院，但后来出现咳痰、劳力性呼吸困难加重和腹泻。发现他肌钙蛋白和N末端脑钠肽前体升高、乳酸酸中毒、TTE和右心导管检查显示严重双心室功能障碍进展，且心脏指数低（1.0 L/min/m）需要使用正性肌力药物。随后他需要左心室辅助装置作为OHT的过渡。心尖活检病理诊断GCM为其暴发性心力衰竭的病因。他最终接受了心脏移植，但出现了复发性GCM并发症。治疗需要强化免疫抑制方案，这导致了多种感染后遗症，包括诺如病毒、产志贺样毒素菌，以及肺部和脑部播散性诺卡菌感染。截至最近一次随访，患者目前临床稳定。
尽管文献中已报道OHT后复发性GCM，但预后尚不清楚，且关于管理尚无明确指南。本病例总结了复发性GCM治疗的临床考量、治疗策略及不良反应。
