PubMed · 16463553
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Bettina Polk. 2006. [Ibandronate once a month].. https://pubmed.ncbi.nlm.nih.gov/16463553/
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Osteogenesis imperfecta is a relatively common hereditary connective tissue disorder characterized by bone fragility and fractures. Other frequently affected tissues include tendons, ligaments, skin, sclera, teeth, and middle and inner ear. Molecular studies have demonstrated that most cases result from mutations affecting the genes responsible for the formation of type 1 collagen. The phenotypic presentation varies from mild to lethal. Commonly observed dental abnormalities include dentinogenesis imperfecta and malocclusion. Medical therapies using bisphosphonates have resulted in reduced fracture risk and decreased bone pain. To date, no cases of bisphosphonate-associated osteonecrosis have been reported. With appropriate precautions, the patient with osteogenesis imperfecta can tolerate and benefit from the delivery of necessary dental care to control oral disease, improve function, and improve esthetics.
We report a juvenile case of diffuse sclerosing osteomyelitis of the mandible that showed a favorable response to pamidronate, a bisphosphonate derivative. Although conventional treatments had been ineffective for 5 years, pamidronate administration brought about conspicuous improvement both clinically and radiographically. Severe adverse reaction was not found except for low-grade fever and lassitude on the day following administration. During the course of the treatment, however, nonsuppurative osteomyelitis of the right humerus also occurred, leading to the established diagnosis of chronic recurrent multifocal osteomyelitis. Pamidronate therapy was again performed successfully with near disappearance of clinical symptoms. Both bone-specific alkaline phosphatase (bone formation marker) and pyridinoline cross-linked carboxyterminal telopeptide of type I collagen (bone resorption marker) showed a marked decrease with pamidronate therapy, suggesting that pamidronate is useful for the treatment of chronic recurrent multifocal osteomyelitis with inhibitory effect on bone turnover.