Long-Term Follow-Up of Bimaxillary Osteomyelitis Associated with Autosomal Dominant Osteopetrosis: A Case Report

Abstract

Osteopetrosis (OP) is a rare metabolic bone disease characterized by a generalized increase in skeletal mass. The disease is characterized by increased susceptibility to develop osteomyelitis of the jaws. We report a case of clinical and radiological progression of bimaxillary osteomyelitis in a patient with autosomal dominant OP. The patient presented non simultaneous osteomyelitis in both upper and lower jaws with time interval of 10 years. The osteomyelitis of maxilla resulted in oroantral fistula formation and required surgical closure with Bichat fat pad flap. The mandibular osteomyelitis resulted in sequestra formation and pathological fracture and required multiple debridement procedures. Both maxillary and mandibular osteomyelitic foci were persistent and healing required more than 2 years. The case confirms the possibility of development of non simultaneous bimaxillary osteomyelitis in patients with OP. The case enhances the need of close follow- up and preventive measures in patients with OP.

Case report of osteomyelitis of the mandible in osteopetrosis and management considerations

“…Tooth extraction, bone trimming, fistula cures, and antibiotic treatment, stabilized the infection. However, bone exposure remained and the risk of recurrence of infection is high due to the persistence of ischemia and contamination of the bone [ 18 ]. The prospect is to continue regular patient follow-up in our department.…”

“…Patients have immune fragility with, in particular, anemia, hypocalcemia, thrombocytopenia. Firstly, these must be diagnosed and corrected to improve tolerance and effectiveness of future treatments [ 18 ]. Antibiotic therapy generally uses Amoxicillin with clavulanic acid as the main choice, then Clindamycin as follow up but no study suggests a specific duration.…”

Osteopetrosis: presenting as maxillary osteomyelitis

“…Investigations proved a severe anaemia (Hb 56 g/L) but normal white blood count of 7200/mm 3 and mild thrombocytopenia (120/µL). His serum calcium, phosphate and alkaline phosphatase, arterial blood gas analysis, liver and kidney function tests were all normal.…”

“…As a result of diffuse generalised osteosclerosis of bones, these patients are prone to serious oral complications such as osteomyelitis, which arises in 10% of cases, involving the mandible 1 or sometimes the maxilla, scapula and other extremities. 3–5…”

“…2 As a result of diffuse generalised osteosclerosis of bones, these patients are prone to serious oral complications such as osteomyelitis, which arises in 10% of cases, involving the mandible 1 or sometimes the maxilla, scapula and other extremities. [3][4][5] Here we discuss a boy who presented with recurrent dental infections and osteomyelitis of the maxilla and was found to have underlying osteopetrosis of the infantile (malignant) type.…”