孕妇抗pr3anca血管炎与抗gbm血管炎双阳性1例

IF 1.9
Sumedh Jayanti, Jennifer Li, Jasveen Renthawa, Ming-Wei Lin, Vincent Lee
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引用次数: 0

摘要

由双阳性anca相关血管炎和抗gbm疾病引起的肺肾综合征(PRS)是罕见的,治疗主要以病例系列证据为指导。我们提出一个更罕见的情况下,一位36岁的女性谁发展PRS在妊娠早期由于双阳性疾病。她需要重症监护以获得呼吸支持,并接受大剂量类固醇和两剂利妥昔单抗(1g)治疗,取得了良好的肺反应。然而,她的肾功能随后恶化。考虑到与她的病情相关的母婴风险高,她选择在8周时终止妊娠。随后的肾活检显示继发于抗gbm疾病的新月形肾小球肾炎。她接受血浆置换和环磷酰胺治疗,使她的肾功能恢复正常。停用强的松并完成一个疗程的静脉脉冲环磷酰胺(每两周500 mg ×6)。在诊断后1年,患者每6个月维持一次利妥昔单抗,维持生化和临床缓解。该病例突出了妊娠期双阳性疾病管理的复杂性,证据有限,决策需要仔细考虑孕产妇和胎儿的风险。此外,它强调了早期抗gbm特异性治疗(血浆置换和环磷酰胺)在实现缓解中的重要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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.

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