Hormone Research in Paediatrics

Original Paper

Normal Tempo of Bone Formation in Turner Syndrome despite Signs of Accelerated Bone Resorption

Cleemann L.a · Holm K.a · Kobbernagel H.b · Skouby S.O.c · Kristensen B.b · Smedegaard H.a · Andersson A.-M.d · Cohen A.e · Gravholt C.H.f

Author affiliations

Departments of aPediatrics and bClinical Physiology, Hillerød Hospital, Hillerød, cDepartment of Gynecology and Obstetrics, Herlev University Hospital, Herlev, dDepartment of Growth and Reproduction, Copenhagen University Hospital and eStatens Serum Institut, Copenhagen, and fDepartment of Endocrinology and Internal Medicine and the Medical Research Laboratories, Aarhus Sygehus NBG, Aarhus University Hospital, Aarhus, Denmark

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Horm Res Paediatr 2011;76:193–201

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Article / Publication Details

First-Page Preview
Abstract of Original Paper

Received: November 05, 2010
Accepted: May 04, 2011
Published online: July 26, 2011
Issue release date: October 2011

Number of Print Pages: 9
Number of Figures: 2
Number of Tables: 4

ISSN: 1663-2818 (Print)
eISSN: 1663-2826 (Online)

For additional information: https://www.karger.com/HRP

Abstract

Aims: To evaluate area bone mineral density (aBMD) and volumetric BMD (vBMD) by dual-energy X-ray absorptiometry, and relations to bone markers and hormones in adolescent women with Turner syndrome (TS). Methods: Cross-sectional study in TS patients (n = 37, 16.7 ± 3.4 years) and control group (n = 49), assessed by dual-energy X-ray absorptiometry, bone markers and hormones. TS patients were divided into a young group receiving (‘ongoing’) GH (n = 15) and an older group previously receiving (‘previous’) GH (n = 22). Results: vBMDspine was similar in ‘ongoing GH’ TS, but higher in ‘previous GH’ TS, compared to controls. vBMDhip was lower in ‘ongoing GH’ TS, but similar in ‘previous GH’. z scores for aBMD were uniformly reduced in ‘ongoing TS’, but near-normalized in ‘previous GH’ TS. Bone formation and resorption markers were increased in ‘ongoing GH’ TS, while ‘previous GH’ TS had elevated bone resorption markers. Conclusion: BMD increased in parallel with age in TS patients receiving optimal estradiol replacement therapy and GH according to consensus guidelines, and in controls. Young TS undergoing pubertal induction and still receiving GH have lower z score BMD than older TS patients receiving hormonal replacement therapy, where a near-normalization of BMD was achieved. TS patients previously receiving GH showed signs of increased bone resorption.

© 2011 S. Karger AG, Basel




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References

  1. Landin-Wilhelmsen K, Bryman I, Windh M, Wilhelmsen L: Osteoporosis and fractures in Turner syndrome – importance of growth promoting and oestrogen therapy. Clin Endocrinol (Oxf) 1999;51:497–502.
  2. Gravholt CH, Vestergaard P, Hermann AP, Mosekilde L, Brixen K, Christiansen JS: Increased fracture rates in Turner’s syndrome: a nationwide questionnaire survey. Clin Endocrinol (Oxf) 2003;59:89–96.
  3. Bakalov VK, Chen ML, Baron J, Hanton LB, Reynolds JC, Stratakis CA, Axelrod LE, Bondy CA: Bone mineral density and fractures in Turner syndrome. Am J Med 2003;115:259–264.
  4. Gravholt CH, Juul S, Naeraa RW, Hansen J: Morbidity in Turner syndrome. J Clin Epidemiol 1998;51:147–158.
  5. Carrascosa A, Gussinye M, Terradas P, Yeste D, Audi L, Vicens-Calvet E: Spontaneous, but not induced, puberty permits adequate bone mass acquisition in adolescent Turner syndrome patients. J Bone Miner Res 2000;15:2005–2010.
  6. Hogler W, Briody J, Moore B, Garnett S, Lu PW, Cowell CT: Importance of estrogen on bone health in turner syndrome: a cross-sectional and longitudinal study using dual-energy X-ray absorptiometry. J Clin Endocrinol Metab 2004;89:193–199.
  7. Gordon CM, Bachrach LK, Carpenter TO, Crabtree N, El-Hajj FG, Kutilek S, Lorenc RS, Tosi LL, Ward KA, Ward LM, Kalkwarf HJ: Dual energy X-ray absorptiometry interpretation and reporting in children and adolescents: the 2007 ISCD Pediatric Official Positions. J Clin Densitom 2008;11:43–58.
  8. Pors NS, Kolthoff N, Barenholdt O, Kristensen B, Abrahamsen B, Hermann AP, Brot C: Diagnosis of osteoporosis by planar bone densitometry: can body size be disregarded? Br J Radiol 1998;71:934–943.
  9. Shaw NJ, Rehan VK, Husain S, Marshall T, Smith CS: Bone mineral density in Turner’s syndrome – a longitudinal study. Clin Endocrinol Oxf 1997;47:367–370.
  10. Bertelloni S, Cinquanta L, Baroncelli GI, Simi P, Rossi S, Saggese G: Volumetric bone mineral density in young women with Turner’s syndrome treated with estrogens or estrogens plus growth hormone. Horm Res 2000;53:72–76.
  11. Gravholt CH, Lauridsen AL, Brixen K, Mosekilde L, Heickendorff L, Christiansen JS: Marked dysproportionality in bone size and mineral, and distinct abnormalities in bone markers and calcitropic hormones in adult Turner syndrome. A cross-sectional study. J Clin Endocrinol Metab 2002;87:2798–2808.
  12. Bakalov VK, Bondy CA: Fracture risk and bone mineral density in Turner syndrome. Rev Endocr Metab Disord 2008;9:145–151.
  13. Schoenau E, Saggese G, Peter F, Baroncelli GI, Shaw NJ, Crabtree NJ, Zadik Z, Neu CM, Noordam C, Radetti G, Hochberg Z: From bone biology to bone analysis. Horm Res 2004;61:257–269.
  14. Szulc P, Seeman E, Delmas PD: Biochemical measurements of bone turnover in children and adolescents. Osteoporos Int 2000;11:281–294.
  15. Garnero P: Biomarkers for osteoporosis management: utility in diagnosis, fracture risk prediction and therapy monitoring. Mol Diagn Ther 2008;12:157–170.
  16. Davies MC, Gulekli B, Jacobs HS: Osteoporosis in Turner’s syndrome and other forms of primary amenorrhoea. Clin Endocrinol Oxf 1995;43:741–746.
  17. Gallicchio CT, Figueiredo-Alves ST, Prendin TR, Mendonca LM, Farias ML, Guimaraes MM: Effect of puberty on the relationship between bone markers of turnover and bone mineral density in Turner’s syndrome. Horm Res 2004;61:193–199.
  18. Zuckerman-Levin N, Yaniv I, Schwartz T, Guttmann H, Hochberg Z: Normal DXA bone mineral density but frail cortical bone in Turner’s syndrome. Clin Endocrinol (Oxf) 2007;67:60–64.
  19. Bachrach LK: Osteoporosis in children: still a diagnostic challenge. J Clin Endocrinol Metab 2007;92:2030–2032.
  20. Marshall WA, Tanner JM: Variations in pattern of pubertal changes in girls. Arch Dis Child 1969;44:291–303.
  21. Greulich WW, Pyle SI: Radiographic Atlas of Skeletal Development of the Hand and Wrist, ed 2. Stanford, Stanford University Press, 1959.
  22. Gomez B Jr, Ardakani S, Ju J, Jenkins D, Cerelli MJ, Daniloff GY, Kung VT: Monoclonal antibody assay for measuring bone-specific alkaline phosphatase activity in serum. Clin Chem 1995;41:1560–1566.
  23. Melkko J, Kauppila S, Niemi S, Risteli L, Haukipuro K, Jukkola A, Risteli J: Immunoassay for intact amino-terminal propeptide of human type I procollagen. Clin Chem 1996;42:947–954.
  24. Bakalov VK, Axelrod L, Baron J, Hanton L, Nelson LM, Reynolds JC, Hill S, Troendle J, Bondy CA: Selective reduction in cortical bone mineral density in Turner syndrome independent of ovarian hormone deficiency. J Clin Endocrinol Metab 2003;88:5717–5722.
  25. Ross JL, Long LM, Feuillan P, Cassorla F, Cutler GB Jr: Normal bone density of the wrist and spine and increased wrist fractures in girls with Turner’s syndrome. J Clin Endocrinol Metab 1991;73:355–359.
  26. Hanton L, Axelrod L, Bakalov V, Bondy CA: The importance of estrogen replacement in young women with Turner syndrome. J Womens Health (Larchmt) 2003;12:971–977.
  27. Khastgir G, Studd JW, Fox SW, Jones J, Alaghband-Zadeh J, Chow JW: A longitudinal study of the effect of subcutaneous estrogen replacement on bone in young women with Turner’s syndrome. J Bone Miner Res 2003;18:925–932.
  28. Sas TC, de Muinck Keizer-Schrama SM, Stijnen T, Van TA, Van Leeuwen WJ, Asarfi A, Van Rijn RR, Drop SL: Bone mineral density assessed by phalangeal radiographic absorptiometry before and during long-term growth hormone treatment in girls with Turner’s syndrome participating in a randomized dose-response study. Pediatr Res 2001;50:417–422.
  29. Bondy CA, Bakalov VK: Investigation of cardiac status and bone mineral density in Turner syndrome. Growth Horm IGF Res 2006;16(suppl A):S103–S108.
  30. Bakalov VK, Van PL, Baron J, Reynolds JC, Bondy CA: Growth hormone therapy and bone mineral density in Turner syndrome. J Clin Endocrinol Metab 2004;89:4886–4889.
  31. Kalkwarf HJ, Zemel BS, Gilsanz V, Lappe JM, Horlick M, Oberfield S, Mahboubi S, Fan B, Frederick MM, Winer K, Shepherd JA: The bone mineral density in childhood study: bone mineral content and density according to age, sex, and race. J Clin Endocrinol Metab 2007;92:2087–2099.
  32. Boot AM, de Ridder MA, Pols HA, Krenning EP, de Muinck Keizer-Schrama SM: Bone mineral density in children and adolescents: relation to puberty, calcium intake, and physical activity. J Clin Endocrinol Metab 1997;82:57–62.
  33. Schoenau E, Land C, Stabrey A, Remer T, Kroke A: The bone mass concept: problems in short stature. Eur J Endocrinol 2004;151(suppl 1):S87–S91.
  34. von SE: Pediatric bone density and fracture. Curr Osteoporos Rep 2007;5:128–134.
  35. Schlemmer A, Hassager C, Jensen SB, Christiansen C: Marked diurnal variation in urinary excretion of pyridinium cross-links in premenopausal women. J Clin Endocrinol Metab 1992;74:476–480.
  36. Shimizu M, Onoe Y, Mikumo M, Miyabara Y, Kuroda T, Yoshikata R, Ishitani K, Okano H, Ohta H: Variations in circulating osteoprotegerin and soluble RANKL during diurnal and menstrual cycles in young women. Horm Res 2009;71:285–289.
  37. Hansen TB, Brixen K, Vahl N, Jorgensen JO, Christiansen JS, Mosekilde L, Hagen C: Effects of 12 months of growth hormone (GH) treatment on calciotropic hormones, calcium homeostasis, and bone metabolism in adults with acquired GH deficiency: a double blind, randomized, placebo-controlled study. J Clin Endocrinol Metab 1996;81:3352–3359.
  38. Cleemann L, Hjerrild BE, Lauridsen AL, Heickendorff L, Christiansen JS, Mosekilde L, Gravholt CH: Long-term hormone replacement therapy preserves bone mineral density in Turner syndrome. Eur J Endocrinol 2009;161:251–257.
  39. Seibel MJ: Biochemical markers of bone turnover. I. Biochemistry and variability. Clin Biochem Rev 2005;26:97–122.
    External Resources

Article / Publication Details

First-Page Preview
Abstract of Original Paper

Received: November 05, 2010
Accepted: May 04, 2011
Published online: July 26, 2011
Issue release date: October 2011

Number of Print Pages: 9
Number of Figures: 2
Number of Tables: 4

ISSN: 1663-2818 (Print)
eISSN: 1663-2826 (Online)

For additional information: https://www.karger.com/HRP


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