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  • 1
    Electronic Resource
    Electronic Resource
    The Microscopic Structure of Bone in Normal Children and Patients with Osteogenesis Imperfecta: A Survey Using Backscattered Electron Imaging (1999)
    Springer
    Calcified tissue international 64 (1999), S. 8-17 
    Details
    ISSN: 1432-0827
    Keywords: Key words: Osteogenesis imperfecta — Normal bone — Children — Backscattered electron imaging scanning electron microscopy.
    Source: Springer Online Journal Archives 1860-2000
    Topics: Biology , Medicine , Physics
    Notes: Abstract. The microstructure of iliac crest biopsies from normal children or from those afflicted with osteogenesis imperfecta (OI) has not previously been studied to determine the tissue histology in the context of the degree of mineralization. The material in this study comprised 112 iliac crest biopsies from children aged 1.9–22.9 years. Fifty-eight were reference biopsies taken from children with no bone disease and the remainder were biopsies from children diagnosed as having OI (23 were Type I, 8 Type III, 18 Type IV, and 5 Type V). The specimens, which had been embedded in polymethylmethacrylate (PMMA), were micromilled and carbon coated to permit backscattered electron imaging. Reference biopsies from very young children often contained densely mineralized cartilage, and evidence of rapid cortical drift. Circumferential lamellae became a prominent feature after the toddler stage, and active remodeling and slower cortical drift continued through childhood. The biopsies from older teenagers and young adults were indistinguishable. Occasional mineralized osteocyte lacunae were detected in even the youngest children. Bone from children with OI Type I often appeared normal in microstructure and amount, but in some there was a dearth of bone and an abundance of osteocytes. Compared with age-matched controls, cortical and trabecular bone from children with OI Types III and IV were markedly sparse and very cellular, and primary osteonal systems continued to be formed later than expected. A distinguishing feature of the bone from OI Type V patients was the failure of patches of bone to mineralize, especially adjoining a reversal line. Packets of bone tissue exhibiting either considerably higher than normal or deficient mineralization would contribute to the characteristic trait of mechanical weakness.
    Type of Medium: Electronic Resource
    URL: http://dx.doi.org/10.1007/s002239900571
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  • 2
    Electronic Resource
    Electronic Resource
    The Mineralization Density of Iliac Crest Bone from Children with Osteogenesis Imperfecta (1999)
    Springer
    Calcified tissue international 64 (1999), S. 185-190 
    Details
    ISSN: 1432-0827
    Keywords: Key words: Bone density — Osteoporosis — Osteopenia — Osteogenesis imperfecta — Quantitative backscattered electron image analysis.
    Source: Springer Online Journal Archives 1860-2000
    Topics: Biology , Medicine , Physics
    Notes: Abstract. We studied iliac crest biopsy cores taken from young individuals with osteogenesis imperfecta of several types, and from age-matched normals; the same samples had been used in prior studies using conventional light microscopic histomorphometric procedures. The PMMA blocks were micro-milled to a fine finish, carbon coated, and imaged using backscattered electrons (BSE) in an automated digital scanning electron microscope (SEM). For comparison of BSE signal levels between samples, microscope operation parameters were standardized by reference to halogenated dimethacrylate standards, and recording data from stereological arrays of 512*512 nonoverlapping pixels at 3.5 μm separation. All OI types showed higher average mineralization densities than age- and site-matched normals. This is interpreted as the result of the failure in matrix assembly, such that it has a higher water volume fraction available for mineral deposition. Added to the net deficit in bone quantity, the predicted higher stiffness of the more mineralized bone will account for much of the observed `brittleness' that characterizes this class of genetic disease. The mean mineralization density, which was higher in types III, IV, and V than in type I, appears to be correlated with disease severity.
    Type of Medium: Electronic Resource
    URL: http://dx.doi.org/10.1007/s002239900600
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  • 3
    Electronic Resource
    Electronic Resource
    Bisphosphonate (2000)
    Springer
    Monatsschrift Kinderheilkunde 148 (2000), S. 334-341 
    Details
    ISSN: 1433-0474
    Keywords: Schlüsselwörter Bisphosphonate ; Knochenstoffwechsel ; Metabolische Osteopathien ; Osteogenesis imperfecta ; Key words Bisphosphonates ; Bone turnover ; Metabolic osteopathy ; Osteogenesis imperfecta
    Source: Springer Online Journal Archives 1860-2000
    Topics: Medicine
    Description / Table of Contents: Summary Bisphosphonates are a class of potent inhibitors of bone resorption which are increasingly used in pediatrics. Potential applications tested so far include short term treatment of hypercalcemia and continuous therapy in disorders with decreased bone mass (osteogenesis imperfecta, idiopathic juvenile osteoporosis, secondary osteoporosis) or increased bone turnover (hyperphosphatasia, fibrous dysplasia). The efficacy of bisphosphonates is best documented in the treatment of osteogenesis imperfecta, where they were shown to decrease fracture rate and to increase mobility. While these results are very encouraging, very little is known on the effect of long term bisphosphonate treatment on bone development. This remains the most critical aspect of their use in pediatrics. Therefore, pediatric use of bisphosphonates should at present be limited to specialised centers.
    Notes: Zusammenfassung Medikamente aus der Klasse der Bisphosphonate sind sehr wirksame Inhibitoren der Knochenresorption, die in jüngster Zeit zunehmend auch in der Pädiatrie Verwendung finden. Bisher beschriebene Einsatzgebiete sind die Akuttherapie von Hyperkalzämiezuständen und die Dauertherapie von Erkrankungen, welche mit erniedrigter Knochenmasse (Osteogenesis imperfecta, idiopathische juvenile Osteoporose, sekundäre Osteoporose) oder erhöhtem Knochenumsatz (Hyperphosphatasie, fibröse Dysplasie) einhergehen. Am besten dokumentiert ist die Wirksamkeit von Bisphosphonaten bei der Therapie der Osteogenesis imperfecta, wo sie die Frakturrate verringern und die Mobilität deutlich verbessern können. Wenn auch die bisherigen Erfahrungen sehr ermutigend sind, bleibt doch der nicht ausreichend bekannte Einfluß einer Dauertherapie auf die Knochenentwicklung weiterhin der kritischste Punkt in der pädiatrischen Anwendung. Daher sollte derzeit die Therapie von Kindern mit Bisphosphonaten auf spezialisierte Zentren beschränkt bleiben.
    Type of Medium: Electronic Resource
    URL: http://dx.doi.org/10.1007/s001120050556
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  • 4
    Electronic Resource
    Electronic Resource
    Congenital primary cutaneous osteoma: Biochemical and histological studies (1983)
    Springer
    Archives of dermatological research 275 (1983), S. 114-117 
    Details
    ISSN: 1432-069X
    Keywords: Cutaneous osteoma ; Collagen ; Ectopic osteogenesis ; Collagen crosslinks
    Source: Springer Online Journal Archives 1860-2000
    Topics: Medicine
    Notes: Summary Biopsies of a cutaneous osteoma and of normal-looking skin from a 1-year-old girl were studied for histological appearance and collagen biochemistry. The mineralized tissue contained a matrix similar to bone: Only type I collagen, with a hydroxylysine content (0.48%) higher than in the skin (0.35%) and dihydroxylysinonorleucine as the major reducible crosslink. As expected, the normal skin adjacent to the lesions contained type I and type III collagen and as major crosslinks hydroxylysinonorleucine and histidinohydroxymerodesmosine. Histological studies showed the presence of woven bone with very little trabeculation. Numerous active osteoblasts were laying down a rapidly calcified non-lamellar matrix. Osteocytes and multinucleated osteoclasts were also noted. The study demonstrates the osseous nature of the lesion and suggests that an abnormal cell differentiation is associated with this form of osteoma.
    Type of Medium: Electronic Resource
    URL: http://dx.doi.org/10.1007/BF00412886
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    Paper (German National Licenses)
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