Bone structure assessed by HR-pQCT, TBS and DXL in adult patients with different types of osteogenesis imperfecta

Kocijan, Roland; Muschitz, Christian; Haschka, Judith; Hans, Didier; Nia, Arastoo; Geroldinger, Angelika; Ardelt, Michael; Wakolbinger, Robert; Resch, Heinrich

doi:10.1007/s00198-015-3156-4

Cited by 50 publications

(52 citation statements)

References 39 publications

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“…For example, several groups have compared adults with osteogenesis imperfecta (OI) to apparently healthy subjects and showed severe trabecular deficits in radial and tibial parameters, especially in Tb.vBMD (−21% to −38.5%), Tb.N (−21.4% to −49.4%), and Tb.Sp. (+54.5% to +77.8%) . Although alterations were more severe in moderate–severe OI (type III/IV) than in mild OI (type I), the trabecular deterioration in all types of OI exceeded that observed in fracture subjects in the current study.…”

Section: Discussioncontrasting

confidence: 61%

“…(+54.5% to +77.8%). (64)(65)(66) Although alterations were more severe in moderate-severe OI (type III/IV) than in mild OI (type I), (65) the trabecular deterioration in all types of OI exceeded that observed in fracture subjects in the current study. Recently, Rolvien and colleagues used HR-pQCT to compare bone microarchitecture in adults with OI and sex-and agematched subjects with early-onset osteoporosis (EOOP; a condition distinct from postmenopausal osteoporosis in that it is characterized by low bone mass and the occurrence of fragility fractures before the age of 50 years) and healthy subjects.…”

Section: Hr-pqct and Bone Diseasescontrasting

confidence: 61%

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HR-pQCT Measures of Bone Microarchitecture Predict Fracture: Systematic Review and Meta-Analysis

Mikolajewicz

Bishop

Burghardt

et al. 2019

Journal of Bone and Mineral Research

106

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High‐resolution peripheral quantitative computed tomography (HR‐pQCT) is a noninvasive imaging modality for assessing volumetric bone mineral density (vBMD) and microarchitecture of cancellous and cortical bone. The objective was to (1) assess fracture‐associated differences in HR‐pQCT bone parameters; and (2) to determine if HR‐pQCT is sufficiently precise to reliably detect these differences in individuals. We systematically identified 40 studies that used HR‐pQCT (39/40 used XtremeCT scanners) to assess 1291 to 3253 and 3389 to 10,687 individuals with and without fractures, respectively, ranging in age from 10.9 to 84.7 years with no comorbid conditions. Parameters describing radial and tibial bone density, microarchitecture, and strength were extracted and percentage differences between fracture and control subjects were estimated using a random effects meta‐analysis. An additional meta‐analysis of short‐term in vivo reproducibility of bone parameters assessed by XtremeCT was conducted to determine whether fracture‐associated differences exceeded the least significant change (LSC) required to discern measured differences from precision error. Radial and tibial HR‐pQCT parameters, including failure load, were significantly altered in fracture subjects, with differences ranging from −2.6% (95% confidence interval [CI] −3.4 to −1.9) in radial cortical vBMD to −12.6% (95% CI −15.0 to −10.3) in radial trabecular vBMD. Fracture‐associated differences reported by prospective studies were consistent with those from retrospective studies, indicating that HR‐pQCT can predict incident fracture. Assessment of study quality, heterogeneity, and publication biases verified the validity of these findings. Finally, we demonstrated that fracture‐associated deficits in total and trabecular vBMD and certain tibial cortical parameters can be reliably discerned from HR‐pQCT‐related precision error and can be used to detect fracture‐associated differences in individual patients. Although differences in other HR‐pQCT measures, including failure load, were significantly associated with fracture, improved reproducibility is needed to ensure reliable individual cross‐sectional screening and longitudinal monitoring. In conclusion, our study supports the use of HR‐pQCT in clinical fracture prediction. © 2019 American Society for Bone and Mineral Research.

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Section: Discussioncontrasting

confidence: 61%

Section: Hr-pqct and Bone Diseasescontrasting

confidence: 61%

HR-pQCT Measures of Bone Microarchitecture Predict Fracture: Systematic Review and Meta-Analysis

Mikolajewicz

Bishop

Burghardt

et al. 2019

Journal of Bone and Mineral Research

106

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“…Altered bone microstructure and bone geometry, evaluated by HR‐pQCT, has been reported in patients with OI types I, III, and IV 28, 29. However, in two mosaic carriers, normal skeletal growth, bone density, and bone histology were found in spite of 40–75% burden of osteoblasts heterozygous for a COL1A1 mutation 30, which is concordant with our findings.…”

Section: Discussionsupporting

confidence: 92%

Asymptomatic parental mosaicism for osteogenesis imperfecta associated with a new splice site mutation in COL1A2

Frederiksen

Dunø

Johnsen

et al. 2016

Clinical Case Reports

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“…Surprisingly, in addition to significant relationships between TBS and trabecular microarchitecture, TBS was also related to cortical parameters and whole bone rather than trabecular stiffness. In contrast, in a cohort of patients with osteogenesis imperfecta, Kocijan et al (27) reported that TBS was related to trabecular but not cortical parameters. In our cross-sectional analyses, we found that, in patients with kidney disease before transplantation, TBS was related to trabecular microarchitecture and wholebone stiffness and failure load.…”

Section: Discussionmentioning

confidence: 88%

Spine Trabecular Bone Score as an Indicator of Bone Microarchitecture at the Peripheral Skeleton in Kidney Transplant Recipients

Luckman

Hans

Cortez³

et al. 2017

CJASN

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Bone structure assessed by HR-pQCT, TBS and DXL in adult patients with different types of osteogenesis imperfecta

Abstract: Trabecular BMD and trabecular bone microstructure in particular are severely altered in patients with clinical OI-I and OI-III-IV. Low TBS and DXL and their significant associations to HR-pQCT parameters of trabecular bone support this conclusion.

Cited by 50 publications

References 39 publications

HR-pQCT Measures of Bone Microarchitecture Predict Fracture: Systematic Review and Meta-Analysis

HR-pQCT Measures of Bone Microarchitecture Predict Fracture: Systematic Review and Meta-Analysis

Asymptomatic parental mosaicism for osteogenesis imperfecta associated with a new splice site mutation in COL1A2

Spine Trabecular Bone Score as an Indicator of Bone Microarchitecture at the Peripheral Skeleton in Kidney Transplant Recipients

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