一名患有单克隆丙种球蛋白病和硬皮样综合征的患者双击氢氯喹和胺碘酮诱发肾磷脂病:病例报告和文献综述

J. C. De la Flor, Pablo Rodríguez-Doyágüez, Daniel Villa, Rocío Zamora, Francisco Díaz
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摘要

磷脂病是一种罕见的疾病,主要表现为细胞内磷脂过度堆积,用电子显微镜观察会出现斑马体或片状体。这种疾病与某些遗传疾病有关,或继发于药物或毒素。药物性磷脂病包括多种类型的药物,最常见的是氯喹、胺碘酮或环丙沙星。从临床和组织学角度看,肾脏受累的情况千变万化,只有在电子显微镜下看到斑马体后才能确诊。这些发现可能需要进行基因检测,以确定是否为法布里病,因为其组织学结果是无法区分的。大多数致病化学物质都是阳离子两亲药物,人们对斑马体的形成及其致病意义提出了多种机制假设。然而,药物毒性与磷脂蓄积、斑马体和器官功能障碍之间的关系以及停药对肾脏造成的后果仍然是个谜。据我们所知,这是第一例急性肾损伤病例报告,患者伴有肾脏单克隆丙种球蛋白病、病变和硬皮样综合征,斑马体与开始使用羟氯喹和胺碘酮治疗有关,是药物诱发磷脂病的一个实例。
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Double Hit of Hydroxichloroquine and Amiodarone Induced Renal Phospholipidosis in a Patient with Monoclonal Gammopathy and Sclerodermiform Syndrome: A Case Report and Review of the Literature
Phospholipidosis is a rare disorder which consists of an excessive intracellular accumulation of phospholipids and the appearance of zebra bodies or lamellar bodies when looking at them using electron microscopy. This disease is associated with certain genetic diseases or is secondary to drugs or toxins. Drug-induced phospholipidosis encompasses many types of pharmaceuticals, most notably chloroquine, amiodarone or ciprofloxacin. Clinically and histologically, renal involvement can be highly variable, with the diagnosis not being made until the zebra bodies are seen under an electron microscope. These findings may require genetic testing to discount Fabry disease, as its histological findings are indistinguishable. Most of the chemicals responsible are cationic amphiphilic drugs, and several mechanisms have been hypothesized for the formation of zebra bodies and their pathogenic significance. However, the relationship between drug toxicity and phospholipid accumulation, zebra bodies and organ dysfunction remains enigmatic, as do the renal consequences of drug withdrawal. We present, to our knowledge, the first case report of acute renal injury with a monoclonal gammopathy of renal significance, lesions, and sclerodermiform syndrome, with zebra bodies that were associated with the initiation of a hydroxychloroquine and amiodarone treatment, as an example of drug-induced-phospholipidosis.
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