Adrenocortical hyperresponsivity to adrenocorticotropic hormone: a mechanism favoring the normal production of cortisol in 21-hydroxylase-deficient nonclassic adrenal hyperplasia

Fertil Steril. 2000 Aug;74(2):329-34. doi: 10.1016/s0015-0282(00)00631-2.

Abstract

Objective: To test the hypothesis that patients with nonclassic adrenal hyperplasia (NCAH) exhibit a generalized exaggeration in their response to ACTH stimulation that favors the normal production of F. Patients with 21-hydroxylase (21-OH)-deficient NCAH do not demonstrate cortisol (F) deficiency.

Design: Prospective controlled study.

Setting: Tertiary university clinic.

Patient(s): Twenty-four untreated patients with NCAH diagnosed by a 17 alpha-hydroxyprogesterone (17-HP) level of >30.3 nmol/L (>10 ng/mL), and 37 age- and body mass-matched healthy eumenorrheic nonhirsute controls.

Intervention(s): All study subjects underwent a 60 minute acute stimulation using 0.25 mg of ACTH-(1-24) i.v.

Main outcome measure(s): Basal and stimulated serum levels of pregnenolone (PREG), 17-hydroxypregnenolone (17-HPREG), dehydroepiandrosterone (DHA), progesterone (P4), 17-HP, androstenedione (A4), 11-deoxycortisol (S), and cortisol (F).

Result(s): The median basal (i.e., Steroid(0)) or ACTH-stimulated (i. e., Steroid(60)) serum levels of PREG, 17-HPREG, DHA, P4, 17-HP, A4 and, most importantly, S were higher in NCAH patients than in controls. In contrast, the levels of F at either 0 minute or 60 minutes of stimulation were similar between NCAH and control women. The proportion of NCAH patients with stimulated steroids levels of >the 95th percentile of controls were as follows: 84.21% for PREG(60), 87.5% for 17-HPREG(60), 95.8% for DHA(60), 89.5% for P4(60), 100% for 17-HP(60), 91.7% for A4(60), 29.2% for S(60), and 4. 1% for F(60).

Conclusion(s): A generalized adrenocortical hyperresponsivity to ACTH stimulation seems to be present in patients with 21-OH-deficient NCAH, with an exaggerated production of S evident in approximately 30%. The excess production of S in these NCAH patients may, in part, account for their normal F production.

Publication types

  • Comparative Study

MeSH terms

  • 17-alpha-Hydroxypregnenolone / blood
  • Adrenal Cortex / drug effects
  • Adrenal Cortex / metabolism
  • Adrenal Hyperplasia, Congenital / drug therapy*
  • Adrenal Hyperplasia, Congenital / metabolism*
  • Adrenocorticotropic Hormone / pharmacology*
  • Adult
  • Androstenedione / blood
  • Case-Control Studies
  • Cortodoxone / blood
  • Dehydroepiandrosterone / blood
  • Female
  • Hirsutism / metabolism
  • Humans
  • Hydrocortisone / metabolism*
  • Pregnenolone / blood
  • Progesterone / blood
  • Prospective Studies
  • Steroid 21-Hydroxylase / drug effects*
  • Steroid 21-Hydroxylase / genetics

Substances

  • 17-alpha-Hydroxypregnenolone
  • Androstenedione
  • Dehydroepiandrosterone
  • Progesterone
  • Pregnenolone
  • Adrenocorticotropic Hormone
  • Steroid 21-Hydroxylase
  • Cortodoxone
  • Hydrocortisone