HRpQCT in Osteogenesis Imperfecta
Research type
Research Study
Full title
Employing High-Resolution peripheral Quantitative Computed Tomography (HRpQCT) towards a better understanding of children’s bone geometry and strength in osteogenesis imperfecta
IRAS ID
154418
Contact name
Amaka Offiah
Contact email
Sponsor organisation
Sheffield Children's Hospital NHS Foundation Trust
Duration of Study in the UK
0 years, 11 months, 31 days
Research summary
Summary of Research
The UK lags behind North America and Europe in terms of research into children’s bone health using high resolution peripheral quantitative computed tomography (HRpQCT). The current gold standard for assessing children’s bone strength is based on measuring how dense they are with dual energy x-ray absorptiometry (DXA).
DXA is difficult to use in children below four years of age (limited reference data exists for current instruments) and DXA is poor at predicting future fracture risk at any age (Including adults).
We identify a wide range of research projects that could be addressed with HRpQCT.
The first of these we would like to address is a test of the feasibility of imaging children with fragile bones due to a disease called osteogenesis imperfecta (OI). These children may have significant bone deformities following multiple fractures. Sheffield Children’s Hospital SCH), under the leadership of Professor Nick Bishop is recognised as a leading centre for the treatment of children with OI. Currently children with OI are being monitored with DXA. We would like to monitor them with HRpQCT. We want to see if, despite their deformities, surgery (including the insertion of metallic rods into the shafts of their bones to prevent further fracture) and treatment with bisphosphonates, we can obtain images of diagnostic quality. If so, we would perform larger definitive studies to ascertain whether HRpQCT parameters can be used as an objective and reliable end point for managing children with severe OI. We do not have the required scanner at SCH, therefore for this study, children and their families will travel to the Northern General Hospital (approximately 5 miles away from SCH) to have their scan.
It is our plan in the future, to apply for external funding to purchase our own HRpQCT scanner as part of an NIHR or MRC programme grant.Summary of Results
Background: Bone health in children with osteogenesis imperfecta is monitored using radiographs and dual energy x-ray absorptiometry (DXA), which have limitations. High resolution peripheral quantitative computed tomography (HRpQCT) can non-invasively derive bone microarchitectural data. Children with severe osteogenesis imperfecta have fragile deformed bones and positioning for HRpQCT may be difficult.
Objectives: To assess the feasibility of HRpQCT in children with osteogenesis imperfecta.
Materials and Methods: Prospective recruitment of children attending an osteogenesis imperfecta clinic. DXA and HRpQCT (ultra-distal radii and tibiae) of patients were compared with those of healthy age and sex-matched controls. Statistical analysis used non-parametric tests.
Results: We recruited nine patients, median age ten years eight months, seven with mild and two with severe osteogenesis imperfecta, two bisphosphonate naïve and seven previously or currently receiving bisphosphonates. All were successfully scanned using HRpQCT; seven of 33 scans were repeated due to movement artefact. DXA showed no significant differences between groups. HRpQCT-derived radial trabecular inhomogeneity, trabecular Von Mises stress and cortical Von Mises stress were 0.191mm, 3.85MPa and 7.88MPa respectively in patients and 0.164mm, 4.93MPa and 8.54MPa respectively in controls (p<0.05 for all). Cortical thickness, area and volumetric bone mineral density were 0.43mm, 25.4mm2 and 251.0mgHA/cm3 respectively in bisphosphonate treated patients and 0.24mm, 12.4mm2 and 214.6mgHA/cm3 respectively in untreated patients (p=0.046 for all). HRpQCT scanning was deemed acceptable by children, with no preference for DXA or HRpQCT.
Conclusion: It is feasible to perform HRpQCT in osteogenesis imperfecta patients over eight years old. HRpQCT provides more detailed information than DXA.REC name
Yorkshire & The Humber - Leeds West Research Ethics Committee
REC reference
14/YH/1050
Date of REC Opinion
1 Sep 2014
REC opinion
Further Information Favourable Opinion