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1例急性狼疮性肾炎发作患者发生环磷酰胺诱发的后部可逆性脑病综合征的罕见病例。

A Rare Case of Cyclophosphamide-Induced Posterior Reversible Encephalopathy Syndrome in a Patient With Acute Lupus Nephritis Flare.

作者信息

Tabot Tabot Mpey K, Ababio Priscilla A, Waldron Shervonne, Rougui Lamiaa, Mehari Alem

机构信息

Internal Medicine, Howard University Hospital, Washington, D.C., USA.

Pulmonary Medicine, Howard University Hospital, Washington, D.C., USA.

出版信息

Cureus. 2023 Jan 30;15(1):e34372. doi: 10.7759/cureus.34372. eCollection 2023 Jan.

DOI:10.7759/cureus.34372
PMID:36874730
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9976264/
Abstract

Posterior reversible encephalopathy syndrome (PRES) is a syndrome encompassing both clinical and radiological manifestations with white matter vasogenic edema predominantly of the posterior and parietal lobes of the brain. It may accompany several medical conditions including immunosuppressive/cytotoxic drugs. We present a case of cyclophosphamide-induced PRES in a patient treated for acute lupus flare with biopsy-proven lupus nephritis. A 23-year-old African American female presented with non-specific symptoms over a six-month period on a medical background of systemic lupus erythematosus and biopsy-proven focal lupus nephritis class III on hydroxychloroquine, prednisone, and mycophenolate mofetil for which she was non-compliant. She was borderline hypertensive, tachycardic, saturating well on ambient air, and alert and oriented. Laboratory workup revealed electrolyte imbalance, elevated serum urea, creatinine, and B-type natriuretic peptide, low serum complements, and elevated double-stranded DNA (dsDNA) with negative lupus anticoagulant, anti-cardiolipin, and B2 glycoprotein antibody. Chest imaging revealed cardiomegaly with small pericardial effusion, left pleural effusion, and trace atelectasis, with no deep vein thrombosis on Doppler ultrasound. She was admitted to the intensive care unit for lupus flare with severe hyponatremia and was continued on mycophenolate mofetil, hydroxychloroquine, and prednisone 60 mg for induction therapy as well as intravenous fluids. Hyponatremia resolved, and blood pressure was controlled. She became fluid overloaded and anuric, with pulmonary edema and worsening hypoxic respiratory failure not responding to diuretic challenges. Daily hemodialysis was started, and she was intubated. Prednisone was tapered down, mycophenolate was switched to cyclophosphamide/mesna. She became agitated, restless, and confused, with waxing and waning consciousness and hallucinations. She was continued on bi-weekly cyclophosphamide for induction therapy. After the second dose of cyclophosphamide, her mentation worsened. Non-contrast MRI showed extensive bilateral cerebral and cerebella deep white matter high-intensity signals suggestive of PRES, which was new compared to one year prior. Cyclophosphamide was held and her mentation improved. She was successfully extubated and discharged to a rehabilitation center. The exact pathophysiological mechanism of PRES is not known. Endothelial damage and vasogenic edema have been hypothesized as possible mechanisms. Severe anemia, fluid overload, and renal failure are some of the causes of endothelial dysfunction and vasogenic edema with disruption of the blood-brain barrier, which were found in our patient, but repeated dosing of cyclophosphamide worsened her condition. Discontinuation of cyclophosphamide led to a significant improvement and complete reversal of her neurologic symptoms, implying that prompt recognition and management of PRES is vital to prevent permanent damage and even death in these patients.

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/87f7/9976264/1fd9257c1e07/cureus-0015-00000034372-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/87f7/9976264/9adbb6ed7ab7/cureus-0015-00000034372-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/87f7/9976264/1fd9257c1e07/cureus-0015-00000034372-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/87f7/9976264/9adbb6ed7ab7/cureus-0015-00000034372-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/87f7/9976264/1fd9257c1e07/cureus-0015-00000034372-i02.jpg
摘要

后部可逆性脑病综合征(PRES)是一种包含临床和影像学表现的综合征,主要表现为大脑后部和顶叶的白质血管源性水肿。它可能伴随多种疾病,包括免疫抑制/细胞毒性药物。我们报告一例环磷酰胺诱导的PRES病例,该患者因活检证实的狼疮性肾炎接受急性狼疮发作治疗。一名23岁的非裔美国女性,在系统性红斑狼疮和活检证实为III级局灶性狼疮性肾炎的病史背景下,在六个月内出现非特异性症状,正在服用羟氯喹、泼尼松和霉酚酸酯,但她未遵医嘱。她血压临界升高,心动过速,在室内空气中氧饱和度良好,神志清醒且定向力正常。实验室检查显示电解质失衡、血清尿素、肌酐和B型利钠肽升高、血清补体降低、双链DNA(dsDNA)升高,狼疮抗凝物、抗心磷脂和B2糖蛋白抗体阴性。胸部影像学检查显示心脏扩大伴少量心包积液、左侧胸腔积液和微量肺不张,多普勒超声检查未发现深静脉血栓。她因严重低钠血症导致狼疮发作而入住重症监护病房,继续服用霉酚酸酯、羟氯喹和60毫克泼尼松进行诱导治疗以及静脉补液。低钠血症得到缓解,血压得到控制。她出现液体超负荷和无尿,伴有肺水肿和缺氧性呼吸衰竭加重,对利尿剂治疗无反应。开始每日血液透析,并对她进行了插管。泼尼松逐渐减量,霉酚酸酯换成环磷酰胺/美司钠。她变得烦躁不安、意识模糊,意识状态时好时坏,并出现幻觉。继续每两周给予环磷酰胺进行诱导治疗。在第二次给予环磷酰胺后,她的精神状态恶化。非增强磁共振成像(MRI)显示双侧大脑和小脑深部白质广泛的高强度信号,提示PRES,与一年前相比是新出现的。停用环磷酰胺后她的精神状态有所改善。她成功拔管并转至康复中心。PRES的确切病理生理机制尚不清楚。内皮损伤和血管源性水肿被认为是可能的机制。严重贫血、液体超负荷和肾衰竭是内皮功能障碍和血管源性水肿的一些原因,伴有血脑屏障破坏(这些在我们的患者中都有发现),但重复给予环磷酰胺使她的病情恶化。停用环磷酰胺导致她的神经症状显著改善并完全逆转,这意味着及时识别和处理PRES对于预防这些患者的永久性损伤甚至死亡至关重要。

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A Rare Case of Cyclophosphamide-Induced Posterior Reversible Encephalopathy Syndrome in a Patient with Anti-GBM Vasculitis, and Review of Current Literature.1例抗肾小球基底膜血管炎患者发生环磷酰胺诱导的后部可逆性脑病综合征的罕见病例及当前文献综述
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