Pamidronate Treatment of Less Severe Forms of Osteogenesis Imperfecta in Children

Zacharin, Margaret; Kanumakala, Shankar

doi:10.1515/jpem.2004.17.11.1511

Cited by 36 publications

(28 citation statements)

References 43 publications

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“…One explanation might be improved vertebral morphology, thus leading to prevention of vertebral compression fractures. Besides us [3], other investigators have indicated that the vertebral shape in OI patients normalizes during pamidronate treatment [5,23,24]. In the current study, we detected positive associations between pamidronate treatment and increased growth of upper body segment which is supported by a previous study [25] showing an association between pamidronate treatment and increased vertebral height ratio.…”

Section: Discussionsupporting

confidence: 91%

Intravenous Pamidronate Treatment Improves Growth in Prepubertal Osteogenesis Imperfecta Patients

Heino¹,

Albrecht²,

Laurenčikas³

et al. 2011

Horm Res Paediatr

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Background/Aims: Pamidronate is widely used to treat pediatric patients with osteogenesis imperfecta (OI). We aimed at delineating the effects of monthly pamidronate therapy on the growth of different body segments in prepubertal OI patients. Methods: The study included 14 prepubertal patients (12 boys, 2 girls) with mild forms of OI (type I and IV). The mean age at treatment start was 7:8 years:months (3:7–11:0). Pamidronate was given as monthly intravenous infusions. The patients were measured 1 year before, at treatment start and 1 and 2 years after treatment start. Results: Height standard deviation score (SDS) and sitting height SDS significantly increased (p < 0.05) during the first year of treatment when compared to the pre-treatment year. No further improvement was detected during the second year of treatment. However, when plotted on disease-specific growth charts (untreated patients with the same OI types), height gain was significant during the first (p < 0.001) and second (p < 0.05) years of treatment. All patients increased their bone mineral density throughout the follow-up. Conclusion: Monthly pamidronate improves the growth of prepubertal patients with mild OI, where the most prominent growth stimulation is seen in the upper body segment.

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Section: Discussionsupporting

confidence: 91%

Intravenous Pamidronate Treatment Improves Growth in Prepubertal Osteogenesis Imperfecta Patients

Heino¹,

Albrecht²,

Laurenčikas³

et al. 2011

Horm Res Paediatr

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“…In other randomised controlled trials with bisphosphonate therapy, an increased BMD and decreased fracture incidence did not clearly influence pain level as well [18,30]. On the other hand, in several uncontrolled studies in children with OI treated with bisphosphonates, beneficial effects in terms of pain level as measured by a VAS score were reported [20,23,31,41]. In these non-controlled trials the beneficial effect of bisphosphonate on pain level was explained by a decreased fracture incidence [20,23,31,41].…”

Section: Discussionmentioning

confidence: 88%

“…On the other hand, in several uncontrolled studies in children with OI treated with bisphosphonates, beneficial effects in terms of pain level as measured by a VAS score were reported [20,23,31,41]. In these non-controlled trials the beneficial effect of bisphosphonate on pain level was explained by a decreased fracture incidence [20,23,31,41]. Within our treatment group a very slight significant decrease in pain level was measured at the end of the study; however, at six months' interval regression coefficients did not differ between the two groups.…”

Section: Discussionmentioning

confidence: 99%

Quality of life in children with osteogenesis imperfecta treated with oral bisphosphonates (Olpadronate): a 2-year randomized placebo-controlled trial

et al. 2007

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In this double-blind randomised placebo-controlled trial it was investigated during a two-year follow-up whether oral bisphosphonates (Olpadronate 10 mg/m 2 /day) influence quality of life in children with osteogenesis imperfecta (OI). Thirty-four children with OI (classified according to Sillence criteria), aged 3 to 18 years of age, with a restricted level of ambulation were included. Randomisation was performed using a list of computer generated random numbers to allocate patients to receive Olpadronate or placebo. Quality of life was measured using self-perception profile for children (SPPC) and health-utility index (HUI). Differences between baseline measurements and measurements at two years follow-up were analysed within the Olpadronate and placebo group using a student's t-test. Differences in HUI and SPPC regression coefficients were analysed by random-effects repeated measures analysis (SAS, Proc Mixed, version 8.2), adjusted for age, gender and type of OI. Within the Olpadronate group there was a significant decrease in pain utility; however, difference in six months' regression coefficients between the placebo and Olpadronate group were not significant. Within the placebo group there was a significant increase in scholastic competence and behavioural conduct. The item behavioural conduct showed a steeper annual regression coefficient favourable for the placebo group. In the other SPPC items none of the annual regression coefficients showed a significant difference between the Olpadronate and the placebo group. Conclusion: We found only slight differences in quality of life in favour of the bisphosphonate group. A small but not significant decrease in pain was detected in the bisphosphonate group.

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“…Its beneficial effects on bone mineral densities (BMD) in the lumbar spine, hips and whole body have been reported in several studies [6,7,8,9,10,11]. There is also increasing evidence for its beneficial effects on patients’ growth, well-being and bone pain [7,12,13].…”

Section: Introductionmentioning

confidence: 99%

Zoledronic Acid Treatment in Children with Osteogenesis Imperfecta

et al. 2011

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Background: Intravenous disodium pamidronate has become an established treatment in osteogenesis imperfecta (OI). Another bisphosphonate, zoledronic acid, has been indicated for the treatment of adult osteoporosis. We studied its efficacy and safety in children with mild OI. Methods: Patients were treated for 1.0–3.2 years with 0.05 mg/kg zoledronic acid intravenously every 6 months as part of their clinical care. They were carefully followed for clinical and biochemical parameters, side effects, bone mineral densities (BMD) and compression fractures. Results: The study included 17 patients (age 1.5–16.8 years) with type I OI. They had sustained altogether 73 fractures; 9 had compression fractures. During the treatment, 6 patients suffered in total 10 new long-bone fractures. The median lumbar spine areal BMD z-score increased from –2.0 to –0.7 during 2 years of treatment. The infusions were associated with a transient decrease in serum calcium and phosphate and a significant increase in serum PTH. Two patients developed symptomatic hypocalcemia. Bone turnover markers decreased during the treatment. Conclusions: Intravenous zoledronic acid is an effective mode of treatment in children with OI. The treatment response is comparable to pamidronate but the infusion protocol is more convenient. Further studies are needed to establish optimal dosing and long-term safety.

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Pamidronate Treatment of Less Severe Forms of Osteogenesis Imperfecta in Children

Cited by 36 publications

References 43 publications

Intravenous Pamidronate Treatment Improves Growth in Prepubertal Osteogenesis Imperfecta Patients

Intravenous Pamidronate Treatment Improves Growth in Prepubertal Osteogenesis Imperfecta Patients

Quality of life in children with osteogenesis imperfecta treated with oral bisphosphonates (Olpadronate): a 2-year randomized placebo-controlled trial

Zoledronic Acid Treatment in Children with Osteogenesis Imperfecta

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