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      Histrelin Implantation and Growth Outcomes in Children With Congenital Adrenal Hyperplasia: An Institutional Experience

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          Abstract

          Background

          Children with congenital adrenal hyperplasia (CAH) because of 21 hydroxylase deficiency (21OHD) are at risk for early or precocious puberty and a short adult height compared to population means and midparental height. The effect of histrelin in suppressing puberty and improving growth in these children has not been reported.

          Methods

          Retrospective cohort analysis of all patients (age ≤ 20) at our institution who underwent histrelin implantation between 2008 and 2017. Treated patients with CAH (classic and nonclassic forms of 21OHD) were identified and their growth data analyzed.

          Results

          Fifteen children with CAH were treated with histrelin for a median of 3 years (range 2–5; age at first implantation 7.7 ± 1.5 years). Bone age (BA) to chronologic age (CA) decreased from 1.57 ± 0.4 to 1.25 ± 0.25 ( P < .01), while predicted adult height (PAH) increased by 7.1 ± 6.6 cm ( P < .01). A subgroup of 10 children reached adult height. Similar changes in BA/CA and PAH were observed with therapy ( P = .02). Adult height z improved compared to pretreatment PAH z (–1.42 ± 0.9 vs. –1.96 ± 1.1 respectively, P < .01), but remained lower than midparental height z ( P = .01).

          Conclusion

          In this retrospective cohort study of children with CAH due to 21OHD and early or precocious puberty, histrelin implantation resulted in a decrease in BA progression compared to CA and an improvement in PAH. In the subgroup who completed growth, adult height remained significantly lower than midparental. These results need to be confirmed with prospective controlled studies.

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          Most cited references19

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          Clinical review: Adult height in patients with congenital adrenal hyperplasia: a systematic review and metaanalysis.

          Mohammad Hassan Murad, Patrick M. Erwin, Julianna Lampropulos … (2010)
          Treatment for patients with congenital adrenal hyperplasia (CAH) may affect the final height of these patients. Our objective was to determine the distribution of achieved height in patients with classic CAH diagnosed at infancy or early childhood and treated with glucocorticoids. We searched MEDLINE, EMBASE, Cochrane Library, ISI Web of Science, and Scopus through September 2008; the reference sections of included studies; and expert files. Eligible studies included patients diagnosed with CAH before age 5 and followed to final height. Reviewers working in duplicate independently extracted data on study characteristics and outcomes and determined each study's risk of bias. The sd score (SDS) for final height and corrected height (defined as final height SDS - midparental height SDS) were estimated from each study and pooled using random-effects metaanalysis. The I(2) statistic was used to assess inconsistency in results across studies. We found 35 eligible studies, most of which were retrospective single-cohort studies. The final height SDS achieved by CAH patients was -1.38 (-1.56 to -1.20; I(2) = 90.2%), and the corrected height SDS was -1.03 (-1.20 to -0.86; I(2) = 63.1%). This was not significantly associated with age at diagnosis, gender, type and dose of steroid, and age of onset of puberty. Mineralocorticoid users had a better height outcome in comparison with the nonusers (P = 0.02). Evidence derived from observational studies suggests that the final height of CAH patients treated with glucocorticoids is lower than the population norm and is lower than expected given parental height.
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            Final Height of Girls with Central Precocious Puberty, Untreated versus Treated with Cyproterone Acetate or GnRH Analogue

            R Kauli, A Galatzer, L Kornreich … (1997)
            This study was designed to determine the benefit of therapy on final height (FHt) in girls with central precocious puberty (CPP). A total of 102 patients were evaluated – 28 untreated, 26 treated with cyproterone acetate (CyA), and 48 treated with GnRH analogue (GnRHA) – and their achieved FHt was compared to the respective target height (THt). Of the untreated girls, half (14/28) had a slow course of puberty and reached THt ± 0.5 SD (FHt 160.2 ± 7.1, THt 159.5 ± 6.6 cm); the other half (14/28) had an accelerated course of puberty with a FHt well below THt (FHt 150.8 ± 4.3, THt 159.2 ± 5.9 cm) and in most cases (10/14) below the height-SDS of both parents. The treated girls (both regimens) reached THt or above (CyA group: FHt 157.8 ± 5.1, THt 156.8 ± 5. 1 cm; GnRHA group: 159.6 ± 6.3, THt 157.7 ± 5.7 cm). We conclude that without treatment the FHt of girls with CPP may be significantly compromised and that therapy is more beneficial if started before bone age exceeds 12 years. Our data also showed that for final height predictions in CPP the Bayley and Pinneau tables for average children should be used, regardless of the advanced bone age of the patients.
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              Hydrocortisone dosing during puberty in patients with classical congenital adrenal hyperplasia: an evidence-based recommendation.

              Dietrich Knorr, Maximilian Schmidt, Walter Bonfig … (2009)
              Patients with congenital adrenal hyperplasia (CAH) are at risk for early pubertal development and diminished pubertal growth. Liberal treatment with glucocorticoids will prevent early puberty but may inhibit growth outright. The aim of the study was to determine an optimal range for hydrocortisone dosing during puberty in children with classical CAH who were exclusively treated with hydrocortisone. The effects of glucocorticoid treatment for classical CAH were retrospectively analyzed in 92 patients (57 females). Growth pattern, final height (FH), and mean daily hydrocortisone dose were recorded. Pubertal growth was significantly reduced in all patients: salt-wasting (SW) females, 13.8 +/- 7.4 cm; simple virilizing (SV) females, 13.1 +/- 6.2 cm; vs. reference, 20.3 +/- 6.8 cm (P < 0.05); and SW males, 17.7 +/- 6.7 cm; SV males, 16.2 +/- 5.7 cm; vs. reference, 28.2 +/- 8.2 cm (P < 0.05). Decreased pubertal growth resulted in FH at the lower limit of genetic potential (corrected FH in SW females, -0.6 +/- 0.9; SV females, -0.3 +/- 0.9; SW males, -0.8 +/- 0.8; and SV males, -1.0 +/- 1.0). During puberty, mean daily hydrocortisone dose was 17.2 +/- 3.4 mg/m(2) in females (SW, 17.0 +/- 3.3; SV, 17.4 +/- 3.5) and 17.9 +/- 2.5 mg/m(2) in males (SW, 17.4 +/- 2.0; SV, 18.7 +/- 3.1). In a logistic regression model, a significant correlation between hydrocortisone dose and FH was found (P < 0.01), and the positive predictive value for short stature rose from below 30% to above 60% when hydrocortisone dose exceeded 17 mg/m(2). With conventional hydrocortisone treatment, pubertal growth is significantly reduced in both sexes, resulting in a FH at the lower limit of genetic potential. These deleterious effects on pubertal growth can be reduced if hydrocortisone does not exceed 17 mg/m(2).
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                Author and article information

                Contributors
                Maria G Vogiatzi:
                ORCID: http://orcid.org/0000-0002-1941-8277
                Journal
                Journal ID (nlm-ta): J Endocr Soc
                Journal ID (iso-abbrev): J Endocr Soc
                Journal ID (publisher-id): jes
                Title: Journal of the Endocrine Society
                Publisher: Oxford University Press (US )
                ISSN (Electronic): 2472-1972
                Publication date Collection: 01 February 2020
                Publication date (Electronic): 13 November 2019
                Publication date PMC-release: 13 November 2019
                Volume: 4
                Issue: 2
                Electronic Location Identifier: bvz004
                Affiliations
                [1 ] Department of Surgery, University of Pennsylvania , Philadelphia, PA, US
                [2 ] Department of Surgery, Columbia University , New York, NY, US
                [3 ] Division of Endocrinology and Diabetes, Children’s Hospital of Philadelphia , Philadelphia, PA, US
                [4 ] Division of General, Thoracic, and Fetal Surgery, Children’s Hospital of Philadelphia , Philadelphia, PA, US
                Author notes
                Correspondence:  Maria G Vogiatzi, MD, Endocrinology and Diabetes, Children’s Hospital of Philadelphia, 3500 Civic Center Blvd, Buerger Building # 12 165, Philadelphia, PA 19104. E-mail: vogiatzim@ 123456email.chop.edu .
                Author information
                http://orcid.org/0000-0002-1592-0120
                http://orcid.org/0000-0003-1810-9533
                http://orcid.org/0000-0002-1941-8277
                Article
                Publisher ID: bvz004
                DOI: 10.1210/jendso/bvz004
                PMC ID: 7035208
                SO-VID: 5345d0d0-78bc-49fe-812f-dfba0d311f7a
                Copyright © © Endocrine Society 2019.
                License:

                This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence ( http://creativecommons.org/licenses/by-nc-nd/4.0/), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

                History
                Date received : 16 July 2019
                Date accepted : 07 November 2019
                Date: 21 February 2020
                Page count
                Pages: 10
                Categories
                Subject: Clinical Research Article

                Keywords: cah,histrelin,leuprolide,final height,puberty
                Data availability:
                Keywords: cah, histrelin, leuprolide, final height, puberty

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