Niimi Teruyuki, Miwa Ryosuke, Kitagawa Ken, Natsume Nagato, Sakamoto Yuki
Division of Research and Treatment for Oral and Maxillofacial Congenital Anomalies, School of Dentistry, Aichi Gakuin University, Nagoya, JPN.
Oral Surgery, Kansai Medical University Medical Center, Moriguchi, JPN.
Cureus. 2025 Jul 29;17(7):e88963. doi: 10.7759/cureus.88963. eCollection 2025 Jul.
Diffuse sclerosing osteomyelitis (DSO) is a rare, chronic aseptic osteomyelitis of the mandible characterized by resistance to conventional therapies including nonsteroidal anti-inflammatory drugs (NSAIDs), antibiotics, corticosteroids, and surgery. Diagnosis is often delayed due to nonspecific symptoms and lack of typical infectious signs, sometimes resulting in misdiagnosis. We report a case of a 58-year-old male patient with persistent mandibular pain initially misdiagnosed as trigeminal neuralgia and treated with microvascular decompression surgery without improvement. Subsequent imaging revealed bone sclerosis and resorption consistent with DSO. IV administration of zoledronic acid resulted in rapid and sustained pain relief, improved mouth opening, and radiographic bone regeneration without adverse events. A review of 139 reported cases treated with antiresorptive agents (ARAs) including bisphosphonates (BPs) and denosumab (DMB) demonstrated a high remission rate (95%), though treatment protocols varied widely. BPs appear preferable to DMB due to longer duration of effect and lower risk of medication-related osteonecrosis of the jaw (MRONJ). This case and literature review underscore the efficacy and safety of ARAs in managing DSO but highlight the need for standardized treatment guidelines and further prospective studies to optimize therapy.
弥漫性硬化性骨髓炎(DSO)是一种罕见的下颌骨慢性无菌性骨髓炎,其特点是对包括非甾体类抗炎药(NSAIDs)、抗生素、皮质类固醇和手术在内的传统治疗方法具有抗性。由于症状不具特异性且缺乏典型的感染体征,诊断往往会延迟,有时会导致误诊。我们报告一例58岁男性患者,其下颌骨持续疼痛最初被误诊为三叉神经痛,并接受了微血管减压手术,但病情并无改善。随后的影像学检查显示骨硬化和吸收,符合弥漫性硬化性骨髓炎的表现。静脉注射唑来膦酸导致疼痛迅速且持续缓解,张口度改善,影像学显示骨再生,且无不良事件发生。对139例使用抗吸收剂(ARAs)治疗的病例进行回顾,这些抗吸收剂包括双膦酸盐(BPs)和地诺单抗(DMB),结果显示缓解率较高(95%),尽管治疗方案差异很大。由于作用持续时间更长且颌骨药物相关性骨坏死(MRONJ)风险较低,双膦酸盐似乎比地诺单抗更可取。该病例及文献回顾强调了抗吸收剂在治疗弥漫性硬化性骨髓炎方面的有效性和安全性,但也突出了需要标准化治疗指南以及进一步的前瞻性研究以优化治疗。