Lee Dae Hyun, Kasprowicz Thomas, Morales Alberto, Sotolongo Ignacio, Fernandez Joel, Korabathina Ravi
Cardiology, University of South Florida, Tampa, USA.
Cardiology, South Tampa Cardiology, Tampa, USA.
Cureus. 2021 Sep 12;13(9):e17914. doi: 10.7759/cureus.17914. eCollection 2021 Sep.
Primary hyperoxaluria is a rare genetic disorder characterized by oxalate crystal deposition, including in the heart. Hyperoxaluria-associated cardiomyopathy manifests as restrictive infiltrative cardiomyopathy. We present a case of a 52-year-old male with a past medical history of type 2 primary hyperoxaluria, end-stage renal disease on hemodialysis, paroxysmal atrial fibrillation, and hypertension, who presented with dyspnea and lethargy. Transthoracic echocardiogram showed cardiomyopathy with ejection fraction (EF) of 35-40% with severe hypokinesis of apical myocardium. Endomyocardial biopsy revealed interstitial fibrosis and crystal deposition consistent with oxalate. Cardiac MRI showed late gadolinium enhancement with subendocardial, nearly transmural fibrosis of lateral wall along with mid myocardial involvement of anterior and septal wall. To the best of our knowledge, this is the first case of type 2 primary hyperoxaluria-associated cardiomyopathy utilizing transthoracic echo, endomyocardial biopsy, and cardiac MRI.
原发性高草酸尿症是一种罕见的遗传性疾病,其特征为草酸盐晶体沉积,包括在心脏中沉积。高草酸尿症相关的心肌病表现为限制性浸润性心肌病。我们报告一例52岁男性病例,其既往有2型原发性高草酸尿症、终末期肾病且正在接受血液透析、阵发性心房颤动和高血压病史,此次因呼吸困难和嗜睡就诊。经胸超声心动图显示心肌病,射血分数(EF)为35%-40%,心尖心肌严重运动减弱。心内膜心肌活检显示间质纤维化和与草酸盐一致的晶体沉积。心脏磁共振成像显示钆延迟强化,侧壁心内膜下几乎透壁性纤维化,以及前壁和间隔壁心肌中层受累。据我们所知,这是首例利用经胸超声心动图、心内膜心肌活检和心脏磁共振成像诊断的2型原发性高草酸尿症相关心肌病病例。