Cyclic administration of pamidronate in children with severe osteogenesis imperfecta.

  title={Cyclic administration of pamidronate in children with severe osteogenesis imperfecta.},
  author={Francis H. Glorieux and N. J. Bishop and Horacio Plotkin and Guillaume Chabot and G Lanoue and Rose Travers},
  journal={The New England journal of medicine},
  volume={339 14},
BACKGROUND Severe osteogenesis imperfecta is a disorder characterized by osteopenia, frequent fractures, progressive deformity, loss of mobility, and chronic bone pain. There is no effective therapy for the disorder. We assessed the effects of treatment with a bisphosphonate on bone resorption. METHODS In an uncontrolled observational study involving 30 children who were 3 to 16 years old and had severe osteogenesis imperfecta, we administered pamidronate intravenously (mean [+/-SD] dose, 6.8… 

Cyclic Pamidronate Therapy in Children with Osteogenesis Imperfecta

There was a significant relief of chronic pain and fatigue but no adverse effects in all children using the drug and cyclic pamidronate administration is effective in improving bone mineralization and reducing fracture incidence in childhood osteogenesis imperfecta.

Effect of intravenous pamidronate treatment in children with osteogenesis imperfecta.

Bisphosphonate seems to be an effective symptomatic treatment for children with osteogenesis imperfecta irrespective of severity of mutation or clinical phenotype and has a positive effect on fracture rate, BMD, mobility score, wellbeing and pain episode.

Pamidronate Treatment of Children with Moderate-to-Severe Osteogenesis Imperfecta: A Note of Caution

Pamidronate with physiotherapy and orthopedic management improved outcomes without delaying fracture healing in 19 (70%) of 27 patients, and should be reserved for severe OI with multiple fractures and/or flattened vertebras.

Beneficial effects of intravenous pamidronate treatment in children with osteogenesis imperfecta under 24 months of age

Treatment with intravenous pamidronate is safe, significantly improves lumbar bone mineral density (L-BMD), and reduces fracture rates in young infants with OI while preserving linear growth.

Cyclic pamidronate infusion for neonatal-onset osteogenesis imperfecta.

Radiological features of bisphosphonate therapy in children with osteogenesis imperfecta.

The radiographic features of cyclic pamidronate administration on the growing skeleton in children with OI, a genetic disorder of type-I collagen, are determined and described.

Low-dose intravenous pamidronate treatment in osteogenesis imperfecta.

Low-dose cyclical pamidronate infusions markedly increased bone density and decreased bone fracture rate and should be considered as a part of a multi-disciplinary treatment in children with osteogenesis imperfecta.

Pamidronate Treatment of Less Severe Forms of Osteogenesis Imperfecta in Children

Pamidronate treatment improves bone quality in children with mild types of OI, improves mobility, reduces fracture frequency and thus improves quality of life and in future is likely to reduce the severity and consequences of adult osteoporosis by improved peak bone mass in these children.

Beneficial effect of long term intravenous bisphosphonate treatment of osteogenesis imperfecta

Disodium pamidronate (APD) was given as monthly intravenous infusions to 28 children and adolescents with severe OI or a milder form of the disease, but with spinal compression fractures to find an effective symptomatic treatment.



Pamidronate treatment of severe osteogenesis imperfecta in children under 3 years of age.

Pamidronate treatment in severely affected OI patients under 3 yr of age is safe, increases BMD, and decreases fracture rate, and no adverse side-effects were noted.

Intravenous pamidronate treatment in osteogenesis imperfecta.

Intravenous pamidronate in juvenile osteoporosis

Intravenous pamidronate appears to be a useful therapeutic option in childhood osteoporosis, but its use in children must still be regarded as experimental and therefore closely monitored.

Lack of effects of human calcitonin in osteogenesis imperfecta.

It was concluded that, with the dose of human calcitonin used, it was impossible to detect any beneficial effect in patients with osteogenesis imperfecta.

Failure of Anabolic Steroids in the Therapy of Osteogenesis Imperfecta

Anabolic steroids have been administered to eight patients with severe osteogenesis imperfecta, who were studied clinically and by metabolic balance techniques, and evidence of increased bone catabolism after withdrawal of therapy was found.

Adverse Effects of Bisphosphonates

SummaryThe bisphosphonates comprise a new class of drugs, and are increasingly being used to treat bone diseases characterised by increased osteoclastic bone resorption. These compounds are generally

Efficacious management with aminobisphosphonate (APD) in Paget's disease of bone.

If the low specific toxicity of the new generations of P-C-Ps is confirmed, it will be possible to induce complete and prolonged remissions through short oral or parenteral treatment courses that are associated with minimal side effects.

Studies of patients with osteogenesis imperfecta.

The over-all results do not warrant the general use of fluoride for the treatment of osteogenesis imperfecta but do warrant further study, especially of the dose-related effect.

Differing Lumbar Vertebral Mineralization Rates in Ambulatory Pediatric Patients with Osteogenesis Imperfecta

It is concluded that vertebral body size is a critical determinant of BMD and BMD Z-score in OI because DXA results are expressed per unit area, not per unit volume.