{"title":"遗传性血管水肿,C1-INH正常,服用小剂量雌激素-黄体酮后发生复发性急腹症1例。","authors":"Tsuyoshi Nakayama, Yasuhiro Tamimoto, Yutaka Shimomura, Hiroshi Tsukamoto","doi":"10.1093/mrcr/rxad017","DOIUrl":null,"url":null,"abstract":"<p><p>Hereditary angio-oedema (HAE) is a rare genetic disease characterised by repeated episodes of temporary organ swelling. Three types of HAE are known, of which HAE with normal C1 inactivator is difficult to be diagnosed due to its lack of laboratory abnormalities. Here, we describe a case of HAE with normal C1 inactivator and recurrent acute abdomen following low-dose oestrogen-progestin therapy. Notably, genetic analysis by Sanger sequencing led to the identification of a recurrent heterozygous missense mutation c.988A > G (p.K330E) in the plasminogen (PLG) gene of the patient. Prophylactic tranexamic acid and on-demand selective bradykinin B2 receptor blockers are used to treat her symptoms.</p>","PeriodicalId":18677,"journal":{"name":"Modern Rheumatology Case Reports","volume":"7 2","pages":"491-494"},"PeriodicalIF":0.0000,"publicationDate":"2023-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"1","resultStr":"{\"title\":\"Hereditary angio-oedema with normal C1-INH, developing recurrent acute abdomen after taking low-dose oestrogen-progestin: A case report.\",\"authors\":\"Tsuyoshi Nakayama, Yasuhiro Tamimoto, Yutaka Shimomura, Hiroshi Tsukamoto\",\"doi\":\"10.1093/mrcr/rxad017\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Hereditary angio-oedema (HAE) is a rare genetic disease characterised by repeated episodes of temporary organ swelling. Three types of HAE are known, of which HAE with normal C1 inactivator is difficult to be diagnosed due to its lack of laboratory abnormalities. Here, we describe a case of HAE with normal C1 inactivator and recurrent acute abdomen following low-dose oestrogen-progestin therapy. Notably, genetic analysis by Sanger sequencing led to the identification of a recurrent heterozygous missense mutation c.988A > G (p.K330E) in the plasminogen (PLG) gene of the patient. Prophylactic tranexamic acid and on-demand selective bradykinin B2 receptor blockers are used to treat her symptoms.</p>\",\"PeriodicalId\":18677,\"journal\":{\"name\":\"Modern Rheumatology Case Reports\",\"volume\":\"7 2\",\"pages\":\"491-494\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2023-06-19\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"1\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Modern Rheumatology Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1093/mrcr/rxad017\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Modern Rheumatology Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1093/mrcr/rxad017","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 1
摘要
遗传性血管水肿(HAE)是一种罕见的遗传性疾病,以反复发作的暂时性器官肿胀为特征。已知有三种类型的HAE,其中C1失活因子正常的HAE由于缺乏实验室异常而难以诊断。在这里,我们描述了一例HAE患者,低剂量雌激素-黄体酮治疗后,C1失活因子正常,急性腹部复发。值得注意的是,Sanger测序的遗传分析导致在患者的纤溶酶原(PLG)基因中鉴定出复发性杂合错义突变c.988A > G (p.K330E)。预防性氨甲环酸和按需选择性缓激肽B2受体阻滞剂用于治疗她的症状。
Hereditary angio-oedema with normal C1-INH, developing recurrent acute abdomen after taking low-dose oestrogen-progestin: A case report.
Hereditary angio-oedema (HAE) is a rare genetic disease characterised by repeated episodes of temporary organ swelling. Three types of HAE are known, of which HAE with normal C1 inactivator is difficult to be diagnosed due to its lack of laboratory abnormalities. Here, we describe a case of HAE with normal C1 inactivator and recurrent acute abdomen following low-dose oestrogen-progestin therapy. Notably, genetic analysis by Sanger sequencing led to the identification of a recurrent heterozygous missense mutation c.988A > G (p.K330E) in the plasminogen (PLG) gene of the patient. Prophylactic tranexamic acid and on-demand selective bradykinin B2 receptor blockers are used to treat her symptoms.