{"title":"一名克罗恩病患者同时患有法布里病和 IgA 肾病:病例报告。","authors":"Heejung Choi, Hyunee Yim, Inwhee Park","doi":"10.1111/nep.14419","DOIUrl":null,"url":null,"abstract":"<p><p>We present a unique case of concurrent Fabry disease (FD) and IgA nephropathy (IgAN) in a 27-year-old female with a 10-year history of Crohn's disease (CD). The patient presented to the nephrology clinic with microscopic haematuria and proteinuria on routine tests. A kidney biopsy revealed mesangial matrix widening, mesangial cell proliferation, and podocyte enlargement with prominent lacy and clear cytoplasm, as observed with haematoxylin and eosin staining. Immunofluorescence staining demonstrated diffuse immunoglobulin A deposits in the mesangium. Electron microscopy identified myelin-like figures in the cytoplasm of podocytes and electron-dense deposits in the mesangium, confirming IgAN and suggesting FD. Subsequent testing showed low alpha-galactosidase A (α-gal) enzyme activity in the patient's white blood cells, confirming the FD diagnosis. Enzyme replacement therapy was initiated following the diagnosis. To our knowledge, this is the first reported case of the coexistence of FD, IgAN, and CD in a single patient. This case highlights the importance of considering FD in patients with proteinuria, emphasising the need for comprehensive diagnostic evaluations in complex cases.</p>","PeriodicalId":19264,"journal":{"name":"Nephrology","volume":"30 1","pages":"e14419"},"PeriodicalIF":2.4000,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Concurrent Fabry disease and IgA nephropathy in a patient with Crohn's disease: A case report.\",\"authors\":\"Heejung Choi, Hyunee Yim, Inwhee Park\",\"doi\":\"10.1111/nep.14419\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>We present a unique case of concurrent Fabry disease (FD) and IgA nephropathy (IgAN) in a 27-year-old female with a 10-year history of Crohn's disease (CD). The patient presented to the nephrology clinic with microscopic haematuria and proteinuria on routine tests. A kidney biopsy revealed mesangial matrix widening, mesangial cell proliferation, and podocyte enlargement with prominent lacy and clear cytoplasm, as observed with haematoxylin and eosin staining. Immunofluorescence staining demonstrated diffuse immunoglobulin A deposits in the mesangium. Electron microscopy identified myelin-like figures in the cytoplasm of podocytes and electron-dense deposits in the mesangium, confirming IgAN and suggesting FD. Subsequent testing showed low alpha-galactosidase A (α-gal) enzyme activity in the patient's white blood cells, confirming the FD diagnosis. Enzyme replacement therapy was initiated following the diagnosis. To our knowledge, this is the first reported case of the coexistence of FD, IgAN, and CD in a single patient. This case highlights the importance of considering FD in patients with proteinuria, emphasising the need for comprehensive diagnostic evaluations in complex cases.</p>\",\"PeriodicalId\":19264,\"journal\":{\"name\":\"Nephrology\",\"volume\":\"30 1\",\"pages\":\"e14419\"},\"PeriodicalIF\":2.4000,\"publicationDate\":\"2025-01-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Nephrology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1111/nep.14419\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"UROLOGY & NEPHROLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Nephrology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1111/nep.14419","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"UROLOGY & NEPHROLOGY","Score":null,"Total":0}
引用次数: 0
摘要
我们报告了一例独特的法布里病(FD)和 IgA 肾病(IgAN)并发病例,患者是一名 27 岁女性,有 10 年克罗恩病(CD)病史。患者在常规检查中出现显微镜下血尿和蛋白尿,遂就诊于肾内科门诊。肾活检显示,系膜基质增宽,系膜细胞增生,荚膜细胞增大,血红素和伊红染色显示,荚膜细胞有突出的花边和清晰的胞浆。免疫荧光染色显示,免疫球蛋白 A 在间质中弥漫沉积。电镜检查在荚膜细胞的细胞质中发现了髓鞘样图形,在系膜中发现了电子致密沉积物,证实了 IgAN 的存在,并提示存在 FD。随后的检测显示,患者白细胞中的α-半乳糖苷酶A(α-gal)酶活性较低,确诊为FD。确诊后,患者开始接受酶替代治疗。据我们所知,这是首例在一名患者中同时存在 FD、IgAN 和 CD 的病例。该病例强调了在蛋白尿患者中考虑 FD 的重要性,并强调了对复杂病例进行全面诊断评估的必要性。
Concurrent Fabry disease and IgA nephropathy in a patient with Crohn's disease: A case report.
We present a unique case of concurrent Fabry disease (FD) and IgA nephropathy (IgAN) in a 27-year-old female with a 10-year history of Crohn's disease (CD). The patient presented to the nephrology clinic with microscopic haematuria and proteinuria on routine tests. A kidney biopsy revealed mesangial matrix widening, mesangial cell proliferation, and podocyte enlargement with prominent lacy and clear cytoplasm, as observed with haematoxylin and eosin staining. Immunofluorescence staining demonstrated diffuse immunoglobulin A deposits in the mesangium. Electron microscopy identified myelin-like figures in the cytoplasm of podocytes and electron-dense deposits in the mesangium, confirming IgAN and suggesting FD. Subsequent testing showed low alpha-galactosidase A (α-gal) enzyme activity in the patient's white blood cells, confirming the FD diagnosis. Enzyme replacement therapy was initiated following the diagnosis. To our knowledge, this is the first reported case of the coexistence of FD, IgAN, and CD in a single patient. This case highlights the importance of considering FD in patients with proteinuria, emphasising the need for comprehensive diagnostic evaluations in complex cases.
期刊介绍:
Nephrology is published eight times per year by the Asian Pacific Society of Nephrology. It has a special emphasis on the needs of Clinical Nephrologists and those in developing countries. The journal publishes reviews and papers of international interest describing original research concerned with clinical and experimental aspects of nephrology.