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唑来膦酸盐治疗成年患者下颌骨弥漫性硬化性骨髓炎的潜力。

Potential of zoledronate for treating diffuse sclerosing osteomyelitis of the mandible in adult patients.

作者信息

Kusumoto Junya, Furudoi Shungo, Muraki Yumi, Warabi Moeka, Takeda Daisuke, Akashi Masaya

机构信息

Department of Oral and Maxillofacial Surgery, Kobe University Graduate School of Medicine, 7 - 5- 2, Kusunoki-Cho Chuo-Ku, Kobe, 650 - 0017, Japan.

Department of Oral Surgery, Konan Medical Center, Kobe, Japan.

出版信息

Clin Rheumatol. 2025 Jun;44(6):2463-2475. doi: 10.1007/s10067-025-07438-4. Epub 2025 Apr 16.

Abstract

INTRODUCTION

Diffuse sclerosing osteomyelitis (DSO) is a rare nonbacterial bone disease associated with recurrent pain and swelling, and its pathogenesis remains unknown. Despite the absence of an established treatment for DSO, bisphosphonates have recently been considered effective in managing this condition. However, the use of zoledronate is off-label, with limited reported cases. Therefore, this study aimed to investigate the effects of zoledronate on pain suppression in DSO.

METHODS

This single-arm retrospective study evaluated adult patients diagnosed with mandibular DSO and treated with zoledronate. Patient demographics, pain suppression effect, recurrence, number of zoledronate administration, adverse reactions to zoledronate, and imaging findings were investigated.

RESULTS

The study included 18 patients (median age of 59.5 years). Zoledronate effectively suppressed pain in all patients, with a median duration of effect onset of 1 day. Symptom recurrence was observed in 66.7% of patients, with a median time of 29 months from the first zoledronate administration to recurrence. Zoledronate was administered multiple times to 44.7% of patients. The median duration of response was 80 months for patients who experienced relief after a single administration of zoledronate compared to 32 months for those who received multiple administrations (p < 0.001). Adverse reactions, including flu-like symptoms, were observed in 72.2% of the patients, and no medication-related osteonecrosis of the jaw was observed during the follow-up period.

CONCLUSIONS

Zoledronate is a relatively safe and effective treatment option for DSO of the mandible. Key Points • Zoledronate effectively suppresses pain in mandibular diffuse sclerosing osteomyelitis. • All condylar lesions recurred after zoledronate administration. • Patients with mandibular canal enlargement required multiple doses of zoledronate. • Adverse reactions to zoledronate administration were minor.

摘要

引言

弥漫性硬化性骨髓炎(DSO)是一种罕见的非细菌性骨病,与反复疼痛和肿胀相关,其发病机制尚不清楚。尽管目前尚无针对DSO的确立治疗方法,但双膦酸盐类药物最近被认为对控制这种疾病有效。然而,唑来膦酸的使用属于超适应症用药,报道的病例有限。因此,本研究旨在探讨唑来膦酸对DSO疼痛抑制的效果。

方法

本单臂回顾性研究评估了诊断为下颌骨DSO并接受唑来膦酸治疗的成年患者。调查了患者的人口统计学特征、疼痛抑制效果、复发情况、唑来膦酸给药次数、对唑来膦酸的不良反应以及影像学表现。

结果

该研究纳入了18例患者(中位年龄59.5岁)。唑来膦酸有效抑制了所有患者的疼痛,中位起效时间为1天。66.7%的患者出现症状复发,从首次使用唑来膦酸给药到复发的中位时间为29个月。44.7%的患者接受了多次唑来膦酸给药。单次使用唑来膦酸后疼痛缓解的患者,中位反应持续时间为80个月,而接受多次给药的患者为32个月(p<0.001)。72.2%的患者出现了包括类流感症状在内的不良反应,随访期间未观察到与药物相关的颌骨坏死。

结论

唑来膦酸是下颌骨DSO相对安全有效的治疗选择。要点:•唑来膦酸有效抑制下颌弥漫性硬化性骨髓炎的疼痛。•唑来膦酸给药后所有髁突病变均复发。•下颌管扩大的患者需要多次使用唑来膦酸。•唑来膦酸给药的不良反应轻微。

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