High-resolution peripheral quantitative computed tomography in children with osteogenesis imperfecta
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High-resolution peripheral quantitative computed tomography in children with osteogenesis imperfecta David J. Fennimore 1 & Maria Digby 1 & Margaret Paggiosi 2 & Paul Arundel 3 & Nick J. Bishop 1,3 & Paul Dimitri 1,3 & Amaka C. Offiah 1,3 Received: 13 November 2019 / Revised: 5 April 2020 / Accepted: 22 May 2020 # The Author(s) 2020
Abstract Bone health in children with osteogenesis imperfecta is monitored using radiographs and dual-energy X-ray absorptiometry, which have limitations. High-resolution peripheral quantitative CT can non-invasively derive bone microarchitectural data. Children with severe osteogenesis imperfecta have fragile deformed bones, and positioning for this scan can be difficult. We assessed the feasibility of high-resolution peripheral quantitative CT in nine children aged 9–15 years with osteogenesis imperfecta and compared results with dual-energy X-ray absorptiometry and with healthy controls. All nine recruited children were successfully scanned and showed no preference for either modality. It therefore appears feasible to perform high-resolution peripheral quantitative CT in children with osteogenesis imperfecta aged 9 years and older. Future studies should focus on understanding the clinical implications of the technology in this patient cohort. Keywords Bone mechanical properties . Bone microstructure . Children . Dual-energy X-ray absorptiometry . Feasibility study . High-resolution peripheral quantitative computed tomography . Osteogenesis imperfecta
Description Bone quality encompasses all material and geometric factors contributing to fracture resistance [1]. A decrease in bone strength results in greater risk of fracture. Osteogenesis imperfecta is characterised by defective Type I collagen and fragile bones that frequently fracture. There are various types of osteogenesis imperfecta, ranging in severity from mild to perinatally lethal. Routine monitoring of these children varies among centres but usually includes 6-monthly dual-energy Xray absorptiometry (for bone mineral density assessment) and either annual lateral spine radiographs or 6-monthly lateral dual-energy X-ray absorptiometry of the spine (for detecting
* Amaka C. Offiah [email protected] 1
Academic Unit of Child Health, University of Sheffield, Damer Street, Sheffield S10 2TH, UK
2
The Mellanby Centre for Bone Research, Academic Unit of Bone Metabolism, Department of Oncology and Metabolism, The University of Sheffield, Sheffield, UK
3
Sheffield Children’s NHS Foundation Trust, Western Bank, Sheffield S10 2TH, UK
vertebral fractures). Given its limitations in assessing bone density, conventional radiography is only performed when fracture or other pathology (e.g., basilar invagination) is suspected. The International Society for Clinical Densitometry states that radiologic investigations should be used to identify children who might benefit from clinical intervention [2]. Dualenergy X-ray absorptiometry is a low-cost technique that exposes children to low radiation dose. However,
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