Remarkable response of juvenile diffuse sclerosing osteomyelitis of mandible to pamidronate.

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We report a juvenile case of diffuse sclerosing osteomyelitis of the mandible thatshowed a favorable response to pamidronate, a bisphosphonate derivative. Althoughconventional treatments had been ineffective for five years, pamidronate administrationbrought about conspicuous improvement both clinically and radiographically. Severeadverse reaction was not found except for low-grade fever and lassitude only on the dayfollowing administration. During the course of the treatment, however,nonsuppurative osteomyelitis of the right humerus also occurred, leading to theestablished diagnosis of chronic recurrent multifocal osteomyelitis. Then, pamidronatetherapy was again performed successfully with almost disappearance of clinicalsymptoms. Both bone-specific alkaline phosphatase (bone formation marker) andpyridinoline cross-linked carboxyterminal telopeptide of type I collagen (ICPT) (boneresorption marker) showed a marked decrease with pamidronate therapy, whichsuggested that pamidronate is useful for the treatment of chronic recurrent multifocalosteomyelitis with inhibitory effect on bone turnover.
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