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Published on: October 16, 2013
Development of scoliosis in young children with osteogenesis imperfecta undergoing intravenous bisphosphonate therapy
Masafumi Kashii1, Sadaaki Kanayama2, Taichi Kitaoka3
1Department of Orthopedic Surgery, Faculty of Medicine, Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan. mkashii-osk@umin.ac.jp.
Insights
Scoliosis affects 23.5% of young children with osteogenesis imperfecta (OI) treated with pamidronate (PAM). Lower bone density and vertebral deformities are key risk factors. Early PAM therapy may prevent scoliosis in severe OI types.
Area of Science:
- Pediatric Orthopedics
- Genetics and Bone Diseases
- Pharmacology
Background:
- Osteogenesis imperfecta (OI) is a genetic disorder characterized by bone fragility.
- Scoliosis is a common complication in children with OI, impacting quality of life.
- Pamidronate (PAM) therapy is used to manage OI, but its effect on scoliosis development requires further clarification.
Purpose of the Study:
- To determine the prevalence of scoliosis in young children with OI receiving pamidronate (PAM) therapy.
- To identify risk factors associated with scoliosis development in this patient population.
- To evaluate the potential of early PAM intervention in preventing scoliosis.
Main Methods:
- Retrospective review of medical records and radiographs of 34 young children with OI.
- Analysis of scoliosis incidence (Cobb angle ≥ 10) in relation to OI type, mobility, bone mineral density (BMD), and treatment parameters.
- Statistical examination of risk factors including vertebral deformities, pelvic tilt, and leg-length discrepancy.
Main Results:
- Scoliosis prevalence was 23.5% among the studied children after a mean follow-up of 4.2 years.
- Significant risk factors for scoliosis included lower lumbar spine BMD Z-scores, vertebral deformities, and corrective lower extremity osteotomies.
- Patients with severe OI types (III/IV) and scoliosis had lower BMD Z-scores and started PAM therapy later compared to those without scoliosis.
Conclusions:
- Scoliosis development in OI is linked to disease severity and bone fragility, persisting even with PAM therapy.
- Lower bone mineral density and vertebral deformities are critical risk factors for scoliosis in OI patients.
- Initiating intravenous PAM therapy in infancy or early childhood may prevent or reduce scoliosis in severe OI types (III/IV).
Abstract:
The purpose of this study was to clarify the prevalence of scoliosis and determine risk factors for the development of scoliosis in young children with osteogenesis imperfecta (OI) who underwent intravenous pamidronate (PAM) therapy. Thirty-four young children with OI who had no scoliosis at the first PAM administration underwent cyclic PAM therapy alone. The medical records and radiographs of these patients were retrospectively reviewed. We examined the relationship between scoliosis (Cobb angle ≥ 10) and type of OI (Sillence classification: types I, III, and IV), physical mobility, Z-scores of bone mineral density in L2-4 of the lumbar spine (L2-4 BMD Z-scores), age of patients at first treatment with PAM, pelvic frontal tilt and leg-length discrepancy. The prevalence of scoliosis was 23.5% in 34 young children with OI who underwent PAM therapy for a mean of 4.2 years. Lower L2-4 BMD Z-scores, the presence of coronal and sagittal vertebral deformities and higher percentage of corrective osteotomy in the lower extremities were significant risk factors for the development of scoliosis. In patients with type III or IV OI, L2-4 BMD Z-scores were significantly lower (p = 0.02) and the percentage of patients who started PAM therapy in early childhood was significantly lower in scoliosis group than in the non-scoliosis group (p = 0.01). Development of scoliosis depends on the severity of OI and has a strong relationship with bone fragility even under PAM therapy. Starting intravenous PAM therapy in infancy or early childhood has a potential to prevent the occurrence and progression of scoliosis associated with bone fragility in young children with severe type III or IV OI.
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