Large osteocyte lacunae in iliac crest infantile bone are not associated with impaired mineral distribution or signs of osteocytic osteolysis

Jandl, Nico Maximilian; Kroge, Simon von; Stürznickel, Julian; Baranowsky, Anke; Stockhausen, Kilian E; Mushumba, Herbert; Beil, Frank Timo; Püschel, Klaus; Amling, Michael; Rolvien, Tim

doi:10.1016/j.bone.2020.115324

Cited by 22 publications

(16 citation statements)

References 39 publications

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“…Indeed, Zimmermann et al reported a similar high osteocyte number in pediatric femoral bone [ 54 ]. In contrast, the OLS density in trabecular bone in our reference cohort is very close to the values reported by Jandl et al in trabeculae of iliac bone from six children [ 57 ]. However, we found a smaller OLS area in trabecular bone (23.63 ± 3.01 µm 2 versus 30.71 ± 4.25 µm 2 ) and in cortical bone (23.19 ± 2.24 µm 2 versus 31.70 ± 7.76 µm 2 ).…”

Section: Discussionsupporting

confidence: 90%

“…Osteocyte density in cortical bone, however, appears rather variable. In the report of Jandl et al, for example, osteocyte density was found to be twice as high than in the present study (537 ± 127 mm 2 versus 274.65 ± 60.14 mm 2 ) [57]. The reason for this discrepancy is possibly that we excluded areas of very porous primary woven bone from our measurements.…”

contrasting

confidence: 79%

“…A further interesting observation is that osteocyte lacunae density is higher in cortical than in trabecular bone, which is in accordance with a higher cortical remodeling activity during skeletal growth [ 57 , 58 , 59 ]. Osteocyte density in cortical bone, however, appears rather variable.…”

Section: Discussionmentioning

confidence: 91%

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Increased Osteocyte Lacunae Density in the Hypermineralized Bone Matrix of Children with Osteogenesis Imperfecta Type I

Mähr

Blouin

Behanová

et al. 2021

IJMS

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Osteocytes are terminally differentiated osteoblasts embedded within the bone matrix and key orchestrators of bone metabolism. However, they are generally not characterized by conventional bone histomorphometry because of their location and the limited resolution of light microscopy. OI is characterized by disturbed bone homeostasis, matrix abnormalities and elevated bone matrix mineralization density. To gain further insights into osteocyte characteristics and bone metabolism in OI, we evaluated 2D osteocyte lacunae sections (OLS) based on quantitative backscattered electron imaging in transiliac bone biopsy samples from children with OI type I (n = 19) and age-matched controls (n = 24). The OLS characteristics were related to previously obtained, re-visited histomorphometric parameters. Moreover, we present pediatric bone mineralization density distribution reference data in OI type I (n = 19) and controls (n = 50) obtained with a field emission scanning electron microscope. Compared to controls, OI has highly increased OLS density in cortical and trabecular bone (+50.66%, +61.73%; both p < 0.001), whereas OLS area is slightly decreased in trabecular bone (−10.28%; p = 0.015). Correlation analyses show a low to moderate, positive association of OLS density with surface-based bone formation parameters and negative association with indices of osteoblast function. In conclusion, hyperosteocytosis of the hypermineralized OI bone matrix associates with abnormal bone cell metabolism and might further impact the mechanical competence of the bone tissue.

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Section: Discussionsupporting

confidence: 90%

contrasting

confidence: 79%

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Increased Osteocyte Lacunae Density in the Hypermineralized Bone Matrix of Children with Osteogenesis Imperfecta Type I

Mähr

Blouin

Behanová

et al. 2021

IJMS

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“…SS1) from our archive, which were obtained in the context of two previous studies. (30,31) This study was performed in accordance with the local ethics committee (PV5364) and the Declaration of Helsinki as informed consent was given by all patients or parents.…”

Section: Methodsmentioning

confidence: 99%

Mice Carrying a Ubiquitous R235W Mutation of Wnt1 Display a Bone-Specific Phenotype

Yorgan

Rolvien

Stürznickel

et al. 2020

Journal of Bone and Mineral Research

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Since a key function of Wnt1 in brain development was established early on through the generation of non‐viable Wnt1‐deficient mice, it was initially surprising that WNT1 mutations were found to cause either early‐onset osteoporosis (EOOP) or osteogenesis imperfecta type XV (OI‐XV). The deduced function of Wnt1 as an osteoanabolic factor has been confirmed in various mouse models with bone‐specific inactivation or overexpression, but mice carrying disease‐causing Wnt1 mutations have not yet been described. Triggered by the clinical analysis of EOOP patients carrying a heterozygous WNT1 mutation (p.R235W), we introduced this mutation into the murine Wnt1 gene to address the question of whether this would cause a skeletal phenotype. We observed that Wnt1+/R235W and Wnt1R235W/R235W mice were born at the expected Mendelian ratio and that they did not display postnatal lethality or obvious nonskeletal phenotypes. At 12 weeks of age, the homozygous presence of the Wnt1 mutation was associated with reduced trabecular and cortical bone mass, explained by a lower bone formation rate compared with wild‐type littermates. At 52 weeks of age, we also observed a moderate bone mass reduction in heterozygous Wnt1+/R235W mice, thereby underscoring their value as a model of WNT1‐dependent EOOP. Importantly, when we treated wild‐type and Wnt1+/R235W mice by daily injection of parathyroid hormone (PTH), we detected the same osteoanabolic influence in both groups, together with an increased cortical thickness in the mutant mice. Our data demonstrate the pathogenicity of the WNT1‐R235W mutation, confirm that controlling skeletal integrity is the primary physiological function of Wnt1, and suggest that osteoanabolic treatment with teriparatide should be applicable for individuals with WNT1‐dependent EOOP. © 2020 American Society for Bone and Mineral Research.

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“…Other groups throughout the world have also used it to measure bone mineral content in pathological situations [33][34][35][36]. Recently, evaluation protocols were developed to also assess osteocyte lacunae number, size, and shape from qBE images [15,18,37,38]. However, it has to be emphasized that this method-in contrast to DXA and HR-pQCT-requires a bone biopsy sample.…”

Section: Introductionmentioning

confidence: 99%

Quantitative Backscattered Electron Imaging of Bone Using a Thermionic or a Field Emission Electron Source

et al. 2021

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Quantitative backscattered electron imaging is an established method to map mineral content distributions in bone and to determine the bone mineralization density distribution (BMDD). The method we applied was initially validated for a scanning electron microscope (SEM) equipped with a tungsten hairpin cathode (thermionic electron emission) under strongly defined settings of SEM parameters. For several reasons, it would be interesting to migrate the technique to a SEM with a field emission electron source (FE-SEM), which, however, would require to work with different SEM parameter settings as have been validated for DSM 962. The FE-SEM has a much better spatial resolution based on an electron source size in the order of several 100 nanometers, corresponding to an about $$10^5$$ 10 5 to $$10^6$$ 10 6 times smaller source area compared to thermionic sources. In the present work, we compare BMDD between these two types of instruments in order to further validate the methodology. We show that a transition to higher pixel resolution (1.76, 0.88, and 0.57 μm) results in shifts of the BMDD peak and BMDD width to higher values. Further the inter-device reproducibility of the mean calcium content shows a difference of up to 1 wt% Ca, while the technical variance of each device can be reduced to $$\pm 0.17$$ ± 0.17 wt% Ca. Bearing in mind that shifts in calcium levels due to diseases, e.g., high turnover osteoporosis, are often in the range of 1 wt% Ca, both the bone samples of the patients as well as the control samples have to be measured on the same SEM device. Therefore, we also constructed new reference BMDD curves for adults to be used for FE-SEM data comparison.

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Large osteocyte lacunae in iliac crest infantile bone are not associated with impaired mineral distribution or signs of osteocytic osteolysis

Cited by 22 publications

References 39 publications

Increased Osteocyte Lacunae Density in the Hypermineralized Bone Matrix of Children with Osteogenesis Imperfecta Type I

Increased Osteocyte Lacunae Density in the Hypermineralized Bone Matrix of Children with Osteogenesis Imperfecta Type I

Mice Carrying a Ubiquitous R235W Mutation of Wnt1 Display a Bone-Specific Phenotype

Quantitative Backscattered Electron Imaging of Bone Using a Thermionic or a Field Emission Electron Source

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