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长期使用左旋咪唑导致的伴有全身累及的特异性血管病:一例报告

Distinctive vasculopathy with systemic involvement due to levamisole long-term therapy: a case report.

作者信息

Aoun Bilal, Alali Mohammad, Degheili Jad A, Sanjad Sami, Vaquin Claudine, Donadieu Jean, Ulinski Tim, Termos Salah

机构信息

Division of Pediatric Nephrology, Department of Pediatrics, American University of Beirut, Beirut, Lebanon.

Hepatobiliary and Transplant Unit, Department of Surgery, Al-Amiri Hospital, Kuwait City, Kuwait.

出版信息

J Med Case Rep. 2018 Jul 16;12(1):209. doi: 10.1186/s13256-018-1728-6.

Abstract

BACKGROUND

Levamisole belongs to the antihelminthic class of drugs that are sometimes administered to patients with frequently relapsing or steroid-dependent nephrotic syndrome, owing to its steroid-sparing effects. Neutropenia and skin lesions, compatible with vasculitis, have been reported as drug complications, but they are rarely associated with any systemic involvement.

CASE PRESENTATION

We report a case of a 9-year-old Arab boy with steroid-dependent nephrotic syndrome who was treated with levamisole after his third relapse. The drug was initially well tolerated, but mild isolated neutropenia occurred 6 months after levamisole administration. This was followed by cutaneous vasculitis of both ears and the left cheek. The patient also developed hepatosplenomegaly and anemia. Levamisole was discontinued, and his disease remained in remission. All the systemic manifestations disappeared gradually over the course of 1 month. The patient remained in remission until 1 year after levamisole withdrawal, when clinical nephrosis recurred.

CONCLUSIONS

Despite levamisole's being a useful drug for maintaining remission in steroid-dependent nephrotic syndrome, patients on long-term levamisole therapy should be monitored closely to prevent serious complications that can easily be resolved by simple drug withdrawal.

摘要

背景

左旋咪唑属于抗蠕虫类药物,因其具有激素节省作用,有时会用于频繁复发或依赖类固醇的肾病综合征患者。嗜中性白血球减少症和与血管炎相符的皮肤病变已被报告为药物并发症,但它们很少伴有任何全身受累情况。

病例报告

我们报告一例9岁阿拉伯男孩,患有依赖类固醇的肾病综合征,在第三次复发后接受左旋咪唑治疗。该药物最初耐受性良好,但在服用左旋咪唑6个月后出现轻度单纯性嗜中性白血球减少症。随后双耳和左脸颊出现皮肤血管炎。患者还出现肝脾肿大和贫血。停用左旋咪唑后,其病情仍处于缓解期。所有全身表现于1个月内逐渐消失。该患者在停用左旋咪唑后1年内病情一直缓解,之后临床肾病复发。

结论

尽管左旋咪唑是用于维持依赖类固醇的肾病综合征缓解的有效药物,但长期接受左旋咪唑治疗的患者应密切监测,以预防严重并发症,这些并发症通过简单停药即可轻松解决。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/de20/6047121/f04908f4ceec/13256_2018_1728_Fig1_HTML.jpg

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