The DOI matches its Crossref record; 28 of the 28 references it cites were found in a registry or an index.
Flutamide, Testolactone, and Reduced Hydrocortisone Dose Maintain Normal Growth Velocity and Bone Maturation Despite Elevated Androgen Levels in Children with Congenital Adrenal Hyperplasia
Abstract
Treatment outcome in congenital adrenal hyperplasia is often sub-optimal due to hyperandrogenism, treatment-induced hypercortisolism, or both. We previously reported better control of linear growth, weight gain, and bone maturation in a short term cross-over study of a new four-drug treatment regimen containing an antiandrogen (flutamide), an inhibitor of androgen to estrogen conversion (testolactone), reduced hydrocortisone dose, and fludrocortisone, compared to the effects of a control regimen of hydrocortisone and fludrocortisone. Twenty-eight children have completed 2 yr of follow-up in a subsequent long term randomized parallel study comparing these two treatment regimens. During 2 yr of therapy, compared to children receiving hydrocortisone, and fludrocortisone treatment, children receiving flutamide, testolactone, reduced hydrocortisone dose (average of 8.7 +/- 0.6 mg/m2 x day), and fludrocortisone had significantly (P < or = 0.05) higher plasma 17-hydroxyprogesterone, androstenedione, dehydroepiandrosterone, dehydroepiandrosterone sulfate, and testosterone levels. Despite elevated androgen levels, children receiving the new treatment regimen had normal linear growth rate (at 2 yr, 0.1 +/- 0.5 SD units), and bone maturation (at 2 yr, 0.7 +/- 0.3 yr bone age/yr chronological age). No significant adverse effects were observed after 2 yr. We conclude that the regimen of flutamide, testolactone, reduced hydrocortisone dose, and fludrocortisone provides effective control of congenital adrenal hyperplasia with reduced risk of glucocorticoid excess. A long term study of this new regimen is ongoing.
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In the citing paper
“This was studied in 28 prepubertal children with classic 21OH deficiency using an anti-androgen and aromatase-inhibitor in combination with lower dose hydrocortisone and fludrocortisone, and was successful in normalizing growth over 2 years.144”
From Congenital adrenal hyperplasia (2017) · cites this paper as [144]
In the citing paper
“In a 2-year randomized, parallel study, children with classic CAH (n = 28) receiving low dose hydrocortisone and fludrocortisone combined with the experimental regimen of flutamide (an anti-androgen) and testolactone (an aromatase inhibitor) maintained normal growth velocity and skeletal maturation despite elevations in adrenal androgens14.”
From Management challenges and therapeutic advances in congenital adrenal hyperplasia (2022) · cites this paper as [14]
In the citing paper
“In a 2-year randomized parallel study of 28 children, patients receiving the experimental four-drug regimen had normal growth and bone maturation, despite elevated adrenal steroids (315).”
From Congenital Adrenal Hyperplasia Due to Steroid 21-Hydroxylase Deficiency: An Endocrine Society* Clinical Practice Guideline (2018) · cites this paper as [315]
Citations
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Congenital Adrenal Hyperplasia
Speiser, P., & White, P. C. (1987) · New England Journal of Medicine · doi:10.1056/nejm198711263172211 · Cited by 1,420
Congenital adrenal hyperplasia
Speiser, P. W., & White, P. C. (2003) · New England Journal of Medicine · doi:10.1056/nejmra021561 · Cited by 1,405
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Speiser, P. W., Azziz, R., Baskin, L. S., Ghizzoni, L., Hensle, T. W., Merke, D. P., Meyer-Bahlburg, H. F. L., Miller, W. L., Montori, V. M., Oberfield, S. E., et al. (2010) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2009-2631 · Cited by 1,468
Self-citationCongenital adrenal hyperplasia due to 21-hydroxylase deficiency*
White, P. C., & Speiser, P. W. (2000) · Endocrine Reviews · doi:10.1210/edrv.21.3.0398 · Cited by 1,432
Congenital Adrenal Hyperplasia
Speiser, P., & White, P. C. (1987) · New England Journal of Medicine · doi:10.1056/nejm198711263172211 · Cited by 1,420
Congenital adrenal hyperplasia
Speiser, P. W., & White, P. C. (2003) · New England Journal of Medicine · doi:10.1056/nejmra021561 · Cited by 1,405
Speiser, P. W., Arlt, W., Auchus, R. J., Baskin, L. S., Conway, G. S., Merke, D. P., Meyer-Bahlburg, H. F. L., Miller, W. L., Murad, M. H., Oberfield, S. E., et al. (2018) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2018-01865 · Cited by 1,234
Self-citation
Metadata
- Authors
- Merke, D. P., Keil, M. F., Jones, J. V., Fields, J., Hill, S., & Cutler, G. B.
- Affiliations
- Warren Grant Magnuson Clinical Center (D.P.M., S.H.), Bethesda, Maryland 20892
- Developmental Endocrinology Branch (D.P.M., M.F.K., J.V.J., J.F., G.B.C.), Bethesda, Maryland 20892
- National Institutes of Health (US)
- Corresponding author
- Merke, Deborah P.
- Venue
- The Journal of Clinical Endocrinology & Metabolism
- Publisher
- The Endocrine Society
- Published
- 2000-03-01
- Volume
- 85
- Issue
- 3
- Pages
- 1114-1120
- ISSN
- 0021-972X (ISSN-L)
- 1945-7197
- OpenAlex
- W1997037606
- Cited by
- 190
- References
- 28
- Citations by year
- 2026: 6 · 2025: 5 · 2024: 9 · 2023: 5 · 2022: 10 · 2021: 5 · 2020: 5 · 2019: 9 · 2018: 3 · 2017: 4 · 2016: 5 · 2015: 9 · 2014: 3 · 2013: 6 · 2012: 2
- FWCI
- 3.59
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Sexual Differentiation and Disorders
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Adrenal Hormones and Disorders
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Hormonal Regulation and Hypertension
- Keywords
- congenital adrenal hyperplasia, flutamide, testolactone, fludrocortisone, hyperandrogenism, linear growth velocity, skeletal maturation, glucocorticoid excess, 17-hydroxyprogesterone, androgen to estrogen conversion, pediatric endocrinology
- MeSH terms
- Adrenal Hyperplasia, Congenital, Androgen Antagonists, Androgens, Anti-Inflammatory Agents, Bone Development, Child, Child, Preschool, Female, Flutamide, Follow-Up Studies, Growth, Hormones, Humans, Hydrocortisone, Male, Testolactone, Weight Gain, Antineoplastic Agents, Hormonal
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- CoChat. (2026). Citation check: Flutamide, Testolactone, and Reduced Hydrocortisone Dose Maintain Normal Growth Velocity and Bone Maturation Despite Elevated Androgen Levels in Children with Congenital Adrenal Hyperplasia. https://app.cochat.ai/papers/10.1210/jcem.85.3.6462
- The paper
- Merke, D. P., Keil, M. F., Jones, J. V., Fields, J., Hill, S., & Cutler, G. B. (2000). Flutamide, Testolactone, and Reduced Hydrocortisone Dose Maintain Normal Growth Velocity and Bone Maturation Despite Elevated Androgen Levels in Children with Congenital Adrenal Hyperplasia. The Journal of Clinical Endocrinology & Metabolism. https://doi.org/10.1210/jcem.85.3.6462