Effect of alendronate therapy in children with osteogenesis imperfecta

Vyskocil, Vaclav; Pikner, Richard; Kutı́lek, Štěpán

doi:10.1016/j.jbspin.2004.09.005

Cited by 33 publications

(24 citation statements)

References 44 publications

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“…However, there are also reports of decreased bone formation (11,15) and increased number of trabeculae with calcified cartilageprimary spongiosa (11,15). Similar findings were observed in children treated with oral ALN (27)(28)(29). These clinical findings are supported by experimental animal studies on OI showing similar results, i.e., increased femoral and vertebral bone mineral density, bone volume, number of trabeculae (30), and cortical width (14).…”

Section: Discussionsupporting

confidence: 59%

Effect of alendronate on the mandible and long bones: an experimental study in vivo

2015

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Background:Bisphosphonates are anticatabolic agents that inhibit bone resorption and are widely used to treat osteoporosis and bone metastases in adults. They are also used in young patients with diseases like osteogenesis imperfecta or juvenile osteoporosis. Bone modeling/remodeling is elevated in growing subjects, and inhibition of osteoclastic activity has been shown to interfere with growth. Thus, our objective was to evaluate the effect of alendronate (ALN) on growing animals. Methods: Healthy male Wistar rats, aged 1 mo, received ALN or vehicle for 8 wk. Serum levels (calcemia, phosphatemia, and total alkaline phosphatase) were determined. Morphometric (rat: femur and tibia weight and length and hemimandible growth) and histomorphometric parameters (thickness of tibial epiphyseal cartilage and each cartilage zone, interradicular bone volume in the first lower molar, trabeculae volume, percentage of bone and cartilage, and osteoclast number in mandibular condyles) were assessed. results: ALN caused a significant decrease in femur and tibia length, tibial cartilage thickness, and longitudinal growth of hemimandibles. It increased interradicular bone volume and mandibular condyle trabeculae volume, increasing the percentage of cartilage and osteoclast number. conclusion: These findings indicate that administration of ALN to growing animals alters the endochondral ossification process, and thus alters growth.

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Section: Discussionsupporting

confidence: 59%

Effect of alendronate on the mandible and long bones: an experimental study in vivo

2015

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“…The effect of this on bone fragility is not known. Reassuringly, at least in the paediatric population with OI, IV pamidronate can decrease fracture incidence [6,12,33,34], and a reduction in fracture has also been observed with oral bisphosphonates in some [13,27,35] but not all [14,15,36] studies. There have been only two controlled trials of bisphosphonates in adults with OI type I.…”

Section: Discussionmentioning

confidence: 99%

Risedronate in adults with osteogenesis imperfecta type I: increased bone mineral density and decreased bone turnover, but high fracture rate persists

et al. 2011

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“…The children's mean age (± SD) at the start of therapy was 7.6±5.2 years, 9.6±5.1 years at the end of therapy, and 11.7±5.3 years at last follow-up ( Table 1). The mean length of treatment was 22±2 months (range 6-43, median 24), and that of follow-up 26±2 months (range 18-44, median 25). The cumulative dose of bisphosphonates, calculated as corrected for pamidronate [0.025 mg zoledronic acid = 1 mg pamidronate [20]] and using children's average weight between beginning and end of therapy was 14.7±8.7 mg/kg (range 2.0-37.1, median 13.4).…”

Section: Resultsmentioning

confidence: 99%

Sustained beneficial effect of intravenous bisphosphonates after their discontinuation in children

et al. 2007

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We studied if the beneficial effects of bisphosphonates are maintained after their discontinuation, and whether adverse effects may develop. Seventeen children in whom I.V. bisphosphonates were discontinued for at least 12 months were included. Fracture rate (FR), skeletal pain, bone mineral density of total body (TB) and spine L(2-4), skeletal radiographs, bone markers and kidney functions were compared between: (a) before treatment, (b) end of treatment, and (c) last follow-up. Mean treatment duration was 22+/-2 months (6-43) and follow-up 26+/-2 months (18-44). FR (mean +/- SD) decreased from 0.74+/-0.21/year before treatment to 0.35+/-0.11/year after treatment and 0.20+/-0.09/year at follow-up (p<0.05). Three children had bone pain before treatment, six during treatment and none at end of follow-up (p<0.05). TB Z-score increased from -1.24+/-0.50 at baseline to -0.37+/-0.44 at end of treatment and -0.39+/-0.37 at follow-up (p<0.05). Spinal Z-score increased from -1.65+/-0.57 to -0.34+/-0.56 and 0.19+/-0.49, respectively (p<0.05). Bone turnover markers showed sustained effect of bisphosphonates. No adverse effects on kidney functions or skeletal radiographs were noted. We conclude that I.V. bisphosphonates continue to exert their beneficial effect for a mean of 26+/-2 months after their discontinuation; therefore, once therapeutic goals are achieved, the medication can be withheld, followed by periodic re-evaluation.

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Effect of alendronate therapy in children with osteogenesis imperfecta

Cited by 33 publications

References 44 publications

Effect of alendronate on the mandible and long bones: an experimental study in vivo

Effect of alendronate on the mandible and long bones: an experimental study in vivo

Risedronate in adults with osteogenesis imperfecta type I: increased bone mineral density and decreased bone turnover, but high fracture rate persists

Sustained beneficial effect of intravenous bisphosphonates after their discontinuation in children

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