Skip to main content
Top
Published in: Osteoporosis International 8/2007

01-08-2007 | Case Report

Can bisphosphonate treatment be stopped in a growing child with skeletal fragility?

Authors: K. A. Ward, J. E. Adams, T. J. Freemont, M. Z. Mughal

Published in: Osteoporosis International | Issue 8/2007

Login to get access

Abstract

Summary

Cyclical pamidronate therapy in a 2-year-old child with skeletal fragility resulted in remodelling of vertebral fractures and improvement in bone mineral density (BMD) at distal radial and spinal sites. The BMD at both sites decreased precipitously within 24 months of stopping treatment, raising the question as to whether bisphosphonates can be stopped in a growing child with skeletal fragility.

Introduction

At age 23 months, a male toddler sustained a low trauma fracture of his right femur. Skeletal radiographs revealed generalised osteopenia with multiple vertebral body fractures. He was diagnosed with type IV osteogenesis imperfecta; however, no mutations were found in COL1A1 or COL1A2 genes.

Methods

This case report presents bone densitometry data before, during and after bisphosphonate treatment. Axial QCT was main outcome from 2 years of age; DXA and pQCT were taken after age 5.

Results

QCT confirmed that he had low spinal trabecular volumetric BMD (Z-score −2.4). After 4 years of treatment his vertebral fractures had been remodelled and all bone densitometry values (QCT, DXA and pQCT) were within normal range and therefore treatment was discontinued. Shortly after this he suffered stress fractures of his left mid tibia and at the sclerotic metaphyseal line corresponding to his first APD treatment. He had marked reduction in spinal trabecular and distal radial vBMD; change in BMAD was less marked.

Conclusion

The patient has been restarted on IV APD therapy. This case has led us to consider whether bisphosphonate therapy can be discontinued in a child with fragility fractures before his/her linear growth has ceased?
Footnotes
1
Z-scores for QCT were calculated using the manufacturer’s reference data [11].
 
2
DXA and pQCT were calculated using Manchester Normative Reference Database of 5–18 year old healthy children [12].
 
Literature
1.
go back to reference Thornton J, Ashcroft DM, Mughal Z, Elliott RA, O’Neill TW, Symmons D (2006) Systematic review of effectiveness of bisphosphonates in treatment of low bone mineral density and fragility fractures in juvenile idiopathic arthritis. Arch Dis Child 91:753–761CrossRef Thornton J, Ashcroft DM, Mughal Z, Elliott RA, O’Neill TW, Symmons D (2006) Systematic review of effectiveness of bisphosphonates in treatment of low bone mineral density and fragility fractures in juvenile idiopathic arthritis. Arch Dis Child 91:753–761CrossRef
2.
go back to reference Glorieux FH, Bishop NJ, Plotkin H, Chabot G, Lanoue G, Travers R (1998) Cyclic administration of pamidronate in children with severe osteogenesis imperfecta. N Engl J Med 339:947–952PubMedCrossRef Glorieux FH, Bishop NJ, Plotkin H, Chabot G, Lanoue G, Travers R (1998) Cyclic administration of pamidronate in children with severe osteogenesis imperfecta. N Engl J Med 339:947–952PubMedCrossRef
3.
go back to reference Shaw N, Boivin C, Crabtree N (2000) Intravenous pamidronate in juvenile osteoporosis. Arch Dis Child 83:143–145PubMedCrossRef Shaw N, Boivin C, Crabtree N (2000) Intravenous pamidronate in juvenile osteoporosis. Arch Dis Child 83:143–145PubMedCrossRef
4.
go back to reference Rauch F, Travers R, Plotkin H, Glorieux FH (2002) The effects of intravenous pamidronate on the bone tissue of children and adolescents with osteogenesis imperfecta. J Clin Invest 110:1293–1299PubMedCrossRef Rauch F, Travers R, Plotkin H, Glorieux FH (2002) The effects of intravenous pamidronate on the bone tissue of children and adolescents with osteogenesis imperfecta. J Clin Invest 110:1293–1299PubMedCrossRef
5.
go back to reference Rauch F, Plotkin H, Zeitlin L, Glorieux FH (2003) Bone mass, size, and density in children and adolescents with osteogenesis imperfecta: effect of intravenous pamidronate therapy. J Bone Miner Res 18:610–614PubMedCrossRef Rauch F, Plotkin H, Zeitlin L, Glorieux FH (2003) Bone mass, size, and density in children and adolescents with osteogenesis imperfecta: effect of intravenous pamidronate therapy. J Bone Miner Res 18:610–614PubMedCrossRef
6.
go back to reference Plotkin H, Rauch F, Bishop NJ, Montpetit K, Ruck-Gibis J, Travers R, Glorieux FH (2000) Pamidronate treatment of severe osteogenesis imperfecta in children under 3 years of age. J Clin Endocrinol Metab 85:1846–1850PubMedCrossRef Plotkin H, Rauch F, Bishop NJ, Montpetit K, Ruck-Gibis J, Travers R, Glorieux FH (2000) Pamidronate treatment of severe osteogenesis imperfecta in children under 3 years of age. J Clin Endocrinol Metab 85:1846–1850PubMedCrossRef
7.
go back to reference Munns CF, Rauch F, Travers R, Glorieux FH (2005) Effects of intravenous pamidronate treatment in infants with osteogenesis imperfecta: clinical and histomorphometric outcome. J Bone Miner Res 20:1235–1243PubMedCrossRef Munns CF, Rauch F, Travers R, Glorieux FH (2005) Effects of intravenous pamidronate treatment in infants with osteogenesis imperfecta: clinical and histomorphometric outcome. J Bone Miner Res 20:1235–1243PubMedCrossRef
8.
go back to reference Rauch F, Munns C, Land C, Glorieux FH (2006) Pamidronate in children and adolescents with osteogenesis imperfecta: effect of treatment discontinuation. J Clin Endocrinol Metab 91:1268–1274PubMedCrossRef Rauch F, Munns C, Land C, Glorieux FH (2006) Pamidronate in children and adolescents with osteogenesis imperfecta: effect of treatment discontinuation. J Clin Endocrinol Metab 91:1268–1274PubMedCrossRef
9.
go back to reference Steelman J, Zeitler P (2003) Treatment of symptomatic pediatric osteoporosis with cyclic single-day intravenous pamidronate infusions. J Pediatr 142:417–423PubMedCrossRef Steelman J, Zeitler P (2003) Treatment of symptomatic pediatric osteoporosis with cyclic single-day intravenous pamidronate infusions. J Pediatr 142:417–423PubMedCrossRef
10.
11.
go back to reference Gilsanz V, Gibbens D, Roe T, Carlson M, Senac M, Boechat M, Huang H, Schulz E, Libanati C, Cann C (1988) Vertebral bone density in children: effect of puberty. Radiology 166:847–850PubMed Gilsanz V, Gibbens D, Roe T, Carlson M, Senac M, Boechat M, Huang H, Schulz E, Libanati C, Cann C (1988) Vertebral bone density in children: effect of puberty. Radiology 166:847–850PubMed
12.
go back to reference Ward K, Ashby R, Roberts S, Adams J, Mughal M (2006) United Kingdom reference data for the hologic QDR discovery dual energy x-ray absorptiometry scanner in healthy children aged 6–17 years. Arch Dis Child DOI 10.1136/adc.2006.097642 Ward K, Ashby R, Roberts S, Adams J, Mughal M (2006) United Kingdom reference data for the hologic QDR discovery dual energy x-ray absorptiometry scanner in healthy children aged 6–17 years. Arch Dis Child DOI 10.​1136/​adc.​2006.​097642
13.
go back to reference Carter D, Bouxsein M, Marcus R (1992) New approaches for interpreting projected bone densitometry data. J Bone Miner Res 7:137–145PubMedCrossRef Carter D, Bouxsein M, Marcus R (1992) New approaches for interpreting projected bone densitometry data. J Bone Miner Res 7:137–145PubMedCrossRef
14.
go back to reference Neu C, Manz F, Rauch F, Merkel A, Schonau E (2001) Bone densities and bone size at the distal radius in healthy children and adolescents: a study using peripheral quantitative computed tomography. Bone 28:227–232PubMedCrossRef Neu C, Manz F, Rauch F, Merkel A, Schonau E (2001) Bone densities and bone size at the distal radius in healthy children and adolescents: a study using peripheral quantitative computed tomography. Bone 28:227–232PubMedCrossRef
15.
go back to reference Rauch F, Travers R, Munns C, Glorieux F (2004) Sclerotic metaphyseal lines in a child treated with pamidronate: histomorphometric analysis. J Bone Miner Res 19:1191–1193PubMedCrossRef Rauch F, Travers R, Munns C, Glorieux F (2004) Sclerotic metaphyseal lines in a child treated with pamidronate: histomorphometric analysis. J Bone Miner Res 19:1191–1193PubMedCrossRef
16.
go back to reference Land C, Rauch F, Glorieux FH (2006) Cyclical intravenous pamidronate treatment affects metaphyseal modeling in growing patients with osteogenesis imperfecta. J Bone Miner Res 21:374–379PubMedCrossRef Land C, Rauch F, Glorieux FH (2006) Cyclical intravenous pamidronate treatment affects metaphyseal modeling in growing patients with osteogenesis imperfecta. J Bone Miner Res 21:374–379PubMedCrossRef
Metadata
Title
Can bisphosphonate treatment be stopped in a growing child with skeletal fragility?
Authors
K. A. Ward
J. E. Adams
T. J. Freemont
M. Z. Mughal
Publication date
01-08-2007
Publisher
Springer-Verlag
Published in
Osteoporosis International / Issue 8/2007
Print ISSN: 0937-941X
Electronic ISSN: 1433-2965
DOI
https://doi.org/10.1007/s00198-007-0330-3

Other articles of this Issue 8/2007

Osteoporosis International 8/2007 Go to the issue