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。

IF 0.4 Q4 SURGERY
Journal of Surgical Case Reports Pub Date : 2024-10-11 eCollection Date: 2024-10-01 DOI:10.1093/jscr/rjae644
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}
引用次数: 0

摘要

烷胨尿症(alkaptonuric ochronosis)的特征是同源戊二酸在结缔组织中沉积,通常与烷胨尿症有关,后者是一种罕见的遗传性疾病,由同源戊二酸 1,2-二氧化酶缺乏症引起。尽管赭石症与碱蛋白尿症有关,但它也可能发生在未经事先诊断的人身上。本病例报告讨论了一位患有严重主动脉瓣狭窄和冠状动脉疾病的 64 岁女性,她被发现主动脉瓣和二尖瓣以及主动脉根部内膜和乳头肌有chronotic色素沉着。该病例强调,在怀疑碱蛋白尿时,需要在瓣膜病的鉴别诊断中考虑chronosis。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
CiteScore
0.70
自引率
0.00%
发文量
559
审稿时长
11 weeks
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信