Haruki Nobuhiko, Sumi Naoki, Kobara Satoshi, Tsujimoto Daiki, Iwasaki Yoichiro, Shimizu Takashi, Ishii Hiroshige, Inoue Yoshiaki, Soeda Takeshi, Saito Yuhei, Shirota Kinya
Division of Cardiology, Matsue Red Cross Hospital, Matsue, Japan.
Division of Cardiovascular Surgery, Matsue Red Cross Hospital, Matsue, Japan.
J Cardiol Cases. 2020 Jun 23;22(3):132-135. doi: 10.1016/j.jccase.2020.06.005. eCollection 2020 Sep.
Herein, we report a case of 61-year-old male with congestive heart failure (HF) due to severe aortic regurgitation (AR) caused by ruptured aortic valve aneurysm (VA). Aortic valve replacement (AVR) was performed after improvement of HF symptoms. Pathological examination of resected aortic VA showed neutrophil infiltration. Although he did not have typical clinical features associated with infective endocarditis (IE) such as fever, leukocytosis, or positive blood culture, we misdiagnosed this case as "concealed IE". However, we reconsidered the etiology because prosthetic aortic valve detachment occurred only one year after AVR. When considering causes except IE for perforated aortic VA and early prosthetic valve detachment, Behçet's disease (BD) was more likely based on the clinical course, echocardiography, and pathological findings in this case. The inflammatory process of BD is associated with aortic valvulitis/aortitis, leading to a possible cause of aortic regurgitation due to aortic VA or early prosthetic valve detachment. The diagnosis of BD was challenging in this case because he did not have predominant clinical findings, including recurrent oral ulcer which is a mandatory criterion for the diagnosis by the International Study Group, however, cardiac involvement may have been the initial presentation of BD. < Aortic valve aneurysm and its rupture are rare and most cases are caused by infective endocarditis (IE). However, if there is no typical clinical feature of IE including fever, leukocytosis, or positive blood culture in such case, Behçet's disease BD should be considered even if there are no predominant clinical findings including recurrent oral ulcer. Appropriate immunosuppressive therapy and modification of surgical techniques for the possible cardiac involvement may improve prognosis in patients with BD.>.
在此，我们报告一例61岁男性因主动脉瓣动脉瘤（VA）破裂导致严重主动脉瓣反流（AR）而发生充血性心力衰竭（HF）的病例。在HF症状改善后进行了主动脉瓣置换术（AVR）。对切除的主动脉VA进行病理检查显示有中性粒细胞浸润。尽管他没有感染性心内膜炎（IE）的典型临床特征，如发热、白细胞增多或血培养阳性，但我们将此病例误诊为“隐匿性IE”。然而，由于人工主动脉瓣脱离仅在AVR后一年发生，我们重新考虑了病因。在考虑除IE之外导致主动脉VA穿孔和人工瓣膜早期脱离的原因时，根据该病例的临床病程、超声心动图和病理结果，白塞病（BD）的可能性更大。BD的炎症过程与主动脉瓣炎/主动脉炎相关，导致因主动脉VA或人工瓣膜早期脱离而引起主动脉反流的可能原因。在该病例中，BD的诊断具有挑战性，因为他没有主要的临床发现，包括复发性口腔溃疡，而复发性口腔溃疡是国际研究小组诊断的必要标准，然而，心脏受累可能是BD的初始表现。<主动脉瓣动脉瘤及其破裂很少见，大多数病例由感染性心内膜炎（IE）引起。然而，如果在这种情况下没有IE的典型临床特征，包括发热、白细胞增多或血培养阳性，即使没有包括复发性口腔溃疡在内的主要临床发现，也应考虑白塞病BD。针对可能的心脏受累进行适当的免疫抑制治疗和手术技术改良可能会改善BD患者的预后。>
