Primary chronic osteomyelitis of the jaw: Rapid improvement after hormonal suppression in a girl with precocious puberty

Abstract

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 per se. 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.

Keywords

• Primary chronic osteomyelitis
• Chronic nonbacterial osteomyelitis
• Mandible
• Precocious puberty
• Gonadotropin-releasing hormone