Alex Kamougeros, George Shiakos, Stelios Ioannou, Ioannis Tzanavaros, Zeyad Al-Jazrawi, Beatrice Ioannou
{"title":"一颗黑色的心:一例患有严重主动脉瓣狭窄和冠状动脉疾病的女性患者的主动脉瓣和二尖瓣碱蛋白尿性硬化症。","authors":"Alex Kamougeros, George Shiakos, Stelios Ioannou, Ioannis Tzanavaros, Zeyad Al-Jazrawi, Beatrice Ioannou","doi":"10.1093/jscr/rjae644","DOIUrl":null,"url":null,"abstract":"<p><p>Alkaptonuric ochronosis, characterized by the deposition of homogentisic acid in connective tissues, is commonly linked with alkaptonuria, a rare genetic disorder resulting from homogentisate 1,2-dioxygenase deficiency. Despite its association with alkaptonuria, ochronosis can occur in individuals without a prior diagnosis. This case report discusses a 64-year-old female with severe aortic valve stenosis and coronary artery disease who was found to have ochronotic pigmentation in the aortic and mitral valves, as well as in the aortic root intima and papillary muscles. This case emphasizes the need to consider ochronosis in the differential diagnosis of valvular disease when alkaptonuria is suspected.</p>","PeriodicalId":47321,"journal":{"name":"Journal of Surgical Case Reports","volume":"2024 10","pages":"rjae644"},"PeriodicalIF":0.4000,"publicationDate":"2024-10-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11469645/pdf/","citationCount":"0","resultStr":"{\"title\":\"A heart so black: a case of alkaptonuric ochronosis of the aortic and mitral valves in a female patient with severe aortic valve stenosis and coronary artery disease.\",\"authors\":\"Alex Kamougeros, George Shiakos, Stelios Ioannou, Ioannis Tzanavaros, Zeyad Al-Jazrawi, Beatrice Ioannou\",\"doi\":\"10.1093/jscr/rjae644\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Alkaptonuric ochronosis, characterized by the deposition of homogentisic acid in connective tissues, is commonly linked with alkaptonuria, a rare genetic disorder resulting from homogentisate 1,2-dioxygenase deficiency. Despite its association with alkaptonuria, ochronosis can occur in individuals without a prior diagnosis. This case report discusses a 64-year-old female with severe aortic valve stenosis and coronary artery disease who was found to have ochronotic pigmentation in the aortic and mitral valves, as well as in the aortic root intima and papillary muscles. This case emphasizes the need to consider ochronosis in the differential diagnosis of valvular disease when alkaptonuria is suspected.</p>\",\"PeriodicalId\":47321,\"journal\":{\"name\":\"Journal of Surgical Case Reports\",\"volume\":\"2024 10\",\"pages\":\"rjae644\"},\"PeriodicalIF\":0.4000,\"publicationDate\":\"2024-10-11\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11469645/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of Surgical Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1093/jscr/rjae644\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/10/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q4\",\"JCRName\":\"SURGERY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Surgical Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1093/jscr/rjae644","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/10/1 0:00:00","PubModel":"eCollection","JCR":"Q4","JCRName":"SURGERY","Score":null,"Total":0}
A heart so black: a case of alkaptonuric ochronosis of the aortic and mitral valves in a female patient with severe aortic valve stenosis and coronary artery disease.
Alkaptonuric ochronosis, characterized by the deposition of homogentisic acid in connective tissues, is commonly linked with alkaptonuria, a rare genetic disorder resulting from homogentisate 1,2-dioxygenase deficiency. Despite its association with alkaptonuria, ochronosis can occur in individuals without a prior diagnosis. This case report discusses a 64-year-old female with severe aortic valve stenosis and coronary artery disease who was found to have ochronotic pigmentation in the aortic and mitral valves, as well as in the aortic root intima and papillary muscles. This case emphasizes the need to consider ochronosis in the differential diagnosis of valvular disease when alkaptonuria is suspected.