Yamamoto Hiroyuki, Hashimoto Katsuya, Ikeda Yoshihiko, Isogai Jun, Hashimoto Toru
Department of Cardiovascular Medicine, Narita-Tomisato Tokushukai Hospital, Chiba, Japan.
Department of Pathology, National Cerebral and Cardiovascular Center, Suita, Japan.
Front Cardiovasc Med. 2022 Jul 7;9:913724. doi: 10.3389/fcvm.2022.913724. eCollection 2022.
Eosinophilic granulomatosis with polyangiitis (EGPA) is a systemic vasculitis involving small-to-medium-sized vessels characterized by asthma, vasculitis, and peripheral eosinophilia. EGPA-associated eosinophilic myocarditis (EM) occurs rarely, yet can be fatal if left untreated. Moreover, the accurate diagnosis of EGPA-associated EM without vasculitis is exceptionally difficult because of the overlapping features with EM of other causes. We report a case of probable EGPA with subclinical neurological involvement that presented with acute EM. The constellation of peripheral eosinophilia, left ventricular dysfunction, and normal epicardial coronary arteries raised suspicion of acute EM, which was confirmed by cardiac magnetic resonance (CMR) investigation and endomyocardial biopsy (EMB). Prompt systemic administration of corticosteroids completely restored and normalized myocardial structure and function. Although the patient's history suggested the presumed hypersensitivity myocarditis, EMB revealed EM without vasculitis, not hypersensitivity, leading to a tentative diagnosis of idiopathic hypereosinophilic syndrome. Interestingly, the characteristic findings of vasculitis on CMR imaging strongly suggested EGPA-associated EM. Although the patient had no clinical neurological manifestations, a nerve conduction study confirmed mononeuritis multiplex, leading to the final diagnosis of probable EGPA. Therefore, this case highlights the diagnostic challenge associated with EGPA and the diagnostic synergy of CMR and EMB for an exploratory diagnosis of EGPA-associated EM.
嗜酸性肉芽肿性多血管炎(EGPA)是一种累及中小血管的系统性血管炎,其特征为哮喘、血管炎和外周血嗜酸性粒细胞增多。EGPA相关的嗜酸性粒细胞性心肌炎(EM)很少见,但如果不治疗可能会致命。此外,由于与其他原因引起的EM有重叠特征,准确诊断无血管炎的EGPA相关EM异常困难。我们报告一例可能患有EGPA且有亚临床神经受累的病例,该病例表现为急性EM。外周血嗜酸性粒细胞增多、左心室功能障碍和心外膜冠状动脉正常的组合引起了对急性EM的怀疑,心脏磁共振(CMR)检查和心内膜心肌活检(EMB)证实了这一点。及时全身应用皮质类固醇使心肌结构和功能完全恢复并正常化。尽管患者病史提示可能为过敏性心肌炎,但EMB显示为无血管炎的EM,而非过敏性,从而初步诊断为特发性高嗜酸性粒细胞综合征。有趣的是,CMR成像上血管炎的特征性表现强烈提示EGPA相关EM。尽管患者没有临床神经表现,但神经传导研究证实为多灶性单神经炎,最终诊断为可能的EGPA。因此,该病例突出了与EGPA相关的诊断挑战以及CMR和EMB对EGPA相关EM进行探索性诊断的诊断协同作用。