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Idiopathic juvenile osteoporosis: Evidence of normal osteoblast function by 1,25-dihydroxyvitamin D3 stimulation test

Summary

Idiopathic juvenile osteoporosis (IJO) is a rare form of bone demineralization that occurs during childhood. The mechanism of bone loss is unclear. Some bone hystomorphometric studies have found osteoblast failure and decreased bone formation in the affected patients whereas others have reported increased bone resorption. To elucidate this issue, we studied osteoblast function in six patients with IJO (five males, one female; aged 2.3–14.6 years) and five healthy sex- and age-matched subjects (four males, one female; aged 2.0–15.1 years) measuring serum values of osteocalcin under basal condition and during an osteoblast stimulation test performed by oral 1,25-dihydroxyvitamin D3 [1,25(OH)2D3] administration (1.8 μg/1.73 m2/daily). After a baseline day (day 0), all the subjects (patients and controls) received 1,25(OH)2D3 in four divided doses for 6 days (days 1–6). Fasting blood samples were obtained every morning (0800 h) for the determination of serum osteocalcin. Baseline osteocalcin levels were not significantly different between IJO and controls (13.58±6.05 ng/ml versus 16.04±5.09 ng/ml, respectively) even if two patients had low osteocalcin values. During 1,25(OH)2D3 administration, serum osteocalcin values significantly increased (P<0.001) from baseline in both children with IJO and controls, reaching peak values not significantly different in the two groups. Our results do not support the hypothesis that defective osteoblast function is the primary factor of bone demineralization in IJO.

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References

  1. 1.

    Smith R (1980) Idiopathic osteoporosis in the young. J Bone Joint Surg 62-B:417–427

  2. 2.

    Saggese G, Bertelloni S, Baroncelli GI, Perri G, Calderazzi A (1991) Mineral metabolism and calcitriol therapy in idiopathic juvenile osteoporosis. AJDC 145:457–462

  3. 3.

    Teotia M, Teotia PS, Singh RK (1979) Idiopathic juvenile osteoporosis. AJDC 133:894–900

  4. 4.

    Exner GU, Prader A, Elsasser U, Anlinker M (1984) Idiopathic osteoporosis in a three-year old girl: follow-up over a period of 6 years by computed tomography bone densitometry. Helv Paediatr Acta 39:517–529

  5. 5.

    Kooh SW, Cumming WA, Fraser D, Fraser A (1973) Transient childhood osteoporosis of unknown cause. In: Frame B, Parfit AM, Duncan H (eds) Clinical aspect of bone disease. Excerpta Medica, Princeton, pp 329–332

  6. 6.

    Jowsey J, Johnson KA (1972) Juvenile osteoporosis: bone findings in seven patients. J Pediatr 81:511–517

  7. 7.

    Berglund G, Lindquist B (1960) Osteopenia in adolescence. Clin Orthop 17:259–264

  8. 8.

    Wieczorek E, Jaworsky Z, Pronicka E (1989) Histomorphometric study of bone in idiopathic osteoporosis of the youth (abstract). Ped Res 26:518

  9. 9.

    Price PA (1988) New bone markers. Triangle 27:21–26

  10. 10.

    Price PA, Baukol SA (1980) 1,25-Dihydroxyvitamin D3 increases synthesis of the vitamin K-dependent bone protein by osteosarcoma cells. J Biol Chem 225:11660–11663

  11. 11.

    Zerwekh JE, Sakhaee K, Pak CYC (1985) Short-term 1,25-dihydroxyvitamin D administration raises serum osteocalcin in patients with postmenopausal osteoporosis. J Clin Endocrinol Metab 60:615–617

  12. 12.

    Audran M, Gross M, Kumar R (1986) The physiology of the vitamin D endocrine system. Semin Nephrol 6:4–20

  13. 13.

    Duda RJ, Kumar R, Nelson KI, Zinsmeister AR, Mann KJ, Riggs BL (1987) 1,25-dihydroxyvitamin D stimulation test for osteoblast function in normal and osteoporotic postmenopausal women. J Clin Invest 79:1249–1253

  14. 14.

    Saggese G, Federico G, Ghirri P, Cipriani J, Bertelloni S, Baroncelli GI (1986) Bone mineral content in pediatrics—normal values between 2 and 19 years: first Italian data. Minerva Pediatr 38:545–551

  15. 15.

    Portale AA, Halloran BP, Murphy MM, Morris RC Jr (1986) Oral intake of phosphorus can determine the serum concentrations of 1,25-dihydroxyvitamin D by determining its production rate in humans. J Clin Invest 77:7–12

  16. 16.

    Sena SF, Bowers GN Jr (1988) Measurement of ionized calcium in biological fluids: ion-selective electrode method. Methods Enzymol 158:320–334

  17. 17.

    Cole D, Carpenter T, Gundberg C (1985) Serum osteocalcin concentrations in children with metabolic bone disease. J Pediatr 106:770–776

  18. 18.

    Saggese G, Bertelloni S, Baroncelli GI, Di Nero G, Perri G (1991) Idiopathic juvenile osteoporosis. Riv Ital Pediatr (IJP) 17:1405–1418

  19. 19.

    Hughes A (1986) Handbook of endocrine tests in children. J Wright & Sons Ltd, Bristol, UK

  20. 20.

    Marder HK, Tsang RC, Hug G, Crawford AC (1982) Calcitriol deficiency in idiopathic juvenile osteoporosis. AJDC 136:914–917

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Bertelloni, S., Baroncelli, G.I., Di Nero, G. et al. Idiopathic juvenile osteoporosis: Evidence of normal osteoblast function by 1,25-dihydroxyvitamin D3 stimulation test. Calcif Tissue Int 51, 20–23 (1992). https://doi.org/10.1007/BF00296211

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Key words

  • Idiopathic juvenile osteoporosis
  • Osteoblast function
  • Osteocalcin
  • 1,25(OH)2D3 administration