The Paradox of Prevention—Bilateral Atypical Subtrochanteric Fractures due to Bisphosphonates in Osteogenesis Imperfecta

Manolopoulos, K.; West, A T H; Gittoes, Neil

doi:10.1210/jc.2012-4195

Cited by 24 publications

(11 citation statements)

References 3 publications

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“…As with active treatment regimens, further studies are required to determine the optimal maintenance bisphosphonate treatment regimen in the management of children with OI. This is particularly important given the emerging data regarding atypical fractures and the long-term effects of bisphosphonate therapy [27]. It may be possible to utilise bone turnover markers to help guide appropriate maintenance bisphosphonate doses, but the effects of puberty, disease and recurrent fractures may make this approach challenging [28,29].…”

Section: Discussionmentioning

confidence: 99%

The Long-Term Effects of Switching from Active Intravenous Bisphosphonate Treatment to Low-Dose Maintenance Therapy in Children with Osteogenesis Imperfecta

Biggin

Zheng²,

Briody³

et al. 2015

Horm Res Paediatr

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Background/Aims: Intravenous bisphosphonate therapy is the first-line treatment in moderate-to-severe osteogenesis imperfecta (OI), but there are varied treatment protocols with little data on long-term efficacy. This study evaluates the clinical outcomes when transitioning from active bisphosphonate treatment to maintenance therapy. Methods: A retrospective review was conducted on 17 patients before treatment, following active treatment (zoledronate 0.05 mg/kg 6-monthly or pamidronate 6-9 mg/kg/year) and after establishment on maintenance treatment for more than 2 years (zoledronate 0.025 mg/kg 6-monthly or pamidronate <4 mg/kg/year). Results: There was a significant reduction in mean fracture rate from 1.5 ± 1.1 fractures/year at baseline to 0.7 ± 0.7 fractures/year on active treatment. Z-scores for lumbar spine bone mineral density, bone mineral content, volumetric bone mineral density and bone mineral content for lean tissue mass increased during active treatment. These improvements were maintained during the period of maintenance treatment. Vertebral height improved in fractured thoracic vertebrae from pre-treatment to active therapy and improved further during maintenance treatment. Metacarpal cortical thickness and relative cortical area also increased over the treatment periods. Conclusion: Maintenance intravenous bisphosphonate therapy preserved the beneficial effects of active treatment at the doses stated above. Further studies are required to determine the optimal bisphosphonate treatment regimen in the management of children with OI.

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Section: Discussionmentioning

confidence: 99%

The Long-Term Effects of Switching from Active Intravenous Bisphosphonate Treatment to Low-Dose Maintenance Therapy in Children with Osteogenesis Imperfecta

Biggin

Zheng²,

Briody³

et al. 2015

Horm Res Paediatr

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“…Moreover, long-term use of antiresorptive treatment may be associated with an increased risk of atypical femoral fractures in patients with OI and osteoporosis (6). Therefore, it is important to further investigate anabolic treatment options for adults with OI.…”

Section: Current Therapiesmentioning

confidence: 99%

Osteogenesis imperfecta in adults

Bishop¹,

Walsh²

2014

J. Clin. Invest.

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A 42-year-old premenopausal woman with osteogenesis imperfecta presents to the metabolic bone clinic. She has a daughter with osteogenesis imperfecta who is seen regularly in a specialist pediatric clinic, but the patient herself hasn't had a clinical consultation in years. She has pain and stiffness in her back and is worried for her future bone health. The patient asks, "Am I going to fall apart?" She had numerous fractures in childhood, including fractures of her femur and wrist; fractured her ankles several times in her late teens; and had occasional fractures in adulthood. Her last fracture was a comminuted fracture of her humerus three years ago, when she stumbled and fell forward onto her hands and knees. The woman is hyperextensible and thinks her ankles feel weak. Her bone mineral density T scores are -2.6 at the lumbar spine and -1.9 at the total hip, and spine imaging shows several vertebral endplate deformities, but overall preservation of vertebral height. What are the available pharmacological and nonpharmacological strategies to preserve her skeletal health and function?

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“…The fact that many of these patients have received therapy with bisphosphonates, some of them over several years of treatment, means that they are at risk of suffering this type of fracture. However, there are few references in the literature to the presence of an atypical fracture in the context of treatment for this disease (8)(9)(10)(11). In this paper, the clinical case is presented of a young male patient diagnosed with osteogenesis imperfecta and who received treatment with bisphosphonates for several years, developing over time an atypical diaphyseal femoral fracture which was treated surgically by means of an intramedullary nailing and with later pharmacological treatment with teriparatide, with a satisfactory outcome at one year follow-up.…”

Section: Introductionmentioning

confidence: 99%

An atypical fracture in male patient with osteogenesis imperfecta

Etxebarría-Foronda

Carpintero

2015

ccmbm

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SummarySo-called atypical fractures have been related to prolonged treatment with bisphosphonates. Although there remain unanswered questions with respect to their etiology and physiopathology, it does appear to be a causal relationship. There are many references in the literature about this problem in patients in whom these drugs have been used to treat osteoporosis, but few reports in patients who have received this therapy for the management of osteogenesis imperfecta. The Authors describe a case of a young male patient with osteogenesis imperfecta with a number of historical fractures, and who received treatment with these drugs, initially parenterally and subsequently orally, presenting as a complication of the treatment, an atypical diaphyseal femoral fracture. The characteristics of the fracture are consistent with the updated diagnostic criteria of the American Society for Bone and Mineral Research. The clinical case, its treatment, both surgically and metabolically with teriparatide, and its development over a year, are analysed. The case is notable for, on the one hand, the significance of the presence of this type of fracture in a young patient with this disease, and on the other, because of the administration of teriparatide outside its established clinical indications, with twin objectives: to improve the bone structure of the patient's underlying disease, and to counteract the harmful effects which bisphosphonates may have on this bone.

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The Paradox of Prevention—Bilateral Atypical Subtrochanteric Fractures due to Bisphosphonates in Osteogenesis Imperfecta

Cited by 24 publications

References 3 publications

The Long-Term Effects of Switching from Active Intravenous Bisphosphonate Treatment to Low-Dose Maintenance Therapy in Children with Osteogenesis Imperfecta

The Long-Term Effects of Switching from Active Intravenous Bisphosphonate Treatment to Low-Dose Maintenance Therapy in Children with Osteogenesis Imperfecta

Osteogenesis imperfecta in adults

An atypical fracture in male patient with osteogenesis imperfecta

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