Original Article Mifepristone: Effect on Plasma Corticotropin-Releasing Hormone, Adrenocorticotropic Hormone, and Cortisol in Term Pregnancy James D. Byrne, MD Deborah A. Wing, MD Mhoyra Fraser, PhD Michael J. Fassett, MD T. Murphy Goodwin, MD John R.G. Challis, PhD, Dsc OBJECTIVE: To compare the effects of in vivo mifepristone with placebo on plasma corticotropin-releasing hormone (CRH), adrenocorticotropic hormone (ACTH), and cortisol levels concentrations in term human pregnancies. STUDY DESIGN: In all, 24 women participating in a randomized controlled trial of mifepristone for preinduction cervical ripening were enrolled in this ancillary study. Participants with uncomplicated singleton pregnancies beyond 41 weeks gestation and undilated, uneffaced cervices were randomized to either placebo or mifepristone 200 mg orally and observed for 24 hours prior to receiving either intravaginal misoprostol and/or intravenous oxytocin. Blood samples were obtained before medication administration, 3 and 6 hours later, and then every 6 hours until delivery. Plasma hormone levels were measured by radioimmunoassay. RESULTS: Basal levels of CRH, ACTH, and cortisol were similar in the placebo (n ¼ 13) and mifepristone groups (n ¼ 11). Compared to placebo treatment, exposure to mifepristone resulted in significant elevation of plasma cortisol within 18 hours. Plasma CRH and ACTH were unaffected. Progression of labor was associated with significant increases in cortisol in both groups, while CRH and ACTH levels were not altered. Compared to basal levels within each group, plasma cortisol at delivery was Division of Maternal Fetal Medicine, (J.D.B., D.A.W., M.J.F., T.M.G.), Department of Obstetrics and Gynecology, University of Southern California Keck School of Medicine, Los Angeles, CA, USA; Liggins Institute for Medical Research (M.F.), Faculty of Medicine and Health Science, University of Auckland, Auckland, New Zealand; and Departments of Physiology and Obstetrics and Gynaecology (J.R.G.C.), University of Toronto, MRC Group In Fetal and Neonatal Health and Development, Toronto, Ontario, Canada. This work was supported by the Department of Obstetrics and Gynecology, University of Southern California School of Medicine and by MRC (CIHR) Canada Group Grant in Fetal and Neonatal Health and Development. Address correspondence and reprint requests to James D. Byrne, MD, Department of Obstetrics and Gynecology, Santa Clara Valley Medical Center, 751 South Bascom Avenue, San Jose, CA 95128, USA. significantly elevated within both the mifepristone (156.8±17.7 vs 332.6±48.5 ng/ml, p ¼ 0.008) and the placebo (166.6±34.3 vs 342.4±46.4 ng/ml, p ¼ 0.003) groups. However, plasma CRH, ACTH, and cortisol levels at delivery did not differ between the groups. CONCLUSION: Mifepristone exposure and induced labor were associated with significant increases in plasma cortisol without alterations of systemic CRH or ACTH levels. Journal of Perinatology (2004) 24, 416–420. doi:10.1038/sj.jp.7211127 Published online 20 May 2004 INTRODUCTION Despite intensive basic and clinical research, the factors regulating the duration of pregnancy and the onset of labor are poorly understood. Labor is initiated by a complex confluence of endocrine and paracrine mechanisms.1–3 In many mammals, parturition is preceded by elevation of fetal cortisol, which increases the expression of placental 17-a-hydroxylase/17,20-lyase. This results in progesterone withdrawal and enhanced estrogen output.1,3 Multiple parturition pathways are then activated that increase prostaglandins, oxytocin sensitivity, and calcium channels before culminating in labor. In contrast, humans lack placental 17-ahydroxylase/17,20-lyase activity. Therefore, systemic progesterone withdrawal does not occur prior to human labor despite an equivalent involvement of these same parturition pathways.3 Alternate models suggest that a functional withdrawal of local progesterone may occur at the placenta, thus initiating labor through the pathways mentioned above without altering circulating progesterone.3,4 In vitro research using term human placentas suggests that the increased cortisol levels associated with impending labor competitively block the inhibitory action of progesterone in regulating trophoblastic corticotropin-releasing hormone (CRH) messenger RNA, resulting in increased CRH levels.4 Karalis noted a similar elevation in CRH following mifepristone exposure, which was attributed to antagonism of the progesterone/glucocorticoid receptor. Karalis’s theory of functional progesterone withdrawal correlates with extensive in vitro evidence that placental CRH expression is inhibited by progesterone and stimulated by glucocorticoids.3,5–8 This has led to the speculation that a positive feedback loop involving cortisol and CRH may be involved in labor. Journal of Perinatology 2004; 24:416–420 r 2004 Nature Publishing Group All rights reserved. 0743-8346/04 $30 416 www.nature.com/jp Mifepristone in Term Pregnancy The report by Karalis provides an in vitro experimental model of human labor in which exposure to mifepristone results in local placental CRH elevation in a manner similar to what may naturally occur following competitive inhibition of progesterone by cortisol. Our objective was to assess this model in vivo by observing systemic plasma CRH, adrenocorticotropic hormone (ACTH), and cortisol levels after mifepristone exposure. MATERIALS AND METHODS This was an ancillary study to a randomized placebo-controlled trial evaluating mifepristone for preinduction cervical ripening.9 The study was performed at the University of Southern California School of Medicine. Women who met the criteria for entry and exclusion were invited to participate. Entry criteria were singleton gestation, cephalic presentation, intact membranes, gestational age more than 41 weeks by good dates, and reassuring fetal heart rate patterns. Exclusion criteria were Bishop score greater than 6, more than nine uterine contractions per hour, parity of six or more, prior uterine surgery, contraindications to prostaglandins, or evidence of complications such as infection, hypertension, intrauterine growth restriction, macrosomia, and vaginal bleeding. The primary study of preinduction cervical ripening was double masked and these methods have been reported previously.9 Briefly, subjects received a single dose of either mifepristone 200 mg or placebo (ascorbic acid) by mouth. Following administration of the medication, subjects were observed with continuous external electronic fetal monitoring for 24 hours. At the end of this observation period, women were re-evaluated and labor induction was continued with misoprostol or oxytocin according to the study protocol.9 Maternal venous blood was obtained prior to study medication administration (time 0 hours), at 3 hours, at 6 hours, and then every 6 hours until the final collection at delivery. The women were randomly chosen among eligible study candidates for participation in this adjunct. Blood was drawn into chilled heparinized tubes and immediately centrifuged at 2000 g revolutions/minute for 12 minutes at 41C. Plasma was separated into equal aliquots and stored at 701C until batched extraction and assay. The primary outcome measures were plasma hormones CRH, ACTH, and cortisol. Plasma CRH was measured by radioimmunoassay (RIA) as described by Marinoni et al.8 The lower limit of detection of the assay was 4.3 pmol/l and the intraand interassay coefficients of variation were 6.5 and 12%, respectively. ACTH was measured with a commercial RIA kit (Incstar, Stillwater, MI) with an interassay coefficient of variation of 10.6%. Cortisol was measured by RIA as reported previously.8 The interassay coefficient of variation was 11%. Sample size of was determined on the basis of data demonstrating a two-fold increase in CRH values following Journal of Perinatology 2004; 24:416–420 Byrne et al. mifepristone exposure in vitro.4 We hypothesized that the in vivo response would be less robust, and estimated that a minimum sample of nine women in each group would be adequate to detect an 80% differences in the mean plasma CRH level with a type I error of 0.05 and a type II error of 0.2. The test was one sided as prior in vivo and in vitro reports have never demonstrated a decrease in CRH values after exposure to mifepristone. Hormone results are expressed as mean±SEM. Owing to high individual variation, maternal plasma CRH values were also evaluated as the mean percent change from basal levels. Statistical analysis was performed with SPSS for Windows version 6.1.4 (SPSS Inc., Chicago, IL). Comparisons between normally distributed variables were performed with unpaired t-tests. As hormone levels were not normally distributed, comparisons between groups were performed using Mann–Whitney U-tests and within-group comparisons were made with Wilcoxon’s matched-pairs tests. Tests were two sided with p<0.05 statistically significant. An Investigational New Drug Application was filed with the Food and Drug Administration under the preinduction cervical ripening study. The institutional review board approved both studies. Written informed consent was obtained for each protocol. RESULTS In all, 24 women were enrolled: 13 received placebo and 11 received mifepristone. The two groups were similar with respect to maternal age, parity, gestational age, and birth weight (Table 1). The two groups were also similar with regard to intrapartum events such as misoprostol use and oxytocin administration, as well as route of delivery (Table 1). At enrollment, the mean baseline CRH level for all subjects was 3.8±0.3 ng/ml, mean ACTH was 30.1±4.7 pg/ml, and mean cortisol was 142.8±9.6 ng/ml. Baseline levels were similar in both the placebo and mifepristone groups for CRH (3.6±0.4 vs 4.0±0.5 ng/ml, p ¼ 0.67), ACTH (33.4±7.0 vs 26.1±6.1 pg/ml, p ¼ 0.32), and cortisol (133.5±10.9 vs 152.8±16.1 ng/ml, p ¼ 0.37). All women receiving mifepristone delivered within 42 hours of receiving the medication. Consequently, longitudinal comparisons between groups were limited to the first 36 hours after drug administration. Plasma CRH levels were not altered by mifepristone exposure or by labor. Compared to placebo, mifepristone exposure was not associated with alterations in plasma CRH levels when analyzed as either the mean value (Figure 1) or as the percentage change from basal levels (not presented). No significant CRH alterations from baseline levels occurred within the mifepristone group. Within the placebo group, match-paired CRH levels did not change from basal levels except for an isolated point at 18 hours. Progression of labor and delivery were not associated with significant alterations in plasma CRH levels. 417 Byrne et al. Mifepristone in Term Pregnancy Table 1 Maternal Characteristics Age (years) Parity Gestational age (weeks) Misoprostol use Oxytocin use Mean time from enrollment to active labor (minutes) Mean time from enrollment to delivery Route of delivery Vaginal Instrumented vaginal Cesarean Birth weight (g) Placebo (n ¼ 13) Mifepristone (n ¼ 11) P-value 24.4±1.6 1 (0–3) 41.3±0.1 11/13 (84.6%) 6/13 (46.2%) 2026.2±238.2 2620.6±283.0 29.3±1.9 1 (0–5) 41.2±0.1 9/11 (81.8%) 4/11 (36.4%) 2179.5±211.4 2473.4±216.2 0.07a 0.24b 0.40a 0.85c 0.63c 0.51d 0.69d 7 4 2 3665±167 10 1 0 3780±130 0.12c 0.60a Data presented as mean±SE the mean, median (range or n(5)). a Unpaired t-test. b Mann–Whitney U-test. c 2 w test. d Based on log-transformed data. Placebo Mifepristone 40 CRH ( ng/mL) ACTH (pg/mL) 50 30 20 6.0 5.5 10 * 5.0 4.5 4.0 3.5 3.0 0 3 6 12 18 Time (hr) 24 30 36 Figure 1. Longitudinal changes in plasma ACTH (top) and CRH (bottom). Asterisk, Placebo group CRH level compared to baseline p<0.05, Wilcoxon’s matched-pairs test. Points represent mean; error bars represent SEM. Plasma ACTH levels were not significantly altered by mifepristone exposure, progression of labor, or delivery (Figure 1). Plasma cortisol was significantly elevated following mifepristone exposure. Compared to placebo-treated women, plasma cortisol levels were significantly higher in the mifepristone-treated women beginning 18 hours after administration (Figure 2). Within the mifepristone group, matched-paired comparisons to basal levels revealed that cortisol began to rise 12 hours after administration of the medication. This increase was statistically significant at 18 hours and beyond. A similar trend was noted within the placebo group, but this did not achieve statistical significance during the first 36 hours of sampling. 418 In order to assess hormonal alterations associated with labor, data were evaluated using delivery as a common point of reference. Progression of labor was associated with significant increases in plasma cortisol in both groups. Mifepristone exposure was associated with higher levels of cortisol during labor were compared to placebo (Figure 3). This difference was statistically significant 24 hours (239.0±36.3 vs 153.5±17.7 ng/ml, p ¼ 0.04) and 6 hours (237.5±28.4 vs 171.5±25.72 ng/ml, p ¼ 0.04) prior to delivery. With-in-group matched-paired comparisons to basal levels revealed that plasma cortisol was significantly elevated at the time of delivery in both the mifepristone group (156.8±17.7 vs 332.6±48.5 ng/ml, p ¼ 0.008) and the placebo group (166.6±34.3 vs 342.4±46.4 ng/ml, p ¼ 0.003). Comparison between the mifepristone and placebo groups at delivery noted similar plasma hormone levels for CRH (3.4±0.7 vs 4.3±0.7 ng/ml, p ¼ 0.36), ACTH (26.4±5.9 vs 30.9±5.6 pg/ml, p ¼ 0.50), and cortisol (332.6±48.5 vs 342.4±46.4 ng/ml, p ¼ 0.55). SIGNIFICANCE Our data with regard to plasma CRH, ACTH, and cortisol in late pregnancy contributes to knowledge of endocrinology in pregnancy. This research focus is currently receiving significant investment due to wide clinical implications for maternal and fetal medicine. These include elucidation of potential etiologies and management options for diverse conditions, including preterm labor, intrauterine growth restriction, maternal stress responses and depression, and postnatal disease.3,10–12 Journal of Perinatology 2004; 24:416–420 Mifepristone in Term Pregnancy Byrne et al. 350 *# Placebo Mifepristone Cortisol (ng/mL) 275 *# *# *# 200 125 50 0 3 12 6 18 24 30 36 Time (hr) Figure 2. Longitudinal changes in plasma cortisol. Pound sign, placebo vs mifepristone p<0.05 by Mann–Whitney U-test. Asterisk, mifepristone group cortisol level compared to baseline, p<0.05 by Wilcoxon’s matched-pairs test. Points represent mean; error bars represent SEM. 500 Placebo Mifepristone Cortisol (ng/mL) 400 300 200 * 100 # *# ** * * ** ** ** ** ** 0 -42 -36 -30 -24 -18 -12 -6 -3 0 Time to Delivery (hr) Figure 3. Changes in plasma cortisol in relation to delivery. Pound sign, placebo vs mifepristone, p<0.05 by Mann–Whitney U-test. Cortisol levels compared to delivery noted for Placebo group (double asterisk) and mifepristone group (asterisk) with p<0.05 by Wilcoxon’s matched-pairs test. Points represent mean; error bars represent SEM. We noted baseline CRH levels at gestational age Z41 weeks that were elevated compared to values reported for term pregnancies, consistent with observations that placenta CRH expression increases with advancing gestational age.13–15 CRH levels varied widely between our subjects, a finding consistent with other studies.5,14,16,17 Individual variation in plasma CRH levels in pregnancy is not due to diurnal influences.15,18 Our study did not show elevations in systemic plasma CRH following in vivo mifepristone and may indicate that the CRH alterations observed by Karalis et al. in vitro occur only at a local level. Neither plasma CRH nor ACTH was altered by mifepristone exposure or by progression of labor. Our data corroborates studies Journal of Perinatology 2004; 24:416–420 by Cooper et al.,19,20 who observed that human placental CRH and ACTH immunostaining were not altered by oral mifepristone administration in the first trimester or by term labor. Despite progressive increases in CRH that peak prior to spontaneous labor, our collective data support the hypothesis that labor itself can progress without further increases in systemic CRH levels.13,14 Our findings correlate with those of Florio et al.,17 who found no increases in CRH levels in women in spontaneous labor. The same conclusion is supported for ACTH, refuting reports that levels increase with labor.21,22 Plasma cortisol increased significantly both with mifepristone exposure and with labor. We acknowledge that these latter results may be confounded by the subsequent administration of misoprostol, and in approximately half of the subjects, oxytocin. We were unable to analyze the influence of these two agents on plasma cortisol levels independently, as there was only one subject in the study who received neither oxytocin nor misoprostol. We suggest that the rise in cortisol may be due to alterations in placental cortisol metabolism, rather than to alterations in the maternal hypothalamic-pituitary-adrenal (HPA) axis induced by either mifepristone exposure or the ‘‘stress’’ of labor. It remains possible that there are changes in the concentrations of ACTH in maternal circulation during the course of labor that we cannot detect using current methodology. In addition, mifepristone exposure may have produced further subtle variations in ACTH that have biological effect, but were undetectable with the sampling and assay techniques we have used. This not withstanding, the lack of evidence for alterations in CRH and/or ACTH concentrations strongly suggests that the maternal HPA axis was not involved in the cortisol elevations we observed. In nonpregnant subjects, mifepristone acts within hours of administration to inhibit directly the negative feedback of cortisol on the HPA axis. This results in increased ACTH followed by increased systemic cortisol.23,24 A similar HPA axis response to ‘‘stress’’ has been suggested to explain the rise in plasma cortisol that accompanies the progression of labor.21 This too would be expected to result in ACTH elevation. However, despite elevations in plasma cortisol, we did not observe a sequence of an HPA cascade involving CRH or ACTH. Thus, the rise in cortisol observed is probably not due to direct action on the maternal HPA axis. We acknowledge the limitations of our study, including a small sample size and the possibility of beta-error. Since there are no previous studies of in vivo plasma CRH alteration following mifepristone exposure in term human pregnancy, our ability to calculate statistical power was limited to projections based on in vitro evidence. This in vitro data documented a two-fold increase in CRH following mifepristone. Even though we projected a less robust response, our data failed to demonstrate the 80% differences in the mean plasma CRH levels used in our power calculation. Owing to multiple factors, access to mifepristone for in vivo research involving human pregnancies remains highly restricted and subject 419 Byrne et al. to regulatory control. These limitations to access prevented us from pursing a study redesign and expansion of sample size following the analysis of our data. Lastly, our choice of placebo was based on cost and availability. Our reviews of the literature found little evidence that ascorbic acid administration would significantly affect plasma levels of the hormones in question. 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