Abstract：
BACKGROUND: The role of bisphosphonates (BP) in hypertrophic osteoarthropathy (HPOA) is unclear. We presented a case of primary HPOA and performed a systematic review of literature on the effect of BP on treatment response in primary and secondary HPOA.
METHODS: The study was prospectively registered in PROSPERO (CRD42022343786). We performed a PubMed literature search that restricted to the English language. We included patients diagnosed with primary or secondary HPOA who received BP. The primary endpoint assessed was the effectiveness of BP on response to pain or arthritis. Secondary outcomes included timing, degree, and duration of response, comparison to other HPOA therapies, impact of BP on radiology, bone scan, bone turnover markers, and adverse effects of BP.
RESULTS: Literature search retrieved only case reports. Forty-five patients (21 primary, 24 secondary HPOA) had received BP. Majority(88.3%) experienced improvement in pain or arthritis. Response was gradual for primary HPOA and within a median of 3 to 7 days for secondary HPOA after treatment with BP. Most patients had reduced bone scan uptake after BP. When other HPOA therapies were tried, half responded to BP after not having previously responded to other therapies, while a third received the treatments concurrently, making it difficult to attribute treatment response to a drug. Reporting of other secondary outcomes was very heterogenous and qualitative to draw conclusions. No major adverse effects have been reported for BP in HPOA.
CONCLUSIONS: Bisphosphonates provide an effective and safe treatment option for primary and secondary HPOA. However, there is a lack of randomized controlled trials.
METHODS: The study was prospectively registered in PROSPERO (CRD42022343786). We performed a PubMed literature search that restricted to the English language. We included patients diagnosed with primary or secondary HPOA who received BP. The primary endpoint assessed was the effectiveness of BP on response to pain or arthritis. Secondary outcomes included timing, degree, and duration of response, comparison to other HPOA therapies, impact of BP on radiology, bone scan, bone turnover markers, and adverse effects of BP.
RESULTS: Literature search retrieved only case reports. Forty-five patients (21 primary, 24 secondary HPOA) had received BP. Majority(88.3%) experienced improvement in pain or arthritis. Response was gradual for primary HPOA and within a median of 3 to 7 days for secondary HPOA after treatment with BP. Most patients had reduced bone scan uptake after BP. When other HPOA therapies were tried, half responded to BP after not having previously responded to other therapies, while a third received the treatments concurrently, making it difficult to attribute treatment response to a drug. Reporting of other secondary outcomes was very heterogenous and qualitative to draw conclusions. No major adverse effects have been reported for BP in HPOA.
CONCLUSIONS: Bisphosphonates provide an effective and safe treatment option for primary and secondary HPOA. However, there is a lack of randomized controlled trials.
摘要：
背景：二膦酸盐（BP）在肥厚性骨关节病（HPOA）中的作用尚不清楚。我们介绍了一例原发性HPOA，并对有关BP对原发性和继发性HPOA治疗反应影响的文献进行了系统回顾。
方法：该研究在PROSPERO（CRD42022343786）中进行了前瞻性注册。我们进行了PubMed文献检索，仅限于英语。我们纳入了接受BP的诊断为原发性或继发性HPOA的患者。评估的主要终点是BP对疼痛或关节炎反应的有效性。次要结果包括时机，学位,和响应的持续时间，与其他HPOA疗法相比，BP对放射学的影响，骨扫描,骨转换标记,和BP的不利影响。
结果：文献检索仅检索病例报告。45名患者(21名原发性，24例继发性HPOA）已接受BP。大多数（88.3％）经历了疼痛或关节炎的改善。在用BP治疗后，原发性HPOA的反应是逐渐的，而继发性HPOA的反应在3至7天的中位数内。BP后，大多数患者的骨扫描摄取减少。当尝试其他HPOA疗法时，一半的人在以前对其他疗法没有反应后对血压有反应，三分之一的人同时接受治疗，很难将治疗反应归因于药物。其他次要结果的报告是非常异质和定性的，无法得出结论。没有关于HPOA中BP的主要不良反应的报道。
结论：双膦酸盐为原发性和继发性HPOA提供了有效和安全的治疗选择。然而,缺乏随机对照试验.
方法：该研究在PROSPERO（CRD42022343786）中进行了前瞻性注册。我们进行了PubMed文献检索，仅限于英语。我们纳入了接受BP的诊断为原发性或继发性HPOA的患者。评估的主要终点是BP对疼痛或关节炎反应的有效性。次要结果包括时机，学位,和响应的持续时间，与其他HPOA疗法相比，BP对放射学的影响，骨扫描,骨转换标记,和BP的不利影响。
结果：文献检索仅检索病例报告。45名患者(21名原发性，24例继发性HPOA）已接受BP。大多数（88.3％）经历了疼痛或关节炎的改善。在用BP治疗后，原发性HPOA的反应是逐渐的，而继发性HPOA的反应在3至7天的中位数内。BP后，大多数患者的骨扫描摄取减少。当尝试其他HPOA疗法时，一半的人在以前对其他疗法没有反应后对血压有反应，三分之一的人同时接受治疗，很难将治疗反应归因于药物。其他次要结果的报告是非常异质和定性的，无法得出结论。没有关于HPOA中BP的主要不良反应的报道。
结论：双膦酸盐为原发性和继发性HPOA提供了有效和安全的治疗选择。然而,缺乏随机对照试验.
