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Cited by 190Sexual Differentiation and DisordersAdrenal Hormones and DisordersHormonal Regulation and Hypertension

Flutamide, testolactone, and reduced hydrocortisone dose maintain normal growth velocity and bone maturation despite elevated androgen levels in children with congenital adrenal hyperplasia

Merke, Deborah P., Keil, Margaret F., Jones, Janet V., Fields, Jeremy, Hill, Suvimol, Cutler, Gordon B.·2000·The Journal of Clinical Endocrinology & Metabolism
DOI 10.1210/jcem.85.3.6462
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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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  • An anti-androgen and aromatase-inhibitor in combination with lower dose hydrocortisone and fludrocortisone was successful in normalizing growth over 2 years in 28 prepubertal children with classic 21OH deficiency.

    “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 a 2-year randomized parallel study, children with classic CAH (n=28) receiving low dose hydrocortisone and fludrocortisone plus flutamide and testolactone maintained normal growth velocity and skeletal maturation despite elevations in adrenal androgens.

    “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 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.

    “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]

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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.
ORCID
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
DOI
10.1210/jcem.85.3.6462
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
  • 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 › 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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