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Joint Bone Spine
Volume 72, n° 4
pages 313-318 (juillet 2005)
Doi : 10.1016/j.jbspin.2004.08.011
Received : 19 Mars 2004 ;  accepted : 26 August 2004
Benefits of pamidronate in children with osteogenesis imperfecta: an open prospective study
 

Véronique Forin a, , Asma Arabi b, Vincent Guigonis c, Georges Filipe a, Albert Bensman c, Christian Roux b
a Service d'Orthopédie Pédiatrique, Hôpital Armand Trousseau, Assistance Publique Hôpitaux de Paris, Université Paris VI, Paris, France 
b Service de Rhumatologie, Hôpital Cochin, Assistance Publique Hôpitaux de Paris, Université René Descartes, Paris, France 
c Service de Néphrologie Pédiatrique, Hôpital Armand Trousseau, Assistance Publique Hôpitaux de Paris, Université Paris VI, Paris, France 

*Corresponding author. Hôpital d'enfants Armand Trousseau, 26, avenue du Dr Arnold Netter, 75012 Paris, France. Tél. : +33-1-44-73-62-26; Fax : +33-1-44-73-53-09.

Data presented in part at the 8th international conference on osteogenesis imperfecta, Annecy, 2002

Abstract

Objectives.- To study the efficacy of pamidronate in children with osteogenesis imperfecta (OI).

Patients and methods.- Twenty-nine patients (median age 8.7 years), were given pamidronate in cyclic infusions of 3 days. Patients received 3-13 cycles (median 6), at a dose of 0.5 mg/kg/day in infants (below 2 years of age) and 1 mg/kg/day in children (2 years and older). The interval time between cycles was 2 months in infants and 4 months in children. The median follow-up was 16 months. All patients received daily supplementation of calcium, vitamin D and physical rehabilitation. Assessments were performed at baseline and before each cycle. Fracture rate under treatment was compared to the one in the pre-treatment period.

Results.- Pain decreased after the first infusion cycle (P < 0.0001). The median of fracture incidence decreased from 15 to 0.5 per year in infants and from 2.0 to 1 per year in children (P = 0.04). Alkaline phosphatase decreased by 31.2% and N -telopeptide collagen cross-links decreased by 61.8% (P < 0.001). Bone mineral density (BMD) of the spine increased by a median of 55.4% (P < 0.001). Z -scores increased from a median of -4.7 to -2.6 (P < 0.001). The femoral neck, BMD increased by a median of 16%. The area of the first four lumbar vertebrae increased by a median of 21.5% (P < 0.001). No adverse effect on growth or on fracture healing was observed. Side effects were symptomatic hypocalcemia in one infant, and the transient acute phase reaction.

Conclusion.- Pamidronate increases BMD, decreases bone remodeling markers, pain and fracture rate in infants and children with OI.

The full text of this article is available in PDF format.

Keywords : Children, Fracture, Osteogenesis imperfecta, Pamidronate




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