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Effect of Pamidronate Treatment in Children with Polyostotic Fibrous Dysplasia of Bone

Genetics Unit, Shriners Hospital for Children, Québec, Canada H3G 1A6; and the Departments of Surgery and Pediatrics, McGill University, Montréal, Québec, Canada H3H 1P3

Address all correspondence and requests for reprints to: Frank Rauch, Genetics Unit, Shriners Hospital for Children, 1529 Cedar Avenue, Montréal, Québec, Canada H3G 1A6. E-mail: frauch{at}shriners.mcgill.ca.

Intravenous infusions with the bisphosphonate compound pamidronate decrease bone pain and reportedly can lead to refilling of dysplastic lesions in adults with fibrous dysplasia (FD) of bone. Here we describe the effects of this treatment approach in 18 children and adolescents (age at start of therapy, 6.2–17.5 yr; eight girls) with polyostotic FD, who received pamidronate for 1.2–9.1 yr (median, 3.8 yr). Treatment cycles with pamidronate (1–1.5 mg/kg·d on 3 consecutive days) were given every 4 months. Levels of serum alkaline phosphatase and urinary collagen type I N-telopeptide were elevated at baseline and decreased continuously during the first 3 yr of therapy. There was no radiographic evidence of filling of lytic lesions or thickening of the bone cortex surrounding the lesions in any patient. Histomorphometric results in dysplastic bone tissue of patients receiving pamidronate (n = 7; time of therapy, 1.4–4.8 yr) were similar to those of patients without medical therapy (n = 9). No serious side effects were noted. In conclusion, pamidronate therapy appears to be safe in children and adolescents with polyostotic FD. However, we found no clear evidence that pamidronate has an effect on dysplastic lesions in such patients.

This study was supported by the Shriners of North America. C.M. is the Royal Children’s Hospital Foundation/Woolworths Scholar.

Current address for H.P.: Inherited Metabolic Diseases Section, Children’s Hospital and Department of Pediatrics, University of Nebraska, Omaha, Nebraska 68198-5430.

Abbreviations: BMC, Bone mineral content; FD, fibrous dysplasia; NTX, N-telopeptide of type I collagen; vBMD, volumetric bone mineral density.

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