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Joint Bone Spine. 2005 Jul;72(4):313-8. Epub 2004 Dec 15.

Benefits of pamidronate in children with osteogenesis imperfecta: an open prospective study.

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1
Service d'Orthopédie Pédiatrique, Hôpital Armand Trousseau, Assistance Publique Hôpitaux de Paris, Université Paris VI, Paris, France. veronique.forin@trs.ap-hop-paris.fr

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.

PMID:
16038843
DOI:
10.1016/j.jbspin.2004.08.011
[Indexed for MEDLINE]
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