Collagen peptides in osteogenesis imperfecta, idiopathic juvenile osteoporosis and Ehlers–Danlos syndrome

Prószyńska, K.; Wieczorek, E; Olszaniecka, M; Rs, Lorenc

doi:10.1111/j.1651-2227.1996.tb14126.x

Cited by 19 publications

(9 citation statements)

References 11 publications

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“…Despite recent work disclosing the molecular background and the in vitro protein-chemical abnormalities of collagen I in patients with OI, many questions remain unanswered about the in vivo handling of deficient collagen I. Few studies have addressed the in vivo metabolism of collagen I in different types of OI (13)(14)(15)(16)(17), and this study is the first attempt to correlate the serum markers of bone metabolism with results of SDS-PAGE of collagen I. The results show that the errors of collagen I metabolism found in vitro in OI fibroblasts are reflected in vivo by the levels of collagen I markers.…”

Section: Discussionmentioning

confidence: 99%

Collagen‐derived markers of bone metabolism in osteogenesis imperfecta

et al. 1998

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Markers of bone formation [C‐terminal and N‐terminal propeptides of procollagen I (PICP, PINP), osteocalcin and alkaline phosphatase] and bone resorption [C‐terminal cross‐linked telopeptide of collagen I (ICTP) and hydroxypyridinium cross‐links, pyridinoline (Pyr) and deoxypyridinoline (Dpyr)] were measured in 78 osteogenesis imperfecta (OI) patients to investigate bone metabolism in vivo and relate marker concentrations to phenotype and in vitro collagen I defects, as shown by sodium dodecyl sulphate ‐polyacrylamide gel electrophoresis (SDS‐PAGE). PICP and PINP were generally low, and the serum levels were lower in all children and adults with mild OI and a quantitative collagen defect than in patients with severe OI and a qualitative collagen I defect. ICTP, Pyr and Dpyr were generally normal or reduced, but elevated in severely affected adults with a qualitative collagen I defect. The in vivo findings correlated with in vitro results of collagen I SDS‐PAGE. Bone turnover is reduced in OI children and mildly affected OI adults, whereas bone resorption is elevated in severely affected adults. These findings may prove helpful for diagnosis and decision‐making regarding therapy in OI.

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Section: Discussionmentioning

confidence: 99%

Collagen‐derived markers of bone metabolism in osteogenesis imperfecta

et al. 1998

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“…s-PICP reflects synthesis of collagen I, including formation of collagen in bone (6). As observed by others (13)(14)(15)18), this marker was decreased in most patients with OI and mostly so in patients with OI I and a quantitative collagen I defect, i.e. in those with a presumed haplo-insufficiency of COL1A1.…”

Section: Discussionmentioning

confidence: 55%

“…Few studies have addressed the in vivo metabolism of collagen I in different types of OI (13)(14)(15)(16)(17), and this study is the first attempt to correlate the serum markers of bone metabolism with results of SDS-PAGE of collagen I. The results show that the errors of collagen I metabolism found in vitro in OI fibroblasts are reflected in vivo by the levels of collagen I markers.…”

Section: Discussionmentioning

confidence: 81%

“…Few other studies have measured such markers in OI: one found normal or low s-ICTP in OI children (13), another elevated urinary cross-linked collagen peptides in all types of OI (16), and a third elevated excretion of pyridinolines in 20% of OI adults and low excretion in 40% (18). The present results show that most OI patients with a quantitative collagen I defect had normal u-pyridinoline concentrations and low s-ICTP concentrations, both of which, in adult patients, were lower than those seen in patients with a qualitative collagen I defect.…”

Section: Markers Of Collagen Synthesismentioning

confidence: 99%

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Collagen-derived markers of bone metabolism in osteogenesis imperfecta

et al. 1998

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Markers of bone formation [C-terminal and N-terminal propeptides of procollagen I (PICP, PINP), osteocalcin and alkaline phosphatase] and bone resorption [C-terminal cross-linked telopeptide of collagen I (ICTP) and hydroxypyridinium cross-links, pyridinoline (Pyr) and deoxypyridinoline (Dpyr)] were measured in 78 osteogenesis imperfecta (OI) patients to investigate bone metabolism in vivo and relate marker concentrations to phenotype and in vitro collagen I defects, as shown by sodium dodecyl sulphate-polyacrylamide gel electrophoresis (SDS-PAGE). PICP and PINP were generally low, and the serum levels were lower in all children and adults with mild OI and a quantitative collagen defect than in patients with severe OI and a qualitative collagen I defect. ICTP, Pyr and Dpyr were generally normal or reduced, but elevated in severely affected adults with a qualitative collagen I defect. The in vivo findings correlated with in vitro results of collagen I SDS-PAGE. Bone turnover is reduced in OI children and mildly affected OI adults, whereas bone resorption is elevated in severely affected adults. These findings may prove helpful for diagnosis and decision-making regarding therapy in OI.

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“…Ce marqueur d'ostéoformation peut contribuer au diagnostic d'ostéoge-nèse imparfaite, pouvant même aider au diagnostic diffé-rentiel de maltraitance [2,19]. Il est intéressant surtout en période prépubertaire, perdant par la suite sa sensibilité [15].…”

Section: Discussionunclassified