病例报告:口服皮洛卡品治疗十例后天性特发性全身多汗症病例

Hanako Miyahara, Noriko Kubota, M. Okune, Yoshiyuki Ishii, N. Okiyama, T. Nomura, Junichi Furuta
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摘要

获得性特发性全身无汗症是一种罕见的疾病,其特征是全身无汗或少汗,且无其他系统性疾病。然而,其病因仍不清楚。获得性特发性全身多汗症的发病似乎与自身免疫机制有关。虽然类固醇脉冲疗法是最常用的疗法,但它缺乏高水平的证据。另一方面,皮洛卡平是一种毒蕈碱受体激动剂,可刺激外分泌腺,增加唾液和泪液分泌以及出汗。在此,我们报告我科类固醇脉冲疗法和口服皮洛卡品的治疗进展。2012 年至 2021 年间,我们共治疗了 10 例获得性特发性全身多汗症患者。所有患者均以口服皮洛卡品作为首次治疗,其中3人(30%)出汗增多。观察到轻微的副作用,但没有观察到严重的副作用。五名对口服皮洛卡平没有反应的患者随后接受了类固醇脉冲疗法,其中四名(80%)有明显反应。两名患者随后接受了口服皮洛卡平的治疗后,病情得到了长达 81 个月的缓解。在类固醇脉冲疗法之前,口服匹罗卡品可用于缓解后天性特发性全身性无汗症患者的症状,在类固醇脉冲疗法之后,口服匹罗卡品可作为维持疗法。
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Case report: Ten cases of acquired idiopathic generalized anhidrosis treated with oral pilocarpine
Acquired idiopathic generalized anhidrosis is a rare disease characterized by systemic anhidrosis or hypohidrosis without other systemic diseases. However, its etiology remains unclear. Autoimmune mechanisms seem to be involved in the development of acquired idiopathic generalized anhidrosis. Although steroid pulse therapy is the most commonly used therapy, it lacks a high level of evidence. On the other hand, pilocarpine, a muscarinic receptor agonist that stimulates exocrine glands, increases saliva and tear secretion as well as sweating. Here, we report treatment progresses of steroid pulse therapy and oral pilocarpine in our department. Between 2012 and 2021, we treated 10 patients of acquired idiopathic generalized anhidrosis. All patients were administered oral pilocarpine as the first therapy, three (30%) of whom had increased sweating. Minor side effects were observed, however, no serious side effects were observed. Five patients who did not respond to oral pilocarpine were subsequently treated with steroid pulse therapy to which four (80%) showed significant response. Two patients were subsequently administered oral pilocarpine as post-therapy, and remission was maintained for up to 81 months. Oral pilocarpine could be used to relieve symptoms in patients with acquired idiopathic generalized anhidrosis prior to steroid pulse therapy and as maintenance therapy after steroid pulse therapy.
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