Sumedh Jayanti, Jennifer Li, Jasveen Renthawa, Ming-Wei Lin, Vincent Lee
{"title":"孕妇抗pr3anca血管炎与抗gbm血管炎双阳性1例","authors":"Sumedh Jayanti, Jennifer Li, Jasveen Renthawa, Ming-Wei Lin, Vincent Lee","doi":"10.1111/nep.70124","DOIUrl":null,"url":null,"abstract":"<p><p>Pulmonary-renal syndrome (PRS) caused by double-positive ANCA-associated vasculitis and anti-GBM disease is rare, and management is primarily guided by case series evidence. We present an even rarer case of a 36-year-old female who developed PRS in early pregnancy due to double-positive disease. She required intensive care admission for respiratory support and was treated with high-dose steroids and two doses of rituximab (1 g), achieving a good pulmonary response. However, her renal function subsequently deteriorated. Given the high maternal and foetal risks associated with her condition, she chose to terminate her pregnancy at 8 weeks. A subsequent kidney biopsy revealed crescentic glomerulonephritis secondary to anti-GBM disease. She was treated with plasma exchange and cyclophosphamide, leading to normalisation of her kidney function. She was weaned off prednisone and completed a course of intravenous pulsed cyclophosphamide (500 mg ×6 fortnightly). At 1 year post-diagnosis, she remains in biochemical and clinical remission on maintenance rituximab every 6 months. This case highlights the complexity of managing double-positive disease in pregnancy, where evidence is limited, and decisions require careful consideration of maternal and foetal risks. Furthermore, it underscores the importance of early anti-GBM-specific treatment-plasma exchange and cyclophosphamide-in achieving remission.</p>","PeriodicalId":520716,"journal":{"name":"Nephrology (Carlton, Vic.)","volume":"30 9","pages":"e70124"},"PeriodicalIF":1.9000,"publicationDate":"2025-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Double Positive Anti-PR3 ANCA Vasculitis and Anti-GBM Vasculitis in a Pregnant Woman: Case Report.\",\"authors\":\"Sumedh Jayanti, Jennifer Li, Jasveen Renthawa, Ming-Wei Lin, Vincent Lee\",\"doi\":\"10.1111/nep.70124\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Pulmonary-renal syndrome (PRS) caused by double-positive ANCA-associated vasculitis and anti-GBM disease is rare, and management is primarily guided by case series evidence. We present an even rarer case of a 36-year-old female who developed PRS in early pregnancy due to double-positive disease. She required intensive care admission for respiratory support and was treated with high-dose steroids and two doses of rituximab (1 g), achieving a good pulmonary response. However, her renal function subsequently deteriorated. Given the high maternal and foetal risks associated with her condition, she chose to terminate her pregnancy at 8 weeks. A subsequent kidney biopsy revealed crescentic glomerulonephritis secondary to anti-GBM disease. She was treated with plasma exchange and cyclophosphamide, leading to normalisation of her kidney function. She was weaned off prednisone and completed a course of intravenous pulsed cyclophosphamide (500 mg ×6 fortnightly). At 1 year post-diagnosis, she remains in biochemical and clinical remission on maintenance rituximab every 6 months. This case highlights the complexity of managing double-positive disease in pregnancy, where evidence is limited, and decisions require careful consideration of maternal and foetal risks. Furthermore, it underscores the importance of early anti-GBM-specific treatment-plasma exchange and cyclophosphamide-in achieving remission.</p>\",\"PeriodicalId\":520716,\"journal\":{\"name\":\"Nephrology (Carlton, Vic.)\",\"volume\":\"30 9\",\"pages\":\"e70124\"},\"PeriodicalIF\":1.9000,\"publicationDate\":\"2025-09-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Nephrology (Carlton, Vic.)\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1111/nep.70124\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Nephrology (Carlton, Vic.)","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1111/nep.70124","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
Double Positive Anti-PR3 ANCA Vasculitis and Anti-GBM Vasculitis in a Pregnant Woman: Case Report.
Pulmonary-renal syndrome (PRS) caused by double-positive ANCA-associated vasculitis and anti-GBM disease is rare, and management is primarily guided by case series evidence. We present an even rarer case of a 36-year-old female who developed PRS in early pregnancy due to double-positive disease. She required intensive care admission for respiratory support and was treated with high-dose steroids and two doses of rituximab (1 g), achieving a good pulmonary response. However, her renal function subsequently deteriorated. Given the high maternal and foetal risks associated with her condition, she chose to terminate her pregnancy at 8 weeks. A subsequent kidney biopsy revealed crescentic glomerulonephritis secondary to anti-GBM disease. She was treated with plasma exchange and cyclophosphamide, leading to normalisation of her kidney function. She was weaned off prednisone and completed a course of intravenous pulsed cyclophosphamide (500 mg ×6 fortnightly). At 1 year post-diagnosis, she remains in biochemical and clinical remission on maintenance rituximab every 6 months. This case highlights the complexity of managing double-positive disease in pregnancy, where evidence is limited, and decisions require careful consideration of maternal and foetal risks. Furthermore, it underscores the importance of early anti-GBM-specific treatment-plasma exchange and cyclophosphamide-in achieving remission.