Robertsson Caroline, Sävendahl Lars, Cardemil Carina
Unit of Cranio- & Maxillofacial Surgery, Karolinska University Hospital, Stockholm, Sweden.
Unit of Pediatric Endocrinology, Karolinska University Hospital, Stockholm, Sweden.
Bone Rep. 2021 May 7;14:101089. doi: 10.1016/j.bonr.2021.101089. eCollection 2021 Jun.
Primary chronic osteomyelitis (PCO) of the jaw is a non-infectious, inflammatory state of the jawbone of unknown etiology. In recurrent periods, these patients often exhibit swelling of the cheek, impaired ability to open their mouth as well as pain. Available treatments today include anti-inflammatory or antiresorptive drugs, hyperbaric oxygen, surgical decortication or resection followed by reconstruction where none of them have been described to lead to restored anatomy and complete relief of symptoms. We here report the unexpected complete regression of all clinical symptoms of PCO within three months after initiating pubertal suppression therapy with a gonadotropin-releasing hormone analogue in a 9-year-old girl with PCO of the jaw and early onset of pubertal development. Radiology of the jawbone confirmed complete PCO remission when performed 18 months after starting the hormone suppression therapy. To our knowledge, total regression of PCO in such a short period of time has not been described earlier suggesting an effect of the anti-hormonal therapy . In this case report, we discuss possible underlying mechanisms and hypothesize that anti-hormonal treatment could be a potential effective treatment in patients with PCO of the jaw.
颌骨原发性慢性骨髓炎（PCO）是一种病因不明的颌骨非感染性炎症状态。在复发期，这些患者常出现脸颊肿胀、张口受限以及疼痛。目前可用的治疗方法包括抗炎或抗吸收药物、高压氧、外科去皮质术或切除术，随后进行重建，但均未被描述为能恢复解剖结构并完全缓解症状。我们在此报告，一名9岁患有颌骨PCO且青春期发育过早的女孩，在开始使用促性腺激素释放激素类似物进行青春期抑制治疗后的三个月内，PCO的所有临床症状意外地完全消退。在开始激素抑制治疗18个月后进行的颌骨放射学检查证实PCO已完全缓解。据我们所知，此前尚未有关于PCO在如此短时间内完全消退的报道，这表明抗激素治疗具有一定效果。在本病例报告中，我们讨论了可能的潜在机制，并推测抗激素治疗可能是颌骨PCO患者的一种潜在有效治疗方法。
