Bisphosphonate therapy for osteogenesis imperfecta

Efficacy and Safety of Bisphosphonate Therapy in Children with Osteogenesis Imperfecta: A Systematic Review

“…None of the trials reported increased fractures with BP treatment. These results are generally in line with the review of Dwan et al [39], although they included adult studies and noncontrolled studies as well. Though promising, the reduction in fracture incidence as a result of BP treatment is still not fully understood [40].…”

“…Similar results have also been seen in several uncontrolled trials reporting a reduction in (bone) pain [45,46,47,48]. However, our results in children deviated from the review of Dwan et al [39] in which a reduction in bone pain was found in merely 1 out of 6 studies; however, they included adult studies and studies comparing different doses of BP without control groups [39]. Two included studies in our review did not measure a significant reduction in (bone) pain; however, the patients in these studies already reported a low pain score at baseline [19,33].…”

Efficacy and Safety of Bisphosphonate Therapy in Children with Osteogenesis Imperfecta: A Systematic Review

“…None of the trials reported increased fractures with BP treatment. These results are generally in line with the review of Dwan et al [39], although they included adult studies and noncontrolled studies as well. Though promising, the reduction in fracture incidence as a result of BP treatment is still not fully understood [40].…”

“…Similar results have also been seen in several uncontrolled trials reporting a reduction in (bone) pain [45,46,47,48]. However, our results in children deviated from the review of Dwan et al [39] in which a reduction in bone pain was found in merely 1 out of 6 studies; however, they included adult studies and studies comparing different doses of BP without control groups [39]. Two included studies in our review did not measure a significant reduction in (bone) pain; however, the patients in these studies already reported a low pain score at baseline [19,33].…”

Craniofacial Effects of Zoledronic Acid on the Osteogenesis Imperfecta Mouse (−/−) Model of Severe Osteogenesis Imperfecta

“…The effects of the treatment on osteocytes could also explain this positive trend of collagen matrix organization. BPs are administered in OI patients to increase bone quantity by inhibiting osteoclastic activity, thereby increasing the total mineral density [6]. Most studies on the effects of ZA have focused on long bones, but our study observed similar effects on intramembranous bones in a mouse model of OI.…”

Outcomes of Zoledronic Acid Use in Paediatric Conditions

“…For bone fragility conditions, the study’s treated patients demonstrated improved BMD for primary bone fragility (OI) as previously reported in the literature [1, 8, 13, 15, 19] and at least maintenance of BMD in the face of the continued adverse health of immobility, compared with some high-dose, long-term glucocorticoid-treated conditions, where both cytokine activity and pubertal delay were major contributors to relatively less response. The influence of puberty on bone mass accrual is well known, with a major reduction in fracture frequency after puberty recognized for all types of OI as a consequence of increased cortical accrual and trabecular mineralization.…”

Efficacy and Safety of Bisphosphonate Therapy in Children with Osteogenesis Imperfecta: A Systematic Review

“…None of the trials reported increased fractures with BP treatment. These results are generally in line with the review of Dwan et al [39], although they included adult studies and noncontrolled studies as well. Though promising, the reduction in fracture incidence as a result of BP treatment is still not fully understood [40].…”

“…Similar results have also been seen in several uncontrolled trials reporting a reduction in (bone) pain [45,46,47,48]. However, our results in children deviated from the review of Dwan et al [39] in which a reduction in bone pain was found in merely 1 out of 6 studies; however, they included adult studies and studies comparing different doses of BP without control groups [39]. Two included studies in our review did not measure a significant reduction in (bone) pain; however, the patients in these studies already reported a low pain score at baseline [19,33].…”

Craniofacial Effects of Zoledronic Acid on the Osteogenesis Imperfecta Mouse (−/−) Model of Severe Osteogenesis Imperfecta

“…The effects of the treatment on osteocytes could also explain this positive trend of collagen matrix organization. BPs are administered in OI patients to increase bone quantity by inhibiting osteoclastic activity, thereby increasing the total mineral density [6]. Most studies on the effects of ZA have focused on long bones, but our study observed similar effects on intramembranous bones in a mouse model of OI.…”

Outcomes of Zoledronic Acid Use in Paediatric Conditions

“…For bone fragility conditions, the study’s treated patients demonstrated improved BMD for primary bone fragility (OI) as previously reported in the literature [1, 8, 13, 15, 19] and at least maintenance of BMD in the face of the continued adverse health of immobility, compared with some high-dose, long-term glucocorticoid-treated conditions, where both cytokine activity and pubertal delay were major contributors to relatively less response. The influence of puberty on bone mass accrual is well known, with a major reduction in fracture frequency after puberty recognized for all types of OI as a consequence of increased cortical accrual and trabecular mineralization.…”

Efficacy and Safety of Bisphosphonate Therapy in Children with Osteogenesis Imperfecta: A Systematic Review

“…None of the trials reported increased fractures with BP treatment. These results are generally in line with the review of Dwan et al [39], although they included adult studies and noncontrolled studies as well. Though promising, the reduction in fracture incidence as a result of BP treatment is still not fully understood [40].…”

“…Similar results have also been seen in several uncontrolled trials reporting a reduction in (bone) pain [45,46,47,48]. However, our results in children deviated from the review of Dwan et al [39] in which a reduction in bone pain was found in merely 1 out of 6 studies; however, they included adult studies and studies comparing different doses of BP without control groups [39]. Two included studies in our review did not measure a significant reduction in (bone) pain; however, the patients in these studies already reported a low pain score at baseline [19,33].…”

Craniofacial Effects of Zoledronic Acid on the Osteogenesis Imperfecta Mouse (−/−) Model of Severe Osteogenesis Imperfecta

“…The effects of the treatment on osteocytes could also explain this positive trend of collagen matrix organization. BPs are administered in OI patients to increase bone quantity by inhibiting osteoclastic activity, thereby increasing the total mineral density [6]. Most studies on the effects of ZA have focused on long bones, but our study observed similar effects on intramembranous bones in a mouse model of OI.…”

Outcomes of Zoledronic Acid Use in Paediatric Conditions

“…For bone fragility conditions, the study’s treated patients demonstrated improved BMD for primary bone fragility (OI) as previously reported in the literature [1, 8, 13, 15, 19] and at least maintenance of BMD in the face of the continued adverse health of immobility, compared with some high-dose, long-term glucocorticoid-treated conditions, where both cytokine activity and pubertal delay were major contributors to relatively less response. The influence of puberty on bone mass accrual is well known, with a major reduction in fracture frequency after puberty recognized for all types of OI as a consequence of increased cortical accrual and trabecular mineralization.…”