A 13-year-old boy presented in accident and emergency with a 3-week history of left-sided facial swelling. The presenting symptoms were pain, and trismus with a 20-mm interincisal distance mouth opening recorded. He was medically fit and well with no allergies. In the first instance, an orthopantomagraph (OPG) was taken for dental assessment (Figure 1). Clinical examination showed no obvious intra-oral swelling and no tenderness to percussion of any teeth on the left side. The OPG revealed no dental cause, but bony expansion of the left mandible was evident. There was no history of recent trauma, but his parents could not rule out trauma or a fall in the past. An urgent ultrasound scan was carried out, indicating thickening of the left masseter muscle, which was suggestive of inflammation. There were also some subtle cortical irregularities within the outer cortex of the mandible. The patient was given IV co-amoxiclav and metronidazole as well as analgesics. The swelling reduced and the symptoms subsided after 2 days of IV antibiotics.
The patient returned after a couple of months complaining of swelling and pain from left mandible. A biopsy of this area was taken under general anaesthetic. The histology confirmed reactive bone. A subsequent CT scan was performed. The CT revealed a subperiosteal reaction, which was seen buccally along the mandible extending from the region of the left mental foramen to the left neck of the condyle and left coronoid process (Figures 2 and 3). There was also loss of the appearance of the trabecular bone within the mandible from the LL5 region posteriorly to the ascending ramps, with widespread replacement by sclerotic bone. With the CT scan, the histological findings of reactive bone and given patient's age, it was concluded that the diagnosis was most likely to be sclerosing osteomyelitis of Garré. The swelling gradually reduced in size following treatment with IV antibiotics, but did not resolve completely. The cause of the bony expansion was not established; however, the patient was monitored regularly.
Findings
Garré's osteomyelitis normally arises as a result of dental infection or trauma. Other causes of Garré's osteomyelitis have been reported in relation to dental extraction, mild periodontitis and trauma.6 The swelling typically appears in the molar or premolar region.7 Patients present with hard bony swelling of the mandible, resulting in facial asymmetry.8 Dental infection is caused by tooth decay, which, if left untreated, can develop into a chronic infection leading to pulp necrosis. The infection extends to the bone, which in turn stimulates bony proliferation by the periosteum.9,10 Pain is not usually a complaint, but if the lesion is secondarily infected, then severe pain is a common characteristic.11
Radiographic examinations such as OPG or CT are important diagnostic tools in determining a diagnosis of Garré's osteomyelitis. CT scans show the presence of bony lamellae parallel to each other outside the cortex of the bone involved.5 Scans also show an ‘onion skin’ appearance of new periosteal proliferation located in successive layers parallel to condensed cortical bone.7,12 Kannan et al concluded that the radiographic appearance of Garré's osteomyelitis may determine the duration, progression and the mode of healing of the disease process.10
Garré's osteomyelitis could mimic the appearance of fibrous dysplasia and, therefore, must be investigated thoroughly. Osteoblastic osteosarcoma, Ewing's sarcoma and other benign and malignant tumours must not be ruled out until a definitive diagnosis is reached.3
There are numerous case reports in literature that support development of Garré's osteomyelitis following a dental abscess. In one case4 of a 5 year old, the patient presented with slowly progressive right-sided hard bony swelling of the mandible. An OPG showed a typical periosteal reaction of the inferior cortex of the mandible associated with the infected deciduous mandibular second molar. The patient was treated with antibiotics and removal of the offending tooth. The swelling regressed and resolved over a period of time. Similarly, in another report,9 a 12-year-old girl presented with a 5-month history of left mandibular expansion. Dental radiograph had confirmed the presence of infection associated with the lower left first molar. In this case, a subsequent CT scan was performed to exclude other potential causes. The tooth was extracted and a biopsy was taken. The swelling had gradually subsided by a 3-month review.
In the case8 of a 10-year old boy who presented with pain from the lower left region and extra-oral swelling on the right inferior border of the mandible, extra-oral examination of the right mandible revealed a diffuse hard non-tender swelling with normal skin colour. Intra-oral examination revealed carious lesions related to LR6 and LL6. LR6 was tender to percussion. Occlusal radiographs of the mandible had shown an ‘onion skin’ appearance in relation to peripheral sub-periosteal bone deposition on the right side. Eventually LR6 and LL6 were root-canal treated with intra-canal antibiotics. The canals were irrigated with mixture of metronidazole and gentamicin. After 3 weeks, the swelling had completely subsided and at the 3-month recall, the occlusal radiograph and intra-oral peri-apical radiograph showed complete remodelling of the bone.8
In the literature, a number cases have been presented where pericoronitis has been reported to encourage harbouring of bacteria, which can eventually lead to infection and osteomyelitis.6,13 This highlights the possibility of periodontal involvement in the development of Garré's osteomyelitis in the absence of abscess secondary to dental caries, although it is rare. In a case14 of a 9-year old girl who presented with a 1-month history of painless, bony hard swelling of the right lower face, the patient had developed swelling after being hit by a seesaw. The swelling increased over time. An OPG and occlusal film showed radio-opaque formation without cortical involvement and confirmed an ‘onion ring’ appearance. CT scan further confirmed these findings. Bone scans indicated a high bone remodelling activity of the right mandible and a biopsy confirmed Garré's osteomyelitis. The patient was treated with 2 weeks of antibiotics and non-steroidal anti-inflammatory drugs. The facial asymmetry had shown improvement by the 4-week follow-up appointment.