Ezine E, Pedailles S, Dompmartin A, Morice C
Centre hospitalier universitaire de Caen, Côte-de-Nacre, avenue de la Côte-de-Nacre, 14000 Caen, France.
Centre hospitalier intercommunal Alençon-Mamers, 25, rue de Fresnay, 61000 Alençon, France.
Ann Dermatol Venereol. 2020 Sep;147(8-9):535-541. doi: 10.1016/j.annder.2020.04.021. Epub 2020 Jul 8.
Retinoids are widely used in dermatology. Adverse effects are frequent and require clinical and laboratory monitoring. Herein we report the case of a patient with secondary capillary leak syndrome (SCLS) associated with acitretin. We then present a review of the literature on systemic retinoids and SFCS.
A 57-year-old patient consulted following the onset of severe type I pityriasis rubra pilaris. Treatment was initiated comprising topical corticosteroids combined with acitretin at a dose of 0.5mg/kg/day. On the eighth day, voluminous edema appeared, accompanied by weight gain of 8kg in 48h and hypotension. The laboratory assessment showed hypoalbuminemia and hemoconcentration. Acitretin-induced SCLS was diagnosed based on the triple signs of hemoconcentration, hypoalbuminemia and hypotension, as well as rapid improvement following discontinuation of acitretin.
We collected 7 published clinical cases between 1981 and 2018, including our own case report. Retinoids were indicated only in severe cutaneous diseases. The mean time to onset of SLCS is 9.8 days, with a return to normal 17 days after discontinuation of retinoids. Capillary leak syndrome is a rare and under-diagnosed clinical-laboratory syndrome that must be recognized in order to avoid potentially fatal inappropriate management. It is a rare adverse effect of retinoids used in dermatology and the pathophysiology remains unclear.
维甲酸类药物在皮肤科广泛应用。其不良反应常见，需要临床和实验室监测。在此，我们报告一例与阿维A相关的继发性毛细血管渗漏综合征（SCLS）患者。然后，我们对有关全身性维甲酸类药物和SCLS的文献进行综述。
一名57岁患者因严重的I型毛发红糠疹前来就诊。开始采用局部皮质类固醇联合阿维A治疗，剂量为0.5mg/kg/天。在第8天，出现大量水肿，48小时内体重增加8kg，并伴有低血压。实验室检查显示低白蛋白血症和血液浓缩。根据血液浓缩、低白蛋白血症和低血压三联征，以及停用阿维A后迅速好转，诊断为阿维A诱发的SCLS。
我们收集了1981年至2018年间发表的7例临床病例，包括我们自己的病例报告。维甲酸类药物仅用于严重的皮肤病。SLCS的平均发病时间为9.8天，停用维甲酸类药物后17天恢复正常。毛细血管渗漏综合征是一种罕见且诊断不足的临床-实验室综合征，必须予以识别，以避免潜在的致命性不适当处理。它是皮肤科使用维甲酸类药物罕见的不良反应，其病理生理学仍不清楚。
