Treatment with neridronate in children and adolescents with osteogenesis imperfecta: Data from open-label, not controlled, three-year Italian study

Idolazzi, Luca; Fassio, Angelo; Viapiana, Ombretta; Rossini, Maurizio; Adami, Giovanni; Bertoldo, Francesco; Antoniazzi, Franco; Gatti, Davide

doi:10.1016/j.bone.2017.07.004

Cited by 14 publications

(22 citation statements)

References 34 publications

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“…It should be noted that these results are different from many studies conducted in adults with OI that demonstrate increases in bone density with BPN treatment. Although it is well known that bone in younger subjects with OI is much more responsive to BPN treatment, many previous studies have shown that bone density increases with BPN in adults with OI . The lack of a significant association between BPN treatment and LS aBMD in older patients in our study could at least in part be based on the wide range of age distribution, type of BPN used, and duration of treatment.…”

Section: Discussioncontrasting

confidence: 59%

“…Although it is well known that bone in younger subjects with OI is much more responsive to BPN treatment, many previous studies have shown that bone density increases with BPN in adults with OI. (11,(23)(24)(25)(26) The lack of a significant association between BPN treatment and LS aBMD in older patients in our study could at least in part be based on the wide range of age distribution, type of BPN used, and duration of treatment. Treatment with oral BPNs was more common among older individuals than in the preadolescent age group, with 55% of those 14 years old and older receiving oral treatment compared with 17% of treated individuals in the 0 to 14 age group.…”

Section: Bpn Use and Ls Abmd In Oi-1mentioning

confidence: 68%

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A Multicenter Observational Cohort Study to Evaluate the Effects of Bisphosphonate Exposure on Bone Mineral Density and Other Health Outcomes in Osteogenesis Imperfecta

et al. 2019

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Osteogenesis imperfecta (OI) is characterized by low bone mass and bone fragility. Using data from a large cohort of individuals with OI from the Osteogenesis Imperfecta Foundation's linked clinical research centers, we examined the association between exposure to bisphosphonate (BPN) treatment (past or present) and lumbar spine (LS) areal bone mineral density (aBMD), fractures, scoliosis, and mobility. From 466 individuals, we obtained 1394 participant‐age LS aBMD data points. Though all OI subtypes were examined, primary analyses were restricted to type I OI (OI‐1). Using linear regression, we constructed expected OI‐1 LS aBMD‐for‐age curves from the data from individuals who had never received BPN. LS aBMD in those who had been exposed to BPN was then compared with the computed expected aBMD. BPN exposure in preadolescent years (age <14 years) was associated with a LS aBMD that was 9% more than the expected computed values in BPN‐naïve individuals ( p < 0.01); however, such association was not observed across all ages. Exposure to i.v. BPN and treatment duration > 2 years correlated with LS aBMD in preadolescent individuals. BPN exposure also had a significant association with non‐aBMD clinical outcome variables. Logistic regression modeling predicted that with BPN exposure, a 1‐year increase in age would be associated with an 8.2% decrease in fracture probability for preadolescent individuals with OI‐1, compared with no decrease in individuals who had never received any BPN ( p < 0.05). In preadolescent individuals with OI‐1, a 0.1 g/cm 2 increase in LS aBMD was associated with a 10.6% decrease in scoliosis probability, compared with a 46.8% increase in the BPN‐naïve group ( p < 0.01). For the same changes in age and LS aBMD in preadolescent individuals, BPN exposure was also associated with higher mobility scores ( p < 0.01), demonstrating that BPN treatment may be associated with daily function. © 2018 The Authors. JBMR Plus Published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

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Section: Discussioncontrasting

confidence: 59%

Section: Bpn Use and Ls Abmd In Oi-1mentioning

confidence: 68%

A Multicenter Observational Cohort Study to Evaluate the Effects of Bisphosphonate Exposure on Bone Mineral Density and Other Health Outcomes in Osteogenesis Imperfecta

et al. 2019

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“…Therapy with iv bisphosphonates in children with OI JRPMS significant increase in height in patients with OI type IV 11 . Very few studies were performed with nerindronate, another iv bisphosphonate, in children and adolescents with OI [12][13] . Idolazzi et al reported significant increases of areal BMD at the lumbar spine and total hip versus baseline but no statistically significant effect on fracture risk (p=0.185), although they report a significant reduction in the mean number of fractures occurring during treatment compared to pre-treatment values 12 .…”

Section: Accepted 28 August 2018mentioning

confidence: 99%

“…Very few studies were performed with nerindronate, another iv bisphosphonate, in children and adolescents with OI [12][13] . Idolazzi et al reported significant increases of areal BMD at the lumbar spine and total hip versus baseline but no statistically significant effect on fracture risk (p=0.185), although they report a significant reduction in the mean number of fractures occurring during treatment compared to pre-treatment values 12 . Gatti et al also reported significant increases of lumbar and hip BMD but no significant effect on fracture incidence 13 .…”

Section: Accepted 28 August 2018mentioning

confidence: 99%

Treatment with intravenous bisphosphonates in children and adolescents with Osteogenesis Imperfecta: are we towards a consensus of a protocol?

Stathopoulos¹

2018

JRPMS

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“…Dies kann aus forensischen Gründen bei Verdacht auf Kindesmisshandlung ein wichtiger diagnostischer Baustein sein, ermöglicht aber auch eine genetische Beratung der Eltern und der Patienten bezüglich des Wiederholungsrisikos und der Weitervererbung und ist zukünftig vermutlich die Basis für eine zielgerichtete medikamentöse Therapie (3). Die derzeitige medikamentöse Standardtherapie bei der Osteogenesis imperfecta ist eine antiresorptive Therapie mit intravenös verabreichten Bisphosphonaten (16,17…”

Section: Genetische Untersuchungenunclassified

Diagnostik und Therapie bei Erkrankungen mit erhöhtem Frakturrisiko

Semler¹,

Rehberg²,

Stark³

et al. 2018

Kinder- und Jugendmedizin

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ZusammenfassungFrakturen treten im Kindes- und Jugendalter bei vielen Kindern auf und sind meist durch adäquate Traumata verursacht. Sie können jedoch auch im Zusammenhang mit einer Kindesmisshandlung oder als Zeichen einer systemischen Skeletterkrankung beobachtet werden. Die diagnostischen Schritte umfassen neben Anamnese und klinischer Untersuchung, bildgebende Verfahren, Laboruntersuchungen und Knochendichtemessungen. In der Kinder- und Jugendmedizin ist bei allen diesen Methoden auf die Verwendung von alters- und größenkorrelierten Normwerten zu achten. Hierdurch können primäre und sekundäre Osteoporosen sowie Mineralisierungs- Synthese- und Abbaustörungen voneinander getrennt werden. Im Falle angeborener Erkrankungen können genetische Untersuchungen zur Diagnosesicherung beitragen. Eine sichere Diagnosestellung ist erforderlich, um eine kostenbewusste Diagnostik durchzuführen und letzten Endes eine zielgerichtete Therapie initiieren zu können.

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Treatment with neridronate in children and adolescents with osteogenesis imperfecta: Data from open-label, not controlled, three-year Italian study

Cited by 14 publications

References 34 publications

A Multicenter Observational Cohort Study to Evaluate the Effects of Bisphosphonate Exposure on Bone Mineral Density and Other Health Outcomes in Osteogenesis Imperfecta

A Multicenter Observational Cohort Study to Evaluate the Effects of Bisphosphonate Exposure on Bone Mineral Density and Other Health Outcomes in Osteogenesis Imperfecta

Treatment with intravenous bisphosphonates in children and adolescents with Osteogenesis Imperfecta: are we towards a consensus of a protocol?

Diagnostik und Therapie bei Erkrankungen mit erhöhtem Frakturrisiko

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