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Effective parenteral clodronate treatment of a child with severe juvenile idiopathic osteoporosis

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Abstract

We report on an 8 years and 3 months old boy with severe idiopathic juvenile osteoporosis (IJO). Clinical features included multiple fractures, especially of the vertebrae, and neurological symptoms. Biological studies showed non-parathyroid hormone-mediated excessive bone resorption and massive urinary calcium loss. Although IJO is usually a self-limiting condition after puberty, the severity of our patient’s manifestations required therapeutic intervention. Clodronate (dichloromethylene-bisphosphonate) was administered parenterally every 3 months for a period of 2 years. Dramatic clinical and biochemical improvement was noted within 2 weeks. All parameters of bone resorption normalised and no new fractures occurred. After 6 months of treatment, radiological improvement with healing of fractures and rebuilding of the vertebral plates was documented. Bone mineral density increased to normal within 1 year and growth velocity was accelerated. After 2 years, treatment was stopped at the age of 10 years and 3 months. One year later, back pain and increasing pain in the knee region recurred. A tibial fracture was evident and, again, bone mineral density was far below normal. Bisphosphonate medication was reinstituted leading to rapid improvement. No side-effects were observed. Conclusion:parenteral clodronate therapy is effective in managing severe idiopathic juvenile osteoporosis.

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Abbreviations

DPD :

desoxypyridinoline-crosslinks

IJO :

idiopathic juvenile osteoporosis

PTH :

parathyroid hormone

References

  1. Allgrove J (1996) Bisphosphonates—paediatric indications advantages and possible side effects. In: Schönau E (ed) Paediatric osteology. New developments in diagnosis and therapy: proceedings of the First International Workshop, held in Cologne, Germany 5-7 October 1995 (International Congress Series). Exerpta Medica, Almere, pp 261–273

    Google Scholar 

  2. Astrom E, Soderhall S (1998) Beneficial effect of bisphosphonate during five years of treatment of severe osteogenesis imperfecta. Acta Paediatr 87: 64–68

    Article  CAS  Google Scholar 

  3. Batch JA, Couper JJ, Rodda C, Cowell CT, Zacharin M (2003) Use of bisphosphonate therapy for osteoporosis in childhood and adolescence J Paediatr Child Health 39: 88–92

    CAS  PubMed  Google Scholar 

  4. Bishop NJ, Williams DM, Compston JC, Stirling DM, Prentice A (1995) Osteoporosis in severe congenital neutropenia treated with granulocyte colony-stimulating factor. Br J Haematol 89: 927–928 (published erratum appears in Br J Haematol 90: 492)

    Article  CAS  Google Scholar 

  5. Cassinelli HR, Mautalen CA, Heinrich JJ, Miglietta A, Bergada C (1992) Familial idiopathic hyperphosphatasia (FIH): response to long- term treatment with pamidronate (APD). Bone Miner 19: 175–184

    Article  CAS  Google Scholar 

  6. Chlebna-Sokol D, Loba-Jakubowska E, Sikora A (2001) Clinical evaluation of patients with idiopathic juvenile osteoporosis. J Pediatr Orthop B 10: 259-263

    Article  CAS  Google Scholar 

  7. De Groen PC, Lubbe DF, Hirsch LJ, Daifotis A, Stephenson W, Freedholm D, Pryor Tillotson S, Seleznick MJ, Pinkas H, Wang KK (1996) Esophagitis associated with the use of alendronate (see comments). N Engl J Med 335: 1016–1021

    Article  Google Scholar 

  8. Glorieux FH (2001) The use of bisphosphonates in children with osteogenesis imperfecta. J Pediatr Endocrinol Metab 14[Suppl 6]: 1491–1495

  9. 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: 947–952

    Article  CAS  Google Scholar 

  10. Hoekman K, Papapoulos SE, Peters AC, Bilvoet OL (1985) Characteristics and bisphopsphonate treatment of a patient with juvenile osteoporosis. J Clin Endocrinol Metab 61: 952–956

    Article  CAS  Google Scholar 

  11. Jackson EC, Strife CF, Tsang RC, Marder HK (1988) Effect of calcitonin replacement therapy in idiopathic juvenile osteoporosis Am J Dis Child 142: 1237–1239

    CAS  PubMed  Google Scholar 

  12. Jelonek A, Wyrobek Z, Kruszewska M, Bromboszcz I, Zontek F, Kolodziejska Z (1975) Calcitonin therapy in juvenile idiopathic osteoporosis (in Polish). Chir Narzadow Ruchu Ortop Pol 40: 731–735

    CAS  PubMed  Google Scholar 

  13. Kauffman RP, Overton TH, Shiflett M, Jennings JC (2001) Osteoporosis in children and adolescent girls: case report of idiopathic juvenile osteoporosis and review of the literature. Obstet Gynecol Surv 56: 492–504

    Article  CAS  Google Scholar 

  14. Krassas GE (2000) Idiopathic juvenile osteoporosis. Ann N Y Acad Sci 900: 409–412

    Article  CAS  Google Scholar 

  15. Marhaug G (1993) Idiopathic juvenile osteoporosis. Scand J Rheumatol 22: 45–47

    Article  CAS  Google Scholar 

  16. Marini JC (2003) Do bisphosphonates make children’s bones better or brittle? N Engl J Med 349: 423–426

    Article  Google Scholar 

  17. Rogers MJ (2003) New insights into the molecular mechanisms of action of bisphosphonates. Curr Pharm Des 9: 2643–2658

    Article  CAS  Google Scholar 

  18. Schippers JC (1938) Over een geval van spontane algemene osteoporose bij een klein meisje. Maandschr Kindergeneeskunde 8: 107

    Google Scholar 

  19. Smith R (1995) Idiopathic juvenile osteoporosis: experience of twenty-one patients. Br J Rheumatol 34: 68–77

    Article  CAS  Google Scholar 

  20. Uttley WS, Belton NR, Syme J, Sheppard H (1975) Calcium balance in children treated with diphosphonates. Arch Dis Child 50: 187–190

    Article  CAS  Google Scholar 

  21. Whyte M, Wenkert D, Clements K, McAlister W, Mumm S (2003) Bisphosphonate-induced osteopetrosis. N Engl J Med 349: 457–463

    Article  CAS  Google Scholar 

  22. Wright NM, Metzger DL, Key LL (1995) Estrogen and diclofenac sodium therapy in a prepubertal female with idiopathic juvenile osteoporosis. J Pediatr Endocrinol Metab 8: 135–139

    Article  CAS  Google Scholar 

  23. Zacharin M, Cundy T (2000) Osteoporosis pseudoglioma syndrome: treatment of spinal osteoporosis with intravenous bisphosphonates. J Pediatr 137: 410–415

    Article  CAS  Google Scholar 

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Correspondence to Ralph Melchior.

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Melchior, R., Zabel, B., Spranger, J. et al. Effective parenteral clodronate treatment of a child with severe juvenile idiopathic osteoporosis. Eur J Pediatr 164, 22–27 (2005). https://doi.org/10.1007/s00431-004-1541-7

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  • DOI: https://doi.org/10.1007/s00431-004-1541-7

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