Height, Bone Mineral Density and Bone Markers in Congenital Adrenal Hyperplasia
Abstract
Aim: To evaluate height, bone growth, areal bone mineral density (aBMD), volumetric bone mineral density (vBMD) and markers of bone turnover in a group of patients affected by congenital adrenal hyperplasia (CAH). Patients: There were 50 patients (23 males, 27 females), aged 1–28 years, affected by CAH due to 21-hydroxylase deficiency: 27 with the salt-wasting (SW); 14 with the simple virilizing (SV), and 9 with the nonclassical (NC) forms. Methods: Bone morphometry was evaluated with the metacarpal index (MI) and lumbar aBMD and vBMD (L2–L4) by dual energy X-ray absorptiometry. Serum osteocalcin was used as a marker of bone formation, while urinary cross-linked N-telopeptides of type-I collagen and free deoxypyridinoline levels were evaluated as indexes of bone resorption. Results: The height standard deviation score (SDS) was –0.41 ± 1.4 in SW patients, –0.01 ± 1.9 in SV patients, and –0.01 ± 2.3 in NC patients. There was no significant difference among groups and against zero. The MI SDS was also not different between groups and against zero.aBMD was significantly lower in the pubertal patients compared with normal values, but only when patients with the SW and SV forms were considered together (p < 0.05). vBMD was significantly reduced in all patients with the classical form. Bone markers were not different in patients and controls. Conclusion: Our study shows that normal height can be attained in CAH patients; however, an impairment in bone growth and mineralization may be found in adolescents and young adults affected by the classical form.
Integrity signals
Registry
Cites
Statistics
Claims the paper makes
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
No correlation was found between glucocorticoid exposure and BMD.
“We did not find a correlation between GC exposure and BMD, similar to some prior studies 19, 20.”
From Cortical bone mineral density in patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency (2015) · cites this paper as [19]
Cites
RADIOGRAPHIC ATLAS OF SKELETAL DEVELOPMENT OF THE HAND AND WRIST
GREULICH, W. W., & Pyle, S. I. (1959) · The American Journal of the Medical Sciences · doi:10.1097/00000441-195909000-00030 · Cited by 5,598 · see citing works
Tanner, J. M., Whitehouse, R. H., & Takaishi, M. (1966) · Archives of Disease in Childhood · doi:10.1136/adc.41.220.613 · Cited by 2,560 · see citing works
Body Mass Index variations: centiles from birth to 87 years.
Rolland‐Cachera, M., Cole, T. J., Sempe, M., Tichet, J., Rossignol, C., & Charraud, A.-M. (1991) · PubMed · Cited by 1,127 · see citing works
Boot, A. M., de Ridder, M. A. J., Pols, H. A. P., Krenning, E. P., & Keizer‐Schrama, S. M. P. F. D. M. (1997) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem.82.1.3665 · Cited by 508 · see citing works
Boot, A. M. (1997) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.82.1.57 · Cited by 365 · see citing works
Perspectives bone histomorphometry in glucocorticoid-induced osteoporosis
Dempster, D. W. (1989) · Journal of Bone and Mineral Research · doi:10.1002/jbmr.5650040202 · Cited by 319 · see citing works
Kröger, H., Kotaniemi, A., Kröger, L. M., & Alhava, E. M. (1993) · Bone and Mineral · doi:10.1016/s0169-6009(08)80094-3 · Cited by 269 · see citing works
del Río, L., Carrascosa, A., Pons, F., Gusinyé, M., Yeste, D., & Domenech, F. M. (1994) · Pediatric Research · doi:10.1203/00006450-199403000-00018 · Cited by 211 · see citing works
Glucocorticoids inhibit bone resorption by isolated rat osteoclasts by enhancing apoptosis
Dempster, D., Moonga, B., Stein, L., Horbert, W., & Antakly, T. (1997) · Journal of Endocrinology · doi:10.1677/joe.0.1540397 · Cited by 180 · see citing works
Clinical review 54: genetics, diagnosis, and management of 21-hydroxylase deficiency
Miller, W. L. (1994) · Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.78.2.241 · Cited by 167 · see citing works
Chiodini, I., Carnevale, V., Torlontano, M., Fusilli, S., Guglielmi, G., Pileri, M., Modoni, S., Giorgio, A. D., Liuzzi, A., Minisola, S., et al. (1998) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem.83.6.4880 · Cited by 154 · see citing works
Clinical review 54: Genetics, diagnosis, and management of 21-hydroxylase deficiency.
Miller, W. L. (1994) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem.78.2.8106606 · Cited by 152 · see citing works
Girgis, R., & Winter, J. S. D. (1997) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem.82.12.4320 · Cited by 139 · see citing works
Serum Androgens as a Continuing Index of Adequacy of Treatment of Congenital Adrenal Hyperplasia*
KORTH-SCHÜTZ, S., Virdis, R., Saenger, P. H., CHOW, D. M., Levine, L. S., & New, M. I. (1978) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem-46-3-452 · Cited by 103 · see citing works
Bone density in young patients with congenital adrenal hyperplasia
Mora, S., Saggion, F., Russo, G., Weber, G., Bellini, A., Prinster, C., & Chiumello, G. (1996) · Bone · doi:10.1016/8756-3282(96)00003-8 · Cited by 95 · see citing works
Jääskeläinen, J., & Voutilainen, R. (1996) · Clinical Endocrinology · doi:10.1046/j.1365-2265.1996.8620871.x · Cited by 92 · see citing works
Gussinyé, M., Carrascosa, A., Potau, N., Enrubia, M., Vicens-Calvet, E., Ibáñez, L., & Yeste, D. (1997) · PEDIATRICS · doi:10.1542/peds.100.4.671 · Cited by 90 · see citing works
Kaji, H., Sugimoto, T., Kanatani, M., Nishiyama, K., & Chihara, K. (1997) · Journal of Bone and Mineral Research · doi:10.1359/jbmr.1997.12.5.734 · Cited by 85 · see citing works
Glucocorticoids and Osteoporosis
Adler, R. A., & Rosen, C. J. (1994) · Endocrinology and Metabolism Clinics of North America · doi:10.1016/s0889-8529(18)30090-2 · Cited by 79 · see citing works
<b>Bone turnover and bone mineral density in patients with congenital adrenal hyperplasia</b>
Guo, C., Weetman, A. P., & Eastell, R. (1996) · Clinical Endocrinology · doi:10.1046/j.1365-2265.1996.00851.x · Cited by 79 · see citing works
Role of Growth Hormone and Sex Steroids in Achieving and Maintaining Normal Bone Mass
Holmes, S. J., & Shalet, S. M. (2008) · Hormone Research · doi:10.1159/000184765 · Cited by 75 · see citing works
Zamberlan, N., Radetti, G., Paganini, C., Gatti, D., Rossini, M., Braga, V., & Adami, S. (1996) · European Journal of Pediatrics · doi:10.1007/bf01955265 · Cited by 61 · see citing works
Self-citationThorsen, K., Nordström, P., Lorentzon, R., & Dahlén, G. H. (1999) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem.84.9.5950 · Cited by 31 · see citing works
INCREASED BONE MINERAL DENSITY IN CONGENITAL ADRENAL HYPERPLASIA (CAH)
Speiser, P., New, M. I., & Gertner, J. M. (1993) · Pediatric Research · doi:10.1203/00006450-199305001-00467 · Cited by 15 · see citing works
Hargitai, G., Hosszú, É., Halász, Z., & Sólyom, J. (1999) · Hormone Research in Paediatrics · doi:10.1159/000023449 · Cited by 9 · see citing works
Osteocalcin in Congenital Adrenal Hyperplasia
Lisa, L., Neradilová, M., Tomášová, N., Soutorová, M., & Zimák, J. (1995) · Bone · doi:10.1016/8756-3282(95)80011-e · Cited by 7 · see citing works
Flagged first, then by citations
Cited by
Sorted by citations
Congenital Adrenal Hyperplasia—Current Insights in Pathophysiology, Diagnostics, and Management
Claahsen - van der Grinten, H. L., Speiser, P. W., Ahmed, S. F., Arlt, W., Auchus, R. J., Falhammar, H., Flück, C. E., Guasti, L., Huebner, A., Kortmann, B. B. M., et al. (2022) · Endocrine Reviews · doi:10.1210/endrev/bnab016 · Cited by 556 · see citing works
Fractures and bone mineral density in adult women with 21-hydroxylase deficiency
Falhammar, H., Filipsson, H., Holmdahl, G., Janson, P.-O., Nordenskjöld, A., Hagenfeldt, K., & Thorén, M. (2007) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2007-0744 · Cited by 151 · see citing works
Falhammar, H., & Nordenström, A. (2015) · Endocrine · doi:10.1007/s12020-015-0656-0 · Cited by 127 · see citing works
Long-Term Outcome of Patients with Congenital Adrenal Hyperplasia due to 21-Hydroxylase Deficiency
Bachelot, A., Plu-Bureau, G., Thibaud, E., Laborde, K., Pinto, G., Samara, D., Nihoul-Fékété, C., Kuttenn, F., Polak, M., & Touraine, P. (2007) · Hormone Research in Paediatrics · doi:10.1159/000098017 · Cited by 106 · see citing works
Bone mineral density, bone markers, and fractures in adult males with congenital adrenal hyperplasia
Falhammar, H., Filipsson Nyström, H., Wedell, A., Brismar, K., & Thorén, M. (2013) · European Journal of Endocrinology · doi:10.1530/eje-12-0865 · Cited by 82 · see citing works
Sciannamblo, M., Russo, G., Cuccato, D., Chiumello, G., & Mora, S. (2006) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2005-2823 · Cited by 82 · see citing works
Rangaswamaiah, S., Gangathimmaiah, V., Nordenstrom, A., & Falhammar, H. (2020) · Frontiers in Endocrinology · doi:10.3389/fendo.2020.00493 · Cited by 59 · see citing works
El‐Maouche, D., Collier, S., Prasad, M., Reynolds, J. C., & Merke, D. P. (2015) · Clinical Endocrinology · doi:10.1111/cen.12507 · Cited by 53 · see citing works
Bone mineral status in children with congenital adrenal hyperplasia
Fleischman, A., Ringelheim, J., Feldman, H. A., & Gordon, C. M. (2007) · Journal of Pediatric Endocrinology and Metabolism · doi:10.1515/jpem.2007.20.2.227 · Cited by 27 · see citing works
Sarafoglou, K., Gonzalez‐Bolanos, M. T., Zimmerman, C. L., Boonstra, T., Yaw Addo, O., & Brundage, R. (2015) · The Journal of Clinical Pharmacology · doi:10.1002/jcph.424 · Cited by 20 · see citing works
Bechtold, S., Beyerlein, A., Bonfig, W., Dalla Pozza, R., Putzker, S., Otto, R., Schmidt, H., & Schwarz, H. P. (2014) · Hormone Research in Paediatrics · doi:10.1159/000362759 · Cited by 7 · see citing works
- Authors
- Paganini, C., Radetti, G., Livieri, C., Braga, V., Migliavacca, D., & Adami, S.
- Affiliations
- Ospedale di Bolzano (IT)
- University of Pavia (IT)
- Venue
- Hormone Research in Paediatrics
- Publisher
- S. Karger AG
- Published
- 2000
- Volume
- 54
- Issue
- 4
- Pages
- 164-168
- ISSN
- 1663-2818 (ISSN-L)
- 1663-2826
- OpenAlex
- W2033593865
- Cited by
- 87
- References
- 26
- Citations by year
- 2025: 2 · 2024: 1 · 2023: 2 · 2022: 3 · 2021: 2 · 2020: 2 · 2019: 3 · 2018: 1 · 2016: 2 · 2015: 2 · 2014: 3 · 2013: 4 · 2012: 6
- FWCI
- 1.57
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Hormone Research in Paediatrics (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Sexual Differentiation and Disorders
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Adrenal Hormones and Disorders
- Health Sciences › Medicine › Orthopedics and Sports Medicine › Bone health and osteoporosis research
- Keywords
- congenital adrenal hyperplasia, 21-hydroxylase deficiency, bone mineral density, bone growth, height, bone turnover markers, osteocalcin, N-telopeptide, deoxypyridinoline, salt-wasting form, simple virilizing form, non-classical congenital adrenal hyperplasia, bone mineralization
- MeSH terms
- Adolescent, Adrenal Hyperplasia, Congenital, Adult, Amino Acids, Body Height, Bone Development, Bone Resorption, Child, Child, Preschool, Collagen, Female, Humans, Infant, Lumbar Vertebrae, Male, Metacarpus, Peptides, Biomarkers, Absorptiometry, Photon, Bone Density, Osteocalcin, Collagen Type I
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review