Ouimet-Grennan Erika, Parent Marie-Claude, Tremblay-Gravel Maxime, Pelletier-Galarneau Matthieu, Boulet Jacinthe
Department of Medicine, Division of Cardiology, Montreal Heart Institute, University of Montreal, Montreal, Quebec, Canada.
Division of Nuclear Medicine, Department of Medicine, Montreal Heart Institute, University of Montreal, Montreal, Quebec, Canada.
JACC Case Rep. 2025 Aug 27;30(25):105078. doi: 10.1016/j.jaccas.2025.105078.
Pediatric inflammatory multisystem syndrome (PIMS-TS) is a rare entity observed in children with postinfectious hyperinflammatory syndrome. Cases have been reported, although rarely, after the mRNA SARS-CoV-2 (COVID-19) vaccine.
We present a case of a young man who developed PIMS-TS with fulminant myocarditis after a first dose of COVID-19 vaccination, with persistent evidence of inflammation despite multiple lines of therapy over the course of 2 years. He then experienced fulminant myocarditis that was resistant to anti-inflammatory therapies. Repeat endomyocardial biopsy was consistent with giant cell myocarditis.
Cases of giant cell myocarditis after COVID-19 vaccination have been reported in the past, however to our knowledge, none have been reported 2 years after a single dose and with intercurrent diagnosis of PIMS-TS myocarditis. This raises the question of a possible pathophysiologic association between the 2 diseases and immune dysregulation.
TAKE-HOME MESSAGE: This case illustrates the importance of repeating the investigation and questioning the diagnosis when the progression of the disease is not typical.
儿童炎症性多系统综合征(PIMS-TS)是一种在感染后高炎症综合征患儿中观察到的罕见病症。尽管很少见,但在接种mRNA新冠病毒(COVID-19)疫苗后也有病例报告。
我们报告一例年轻男性,在接种第一剂COVID-19疫苗后发生伴有暴发性心肌炎的PIMS-TS,尽管在2年病程中接受了多种治疗,但仍持续存在炎症迹象。随后他经历了对抗炎治疗耐药的暴发性心肌炎。重复心内膜心肌活检结果符合巨细胞心肌炎。
过去曾有接种COVID-19疫苗后发生巨细胞心肌炎的病例报告,但据我们所知,单剂接种2年后且并发PIMS-TS心肌炎诊断的病例尚无报告。这就引发了这两种疾病之间可能存在病理生理关联以及免疫失调的问题。
本病例说明了当疾病进展不典型时重复进行检查并质疑诊断的重要性。