Intractable bimaxillary osteomyelitis in osteopetrosis: review of the literature and current therapy.

PURPOSE: Osteopetrosis (OP), also known as Albers-Schönberg disease, is a rare metabolic bone disease caused by a congenital defect in the development or function of the osteoclasts resulting in generalized increase in skeletal mass. Osteomyelitis is a recognized complication, and prevention of dental infections can be difficult. The locations of osteomyelitis in the jawbones have been either in the mandible or maxilla. In this article, a case of OP complicated with osteomyelitis of the mandible and the maxilla is presented, and types of OP and treatment options are discussed with a literature review.

MATERIALS AND METHODS: The 18-year-old blind female patient had been diagnosed with OP in the past. She had a purulent discharge at the medial canthus of the left eye, in addition to another discharge of pus in the infraorbital and upper left molar region, and multiple malformed and partially impacted teeth, and there were sequestrum formations in the maxilla and mandible. For the treatment of osteomyelitis, systemic combined antibiotics of sultamicillin 750 mg and ornidazole 500 mg twice daily, were given orally for 5 weeks followed by surgical removal of small sequestra in the jaws. Persistent osteomyelitic bones were removed by saucerization in the mandibular anterior and right premolar region and the left posterior maxilla under the same antibiotic regimen. After wound healing was completely achieved in both jaws, the oroantral opening in the left posterior maxilla was successfully closed with a buccal flap under antibiotic coverage.

RESULTS: The purulent discharge at the medial canthus of her left eye subsided and she had no recurrent infections during the long-term follow-up of 2 years.

CONCLUSION: The case reported shows that osteomyelitis is difficult to treat in subjects suffering from OP. Therefore, it is necessary to be very cautious against infections in even minor surgical interventions such as simple dental extractions in patients with OP, regardless of the location of the teeth. When considering removal of impacted teeth in the posterior maxilla, resultant oroantral communication from osteomyelitis of the extraction site should be regarded as a potential complication.