Rapid induction of clinical remission in SAPHO syndrome using high-dose Tripterygium glycosides A case report
Rapid induction of clinical remission in SAPHO syndrome using high-dose Tripterygium glycosides A case report
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DOI：
10.1097/md.0000000000021102
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发表时间：
2020-07-02
期刊：
影响因子：
1.6
通讯作者：
Li, Chen
中科院分区：
文献类型：
--
作者：
Gong, Liang;Wang, Lun;Li, Chen
原理：滑膜炎、痤疮、脓疱病、骨质增生和骨炎（SAPHO）综合征是一种罕见的疾病，没有标准治疗。雷公藤多甙（Tripterygium wilfordii hook f，TwHF）是一种具有抗炎作用的传统中药，在1例SAPHO患者中，1.0 mg/（kg center dot d）剂量的雷公藤多甙可显著改善SAPHO患者的疾病活动性。然而，TwHF的最佳剂量仍不清楚。在这里，我们报告了第一例SAPHO患者在接受1.5 mg/（kg中心点d）剂量的雷公藤多甙治疗后，临床症状迅速缓解。患者问题：一名67岁女性出现掌跖脓疱病，前胸壁和腰部疼痛。骨显像显示典型的示踪剂积聚特征，磁共振图像显示腰骶椎骨髓水肿。诊断：根据2012年提出的诊断标准，根据皮肤和骨关节表现以及骨关节造影的经典体征进行诊断。干预措施：雷公藤多甙起始剂量为1.5mg/（kg中心·d），连续1个月，以后每2周减量10 mg，直至1.0mg/（kg中心·d），长期维持。结果：临床表现快速缓解，磁共振成像异常明显改善。此外，未观察到明显的副作用。课程：雷公藤多甙1.5mg/（kg中心·d）剂量比1.0mg/（kg中心·d）剂量更能快速诱导缓解，且安全可靠。此外，雷公藤多苷还可能具有抑制骨溶解和增强骨强度的药理作用。
Rationale: Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare disease without standard treatments. Tripterygium wilfordii hook f (TwHF) is a traditional Chinese herb with anti-inflammatory effect, and 1.0 mg/(kg center dot d) dose of Tripterygium glycosides has been reported to significantly improve the disease activity of a SAPHO patient in a case report. However, the optimal dose of TwHF is still unclear. Here, we report the first case of SAPHO patient who achieved rapid remission in clinical symptoms after receiving 1.5 mg/(kg center dot d) dose of Tripterygium glycosides treatment. Patient concerns: A 67-year-old woman noted palmoplantar pustulosis and pain in the anterior chest wall and waist. Bone scintigraphy demonstrated the typical tracer accumulation feature and magnetic resonance images showed bone marrow edema in lumbosacral vertebra. Diagnoses: The diagnosis was made by dermatological and osteoarticular manifestations and classical signs in bone scintigraphy in accordance with the diagnostic criteria proposed in 2012. Interventions: Tripterygium glycosides was given with a primary dose of 1.5 mg/(kg center dot d) for 1 month and then reduced at a rate of 10 mg every 2 weeks until 1.0 mg/(kg center dot d) for a long-term maintenance. Outcomes: Fast-induced remission on clinical manifestations was achieved and magnetic resonance imaging abnormality was improved significantly. Additionally, no apparent side effects were observed. Lessons: 1.5 mg/(kg center dot d) dose of Tripterygium glycosides seems to have fast-induced remission than 1.0 mg/(kg center dot d) with reliable safety. Besides, Tripterygium glycosides may also have a pharmacological effect of inhibiting osteolysis and enhancing bone strength.