Skip to main content
Erschienen in: Endocrine 3/2015

01.04.2015 | Research Letter

Evaluation of impact of steroid replacement treatment on bone health in children with 21-hydroxylase deficiency

verfasst von: M. Delvecchio, L. Soldano, A. Lonero, A. Ventura, P. Giordano, L. Cavallo, M. Grano, G. Brunetti, M. F. Faienza

Erschienen in: Endocrine | Ausgabe 3/2015

Einloggen, um Zugang zu erhalten

Abstract

There are conflicting data regarding the potential impact of chronic glucocorticoid (GC) therapy on the bone mineral density of patients with congenital adrenal hyperplasia (CAH). Previous studies performed by dual-energy X-ray absorptiometry reported conflicting results. The purpose of this study was to assess the impact of chronic GC replacement treatment in children with classical and non classical CAH due to 21-hydroxylase deficiency (21-OHD) by quantitative ultrasonometry (QUS), an easy, cheap, and radiation-free technique. The study population consisted of nineteen 21-OHD patients (nine males) on lifelong GC treatment. Anthropometric, hormonal, and treatment data were recorded for each patient, and bone quality was assessed by QUS measurements. QUS findings (amplitude-dependent speed of sound and bone transmission time) were normal in 21-OHD patients and did not correlate with duration of treatment, daily, total, and yearly hydrocortisone dose. Furthermore, no significant correlation was found between QUS findings and 17α-hydroxy progesterone, Δ4-androstenedione, and testosterone levels. In conclusion, our results provide reassurance that currently used replacement doses of GC do not have a major impact on bone in patients with CAH. QUS seems to be a reliable tool for screening of bone health in children with 21-OHD.
Literatur
1.
Zurück zum Zitat P.C. White, T.A. Bachega, Congenital adrenal hyperplasia due to 21 hydroxylase deficiency: from birth to adulthood. Semin. Reprod. Med. 30, 400–409 (2012)CrossRefPubMed P.C. White, T.A. Bachega, Congenital adrenal hyperplasia due to 21 hydroxylase deficiency: from birth to adulthood. Semin. Reprod. Med. 30, 400–409 (2012)CrossRefPubMed
2.
Zurück zum Zitat P.W. Speiser, R. Azziz, L.S. Baskin, L. Ghizzoni, T.W. Hensle, D.P. Merke, H.F. Meyer-Bahlburg, W.L. Miller, V.M. Montori, S.E. Oberfield, M. Ritzen, P.C. White, Endocrine Society, Congenital adrenal hyperplasia due to steroid 21-hydroxylase deficiency: an Endocrine Society clinical practice guideline. J. Clin. Endocrinol. Metab. 95, 4133–4160 (2010)CrossRefPubMedCentralPubMed P.W. Speiser, R. Azziz, L.S. Baskin, L. Ghizzoni, T.W. Hensle, D.P. Merke, H.F. Meyer-Bahlburg, W.L. Miller, V.M. Montori, S.E. Oberfield, M. Ritzen, P.C. White, Endocrine Society, Congenital adrenal hyperplasia due to steroid 21-hydroxylase deficiency: an Endocrine Society clinical practice guideline. J. Clin. Endocrinol. Metab. 95, 4133–4160 (2010)CrossRefPubMedCentralPubMed
3.
Zurück zum Zitat A. Crown, S. Lightman, Management of patients with glucocorticoid deficiency. Nat. Clin. Pract. Endocrinol. Metab. 1, 62–63 (2005)CrossRefPubMed A. Crown, S. Lightman, Management of patients with glucocorticoid deficiency. Nat. Clin. Pract. Endocrinol. Metab. 1, 62–63 (2005)CrossRefPubMed
4.
Zurück zum Zitat G. Mazziotti, A. Angeli, J.P. Bilezikian, E. Canalis, A. Giustina, Glucocorticoid-induced osteoporosis: an update. Trends Endocrinol. Metab. 17, 144–149 (2006)CrossRefPubMed G. Mazziotti, A. Angeli, J.P. Bilezikian, E. Canalis, A. Giustina, Glucocorticoid-induced osteoporosis: an update. Trends Endocrinol. Metab. 17, 144–149 (2006)CrossRefPubMed
5.
Zurück zum Zitat A. Ventura, G. Brunetti, S. Colucci, A. Oranger, F. Ladisa, L. Cavallo, M. Grano, M.F. Faienza, Glucocorticoid-induced osteoporosis in children with 21-hydroxylase deficiency. Biomed. Res. Int. (2013). doi:10.1155/2013/250462 PubMedCentralPubMed A. Ventura, G. Brunetti, S. Colucci, A. Oranger, F. Ladisa, L. Cavallo, M. Grano, M.F. Faienza, Glucocorticoid-induced osteoporosis in children with 21-hydroxylase deficiency. Biomed. Res. Int. (2013). doi:10.​1155/​2013/​250462 PubMedCentralPubMed
6.
Zurück zum Zitat A. Bachelot, Z. Chakhtoura, D. Samara-Boustani, J. Dulon, P. Touraine, M. Polak, Bone health should be an important concern in the care of patients affected by 21 hydroxylase deficiency. Int. J. Pediatr. Endocrinol. (2010). doi:10.1155/2010/326275 PubMedCentralPubMed A. Bachelot, Z. Chakhtoura, D. Samara-Boustani, J. Dulon, P. Touraine, M. Polak, Bone health should be an important concern in the care of patients affected by 21 hydroxylase deficiency. Int. J. Pediatr. Endocrinol. (2010). doi:10.​1155/​2010/​326275 PubMedCentralPubMed
7.
Zurück zum Zitat H. Falhammar, H. Filipsson, G. Holmdahl, P.O. Janson, A. Nordenskjöld, K. Hagenfeldt, M. Thorén, Fractures and bone mineral density in adult women with 21-hydroxylase deficiency. J. Clin. Endocrinol. Metab. 92, 4643–4649 (2007)CrossRefPubMed H. Falhammar, H. Filipsson, G. Holmdahl, P.O. Janson, A. Nordenskjöld, K. Hagenfeldt, M. Thorén, Fractures and bone mineral density in adult women with 21-hydroxylase deficiency. J. Clin. Endocrinol. Metab. 92, 4643–4649 (2007)CrossRefPubMed
8.
Zurück zum Zitat H. Falhammar, H. Filipsson, H. Nyström, A. Wedell, K. Brismar, M. Thorén, Bone mineral density, bone markers, and fractures in adult males with congenital adrenal hyperplasia. Eur. J. Endocrinol. 168, 331–341 (2013)CrossRefPubMed H. Falhammar, H. Filipsson, H. Nyström, A. Wedell, K. Brismar, M. Thorén, Bone mineral density, bone markers, and fractures in adult males with congenital adrenal hyperplasia. Eur. J. Endocrinol. 168, 331–341 (2013)CrossRefPubMed
9.
Zurück zum Zitat M.F. Faienza, G. Brunetti, S. Colucci, L. Piacente, M. Ciccarelli, L. Giordani, G.C. Del Vecchio, M. D’Amore, L. Albanese, L. Cavallo, M. Grano, Osteoclastogenesis in children with 21-hydroxylase deficiency on long-term glucocorticoid therapy: the role of receptor activator of nuclear factor-kappaB ligand/osteoprotegerin imbalance. J. Clin. Endocrinol. Metab. 94, 2269–2276 (2009)CrossRefPubMed M.F. Faienza, G. Brunetti, S. Colucci, L. Piacente, M. Ciccarelli, L. Giordani, G.C. Del Vecchio, M. D’Amore, L. Albanese, L. Cavallo, M. Grano, Osteoclastogenesis in children with 21-hydroxylase deficiency on long-term glucocorticoid therapy: the role of receptor activator of nuclear factor-kappaB ligand/osteoprotegerin imbalance. J. Clin. Endocrinol. Metab. 94, 2269–2276 (2009)CrossRefPubMed
11.
Zurück zum Zitat G. Brunetti, M.F. Faienza, L. Piacente, A. Ventura, A. Oranger, C. Carbone, A.D. Benedetto, G. Colaianni, M. Gigante, G. Mori, L. Gesualdo, S. Colucci, L. Cavallo, M. Grano, High dickkopf-1 levels in sera and leukocytes from children with 21-hydroxylase deficiency on chronic glucocorticoid treatment. Am. J. Physiol. Endocrinol. Metab. 304, E546–E554 (2013)CrossRefPubMed G. Brunetti, M.F. Faienza, L. Piacente, A. Ventura, A. Oranger, C. Carbone, A.D. Benedetto, G. Colaianni, M. Gigante, G. Mori, L. Gesualdo, S. Colucci, L. Cavallo, M. Grano, High dickkopf-1 levels in sera and leukocytes from children with 21-hydroxylase deficiency on chronic glucocorticoid treatment. Am. J. Physiol. Endocrinol. Metab. 304, E546–E554 (2013)CrossRefPubMed
12.
Zurück zum Zitat M.A. Krieg, R. Barkmann, S. Gonnelli, A. Stewart, D.C. Bauer, L. Del Rio Barquero, J.J. Kaufman, R. Lorenc, P.D. Miller, W.P. Olszynski, C. Poiana, A.M. Schott, Lewiecki E.M, D. Hans, Quantitative ultrasound in the management of osteoporosis: the 2007 ISCD Official Positions. J. Clin. Densitom. 11, 163–187 (2008)CrossRefPubMed M.A. Krieg, R. Barkmann, S. Gonnelli, A. Stewart, D.C. Bauer, L. Del Rio Barquero, J.J. Kaufman, R. Lorenc, P.D. Miller, W.P. Olszynski, C. Poiana, A.M. Schott, Lewiecki E.M, D. Hans, Quantitative ultrasound in the management of osteoporosis: the 2007 ISCD Official Positions. J. Clin. Densitom. 11, 163–187 (2008)CrossRefPubMed
13.
Zurück zum Zitat A. Christoforidis, N. Printza, C. Gkogka, E. Siomou, A. Challa, E. Kazantzidou, K. Kollios, F. Papachristou, Comparative study of quantitative ultrasonography and dual-energy X-ray absorptiometry for evaluating renal osteodystrophy in children with chronic kidney disease. J. Bone Miner. Metab. 29, 321–327 (2011)CrossRefPubMed A. Christoforidis, N. Printza, C. Gkogka, E. Siomou, A. Challa, E. Kazantzidou, K. Kollios, F. Papachristou, Comparative study of quantitative ultrasonography and dual-energy X-ray absorptiometry for evaluating renal osteodystrophy in children with chronic kidney disease. J. Bone Miner. Metab. 29, 321–327 (2011)CrossRefPubMed
14.
Zurück zum Zitat P. Pisani, M.D. Renna, F. Conversano, E. Casciaro, M. Muratore, E. Quarta, M.D. Paola, S. Casciaro, Screening and early diagnosis of osteoporosis through X-ray and ultrasound based techniques. World J. Radiol. 5, 398–410 (2013)CrossRefPubMedCentralPubMed P. Pisani, M.D. Renna, F. Conversano, E. Casciaro, M. Muratore, E. Quarta, M.D. Paola, S. Casciaro, Screening and early diagnosis of osteoporosis through X-ray and ultrasound based techniques. World J. Radiol. 5, 398–410 (2013)CrossRefPubMedCentralPubMed
15.
Zurück zum Zitat G.I. Baroncelli, G. Federico, M. Vignolo, G. Valerio, A. del Puente, M. Maghnie, M. Baserga, G. Farello, G. Saggese, Phalangeal Quantitative Ultrasound Group, Cross-sectional reference data for phalangeal quantitative ultrasound from early childhood to young-adulthood according to gender, age, skeletal growth, and pubertal development. Bone 39, 159–173 (2006)CrossRefPubMed G.I. Baroncelli, G. Federico, M. Vignolo, G. Valerio, A. del Puente, M. Maghnie, M. Baserga, G. Farello, G. Saggese, Phalangeal Quantitative Ultrasound Group, Cross-sectional reference data for phalangeal quantitative ultrasound from early childhood to young-adulthood according to gender, age, skeletal growth, and pubertal development. Bone 39, 159–173 (2006)CrossRefPubMed
16.
Zurück zum Zitat H. Falhammar, M. Thorén, Clinical outcomes in the management of congenital adrenal hyperplasia. Endocrine 41, 355–373 (2012)CrossRefPubMed H. Falhammar, M. Thorén, Clinical outcomes in the management of congenital adrenal hyperplasia. Endocrine 41, 355–373 (2012)CrossRefPubMed
18.
Zurück zum Zitat H. Falhammar, H.F. Nyström, M. Thorén, Quality of life, social situation, and sexual satisfaction, in adult males with congenital adrenal hyperplasia. Endocrine (2014). doi:10.1007/s12020-013-0161-2 H. Falhammar, H.F. Nyström, M. Thorén, Quality of life, social situation, and sexual satisfaction, in adult males with congenital adrenal hyperplasia. Endocrine (2014). doi:10.​1007/​s12020-013-0161-2
19.
Zurück zum Zitat P.O. de Almeida Freire, i S.H. de Valente Lemos-Marin, A. Trevas Maciel-Guerra, A.M. Morcillo, M.T. Matias Baptista, M.P. de Mello, G. Guerra Jr., Classical congenital adrenal hyperplasia due to 21-hydroxylase deficiency: a cross-sectional study of factors involved in bone mineral density. J. Bone Miner. Metab. 21, 396–401 (2003)CrossRefPubMed P.O. de Almeida Freire, i S.H. de Valente Lemos-Marin, A. Trevas Maciel-Guerra, A.M. Morcillo, M.T. Matias Baptista, M.P. de Mello, G. Guerra Jr., Classical congenital adrenal hyperplasia due to 21-hydroxylase deficiency: a cross-sectional study of factors involved in bone mineral density. J. Bone Miner. Metab. 21, 396–401 (2003)CrossRefPubMed
20.
Zurück zum Zitat R.H. Elnecave, C. Kopacek, M. Rigatto, J.K. Brenner, J.A.S. de Castro, Bone mineral density in girls with classical congenital adrenal hyperplasia due to CYP21 deficiency. J. Pediatr. Endocrinol. Metab. 21, 1155–1162 (2008)CrossRefPubMed R.H. Elnecave, C. Kopacek, M. Rigatto, J.K. Brenner, J.A.S. de Castro, Bone mineral density in girls with classical congenital adrenal hyperplasia due to CYP21 deficiency. J. Pediatr. Endocrinol. Metab. 21, 1155–1162 (2008)CrossRefPubMed
21.
Zurück zum Zitat C. Wüster, C. Albanese, D. De Aloysio, F. Duboeuf, M. Gambacciani, S. Gonnelli, C.C. Glüer, D. Hans, J. Joly, J.Y. Reginster, F. De Terlizzi, R. Cadossi, Phalangeal osteosonogrammetry study (PhOS): age related changes, diagnostic sensitivity and discrimination power. J Bone. Miner. Res. 15, 1603–1614 (2000)CrossRefPubMed C. Wüster, C. Albanese, D. De Aloysio, F. Duboeuf, M. Gambacciani, S. Gonnelli, C.C. Glüer, D. Hans, J. Joly, J.Y. Reginster, F. De Terlizzi, R. Cadossi, Phalangeal osteosonogrammetry study (PhOS): age related changes, diagnostic sensitivity and discrimination power. J Bone. Miner. Res. 15, 1603–1614 (2000)CrossRefPubMed
22.
Zurück zum Zitat G.I. Baroncelli, G. Federico, S. Bertelloni, F. Sodini, F. De Terlizzi, R. Cadossi, G. Saggese, Assessment of bone quality by quantitative ultrasound of proximal phalanges of the hand and fracture rate in children and adolescents with bone and mineral disorders. Pediatr. Res. 54, 125–136 (2003)CrossRefPubMed G.I. Baroncelli, G. Federico, S. Bertelloni, F. Sodini, F. De Terlizzi, R. Cadossi, G. Saggese, Assessment of bone quality by quantitative ultrasound of proximal phalanges of the hand and fracture rate in children and adolescents with bone and mineral disorders. Pediatr. Res. 54, 125–136 (2003)CrossRefPubMed
23.
Zurück zum Zitat Z.P. Halaba, J. Konstantynowicz, W. Pluskiewicz, M. Kaczmarski, J. Piotrowska-Jastrzebska, Comparison of phalangeal ultrasound and dual energy X-ray absorptiometry in healthy male and female adolescents. Ultrasound Med. Biol. 31, 1617–1622 (2005)CrossRefPubMed Z.P. Halaba, J. Konstantynowicz, W. Pluskiewicz, M. Kaczmarski, J. Piotrowska-Jastrzebska, Comparison of phalangeal ultrasound and dual energy X-ray absorptiometry in healthy male and female adolescents. Ultrasound Med. Biol. 31, 1617–1622 (2005)CrossRefPubMed
24.
Zurück zum Zitat L. Tauchmanovà, V. Nuzzo, A. Del Puente, F. Fonderico, A. Esposito-Del Puente, Padulla. S., Rossi, A., Bifulco, G., Lupoli, G., Lombardi, G.: reduced bone mass detected by bone quantitative ultrasonometry and DEXA in pre- and postmenopausal women with endogenous subclinical hyperthyroidism. Maturitas 48, 299–306 (2004)CrossRefPubMed L. Tauchmanovà, V. Nuzzo, A. Del Puente, F. Fonderico, A. Esposito-Del Puente, Padulla. S., Rossi, A., Bifulco, G., Lupoli, G., Lombardi, G.: reduced bone mass detected by bone quantitative ultrasonometry and DEXA in pre- and postmenopausal women with endogenous subclinical hyperthyroidism. Maturitas 48, 299–306 (2004)CrossRefPubMed
25.
Zurück zum Zitat R. Di Mase, M. Cerbone, N. Improda, A. Esposito, D. Capalbo, C. Mainolfi, F. Santamaria, C. Pignata, M. Salerno, Bone health in children with long-term idiopathic subclinical hypothyroidism. Ital. J. Pediatr. 38, 56 (2012)CrossRefPubMedCentralPubMed R. Di Mase, M. Cerbone, N. Improda, A. Esposito, D. Capalbo, C. Mainolfi, F. Santamaria, C. Pignata, M. Salerno, Bone health in children with long-term idiopathic subclinical hypothyroidism. Ital. J. Pediatr. 38, 56 (2012)CrossRefPubMedCentralPubMed
Metadaten
Titel
Evaluation of impact of steroid replacement treatment on bone health in children with 21-hydroxylase deficiency
verfasst von
M. Delvecchio
L. Soldano
A. Lonero
A. Ventura
P. Giordano
L. Cavallo
M. Grano
G. Brunetti
M. F. Faienza
Publikationsdatum
01.04.2015
Verlag
Springer US
Erschienen in
Endocrine / Ausgabe 3/2015
Print ISSN: 1355-008X
Elektronische ISSN: 1559-0100
DOI
https://doi.org/10.1007/s12020-014-0332-9

Weitere Artikel der Ausgabe 3/2015

Endocrine 3/2015 Zur Ausgabe

Leitlinien kompakt für die Innere Medizin

Mit medbee Pocketcards sicher entscheiden.

Seit 2022 gehört die medbee GmbH zum Springer Medizin Verlag

Update Innere Medizin

Bestellen Sie unseren Fach-Newsletter und bleiben Sie gut informiert.