Department of Cardiovascular Surgery, Sasebo Central Hospital, 15 Yamato-cho, Sasebo, Nagasaki, 857-1195, Japan.
Gen Thorac Cardiovasc Surg. 2021 Mar;69(3):605-609. doi: 10.1007/s11748-020-01492-2. Epub 2020 Sep 24.
Alkaptonuria is an inherited metabolic disease caused by a genetic deficiency of homogentisate 1,2-dioxygenase and characterized by dark-brown connective tissue related to the deposition of oxidized homogentisic acid. Pigment deposition is also observed in the cardiovascular system, such as in the coronary arteries, cardiac valves, and aorta. Because aortic stenosis may develop secondary to pigment deposition-related calcification at the aortic valve, aortic valve replacement may be necessary for severe aortic valve disease. We report the case of a 75 year-old man with alkaptonuria-associated severe aortic stenosis who was successfully treated with minimally invasive endoscopic aortic valve replacement via right anterior minithoracotomy. The tricuspid aortic valve was severely calcified and both the valve and the aortic intima were ochronotic. No perioperative complications were observed and the postoperative course was uneventful.
尿黑酸尿症是一种遗传性代谢疾病,由 homogentisate 1,2-双加氧酶的遗传缺陷引起,其特征是与氧化 homogentisic 酸沉积有关的深褐色结缔组织。色素沉积也可见于心血管系统,如冠状动脉、心脏瓣膜和主动脉。由于主动脉瓣色素沉积相关钙化可导致主动脉瓣狭窄,因此对于严重的主动脉瓣疾病可能需要主动脉瓣置换。我们报告了一例 75 岁男性,患有尿黑酸尿症相关的严重主动脉瓣狭窄,成功地通过右前胸小切口微创内镜主动脉瓣置换术进行了治疗。三尖瓣主动脉瓣严重钙化,瓣叶和主动脉内膜均呈褐黄病样。未观察到围手术期并发症,术后过程顺利。