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.<Table Float="No" ID="Taba"> <tgroup cols="2"> <colspec align="left" colname="c1" colnum="1" /> <colspec align="left" colname="c2" colnum="2" /> <tbody> <row> <entry nameend="c2" namest="c1"> 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. </entry> </row> </tbody> </tgroup> </Table>

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

期刊论文
发表日期：

2025年4月16日

查看全文

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

摘要

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.