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.
Conclusion
Bisphosphonates provide an effective and safe treatment option for primary and secondary HPOA. However, there is a lack of randomized controlled trials.
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Data availability
No datasets were generated or analysed during the current study.
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All authors contributed to the conception and design of the study. RP conducted the literature search. RP and KS performed the data extraction, qualitative assessment of the studies and the data analysis. KS, VS and RP wrote the manuscript. JS, SK, DN, DH, HD and HR critically reviewed the manuscript. All authors approved the final version of the manuscript.
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Palui, R., Sridharan, K., Sahoo, J. et al. Role of bisphosphonates in hypertrophic osteoarthropathy: a systematic review. Endocrine (2024). https://doi.org/10.1007/s12020-024-03804-5
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DOI: https://doi.org/10.1007/s12020-024-03804-5