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环孢素 A 诱导的膜性肾病患者的鼓室过度增生伴鼻塞。

Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy.

机构信息

Department of Medicine I - Nephrology, Transplantation & Medical Intensive Care, University Witten/Herdecke, Medical Center Cologne-Merheim, Cologne, Germany.

Department of Internal Medicine and Nephrology, University Witten/Herdecke, Dortmund, DE, Germany.

出版信息

Am J Case Rep. 2022 May 21;23:e935362. doi: 10.12659/AJCR.935362.

Abstract

BACKGROUND The immunomodulatory and pharmacokinetic effects of cyclosporine A are used to treat diverse disease entities in different medical fields, including organ transplantation and/or autoimmune diseases. It is also applied in patients with nephrotic range proteinuria as an adjunct to steroids and supportive antihypertensive/antiproteinuric medications. Cyclosporine has a small therapeutic window and is dosed with respect to the underlying disease entity and severity via trough level adaptations. Among its most frequent adverse effects are hypertension, nephrotoxicity, neurotoxicity, and electrolyte disturbances. Hypertrichosis and gingival hyperplasia are obvious and widely recognized adverse effects. CASE REPORT We report on a 66-year-old woman who was treated with cyclosporine A for primary membranous nephropathy. During treatment with cyclosporine, she developed hirsutism and gingival hyperplasia. Later, she reported having impaired nasal breathing and dyspnea on mild physical exercise. Clinical, rhinoscopic, and radiological evaluations showed marked conchal hyperplasia as a potential cause of her symptoms. An extensive medical work-up did not show evidence of allergic, immunologic, or other drug adverse effects, suggesting cyclosporine-induced hyperplasia of the turbinates as a hypothetical causative factor. Dose reductions did not lead to resolution of symptoms but resulted in increasing proteinuria. Therefore, cyclosporine was stopped, and the patient was treated with rituximab. Thereafter, hirsutism and gingival and conchal hyperplasia gradually regressed over 2-4 months, showing complete resolution of conchal hyperplasia on computed-tomography follow-up after 6 months. CONCLUSIONS Cyclosporine can not only result in gingival hyperplasia but also in hyperplasia of the turbinates leading to impaired nasal breathing and shortness of breath on exertion. An extensive search for many other known causes of conchal swelling is warranted to finally suggest an adverse effect of cyclosporine. Discontinuation of cyclosporine resulted in complete remission of conchal hyperplasia as well as other adverse effects.

摘要

背景

环孢素 A 的免疫调节和药代动力学作用被用于治疗不同医学领域的多种疾病实体,包括器官移植和/或自身免疫性疾病。它也被应用于肾病范围蛋白尿的患者中,作为类固醇和支持性降压/抗蛋白尿药物的辅助治疗。环孢素的治疗窗较小,根据基础疾病实体和严重程度通过谷浓度调整剂量。其最常见的不良反应包括高血压、肾毒性、神经毒性和电解质紊乱。多毛症和牙龈增生是明显且广泛公认的不良反应。

病例报告

我们报告了一位 66 岁女性,因原发性膜性肾病接受环孢素 A 治疗。在环孢素治疗期间,她出现了多毛症和牙龈增生。后来,她报告说在轻度体力活动时出现鼻塞和呼吸困难。临床、鼻镜和影像学评估显示鼻甲明显增生,可能是她症状的原因。广泛的医学检查未显示出过敏、免疫或其他药物不良反应的证据,提示环孢素引起的鼻甲增生是一个假设的致病因素。剂量减少并未导致症状缓解,反而导致蛋白尿增加。因此,停止使用环孢素,并使用利妥昔单抗治疗。此后,多毛症、牙龈和鼻甲增生逐渐消退,6 个月后 CT 随访显示鼻甲完全消退。

结论

环孢素不仅可导致牙龈增生,还可导致鼻甲增生,导致鼻塞和运动时呼吸困难。应广泛寻找许多其他已知的鼻甲肿胀原因,最终提示环孢素的不良反应。停用环孢素可导致鼻甲增生和其他不良反应完全缓解。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e34/9131439/960583d3147b/amjcaserep-23-e935362-g001.jpg

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