Juvenile Mandibular Chronic Osteomyelitis: Role of Surgical Debridement and Antibiotics

Juvenile mandibular chronic osteomyelitis (JMCO) is characterized by multiple episodes of pain, swelling, tenderness and limited jaw motion, predominantly occurring in girls. Radiographic and computed tomographic (CT) imaging is consistent with osteomyelitis:  subperiosteal bone formation and mixed radio-dense/radiolucent trabecular pattern. Inflammatory markers (erythrocyte sedimentation rate and C-reactive protein) are often elevated. Because standard bacterial cultures are frequently negative, there is controversy as to whether this is an infectious disease or a non-infectious inflammatory disorder.  We hypothesize that JMCO is an infectious disease and that standard culture techniques simply fail to identify the responsible organisms¹for a variety of reasons.  The purpose of this study was to document the response and long term outcomes of JMCO using a standardized protocol of extra-oral biopsy, culture, decortication and long term antibiotic therapy.  

              This is a retrospective study of children with JMCO treated at Massachusetts General Hospital between 1996 and 2014.  Inclusion criteria included: 1) age 2-18 years, 2) symptoms of recurrent mandibular pain and swelling, 3) radiographic evidence of osteomyelitis, 4) availability of histopathologic and culture data.  Inpatient and outpatient records were reviewed for: demographic (age, sex) and disease related  (signs and symptoms, radiology, operative, pathology and culture results) data.  Outcome variables were disease status at end treatment (cure, intermittent flare-ups), and complications.  Protocol consisted of extra-oral biopsy, debridement and decortication. Specimens were sent for aerobic, anaerobic and fungal cultures. Patients were then started on a 6-12 week course of intravenous antibiotics via peripherally inserted central catheter (PICC) line or, in patients under 10 years, high dose oral antibiotics. Maintenance oral antibiotics were continued for a mean of 10.8 months (1-24 months range). Criteria for discontinuing antibiotics included: symptom free interval of 3 months, radiographic changes indicating bone remodeling and no progression of disease. The histopathology slides were reviewed with one pathologist blinded to the clinical outcomes.  

            A total of 12 patients (11 females), with mean age of onset of 10.7 years (range 3-14 years) met the inclusion criteria. All subjects had pain and/or swelling as the presenting complaint with 5 having limitation of jaw motion. All patients had screening orthopantomograms and CT scans which demonstrated expansion of the mandible, mixed radiolucent/radiodense trabecular patterns, sclerosis and sub-periosteal bone formation. Management in 12/12 patients consisted of biopsy (extraoral n=10; intraoral, n=2), decortication and long term antibiotic therapy.  NSAIDs were administered only as needed for pain control.  Duration of follow up ranged from 1-11 years with mean follow up of 3.79 years.  There was complete resolution of osteomyelitis in 7/12 patients with mean follow-up of 4.3 years (range  1-11). Of these patients, 1 ultimately required mandibular resection, 1 marginal resection and 1 had neuropathic pain in absence of active disease. 5 patients had improvement in symptoms with less frequent flare ups (every week pre-treatment to once every few months post treatment), well controlled with short courses of antibiotics (4-12 weeks)  and  NSAIDs  (mean follow up 1.4 years range 1-3 years). There were no major complications related to  antibiotic therapy. Minor complications included:  rash (n=2), nausea and vomiting (n=1), vaginal candidiasis (n=1).              

             The results of this study indicate that 58.3% of subjects were cured with a mean follow-up of 4.3years.  The cure rate with long term follow-up in this study was higher than that in comparable previously published reports²without the use of antibiotics. A prospective study is planned with extraoral mandibular biopsy and molecular culture techniques to identify specific etiologic organisms.   

REFERENCES

1. Flynn TR, Paster BJ et al. Molecular Methods for Diagnosis of Odontogenic Infections. J Oral Maxillofac Surg, 2012, Vol.70(8), pp.1854-1859
2. Heggie AA, Shand JM, et al: Juvenile mandibular chronic osteomyelitis: A distinct clinical entity. Int. J. Oral Maxillofac. Surg. 2003; 32: 459--468