Impact of hydrocortisone on adult height in congenital adrenal hyperplasia - The Minnesota cohort

Kyriakie Sarafoglou, O. Yaw Addo, Lucie Turcotte, Noelle Otten, Andrea Wickremasinghe, Siobhan Pittock, Jennifer Kyllo, Aida N. Lteif, John H. Himes, Bradley S. Miller

Research output: Contribution to journalArticle

16 Citations (Scopus)

Abstract

Objective To estimate the impact of the average daily dose of hydrocortisone (HC) on the amount of growth attained in children with congenital adrenal hyperplasia (CAH). The effect of glucocorticoid therapy on adult height (AH) in children with CAH has yet to be elucidated. Study design Triple-logistic models estimating components of growth and maturation were fitted to longitudinal records of 104 patients with classic CAH from 3 pediatric medical centers in Minnesota between 1955 and 2012. A total of 3664 clinical encounters were examined. Random-effects longitudinal models with time-related covariates were used to estimate the effect of HC therapy on linear growth. Results The predicted AH z-score (-0.7) was similar between the sexes and among CAH subtypes. The mean growth period HC dose was 18.9 ± 5.6 mg/m 2/day. In the final regression model, HC dose was negatively associated with predicted AH, with each mg/m2/day increase in average growth period HC dose predicting a 0.37-cm decrease in AH (P <.004). Conclusion This study has quantified the fractional reduction in predicted final AH with an incremental increase in HC dose. These findings have important clinical implications in the decision making balance between HC replacement dose and adrenal androgen suppression in children with CAH.

Original languageEnglish (US)
JournalJournal of Pediatrics
Volume164
Issue number5
DOIs
StatePublished - Jan 1 2014

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Congenital Adrenal Hyperplasia
Hydrocortisone
Growth
Glucocorticoids
Androgens
Decision Making
Logistic Models
Pediatrics
Therapeutics

Keywords

  • 21α-OHase
  • 21α-hydroxylase
  • AH
  • Adult height
  • CAH
  • CDC
  • Centers for Disease Control and Prevention
  • Congenital adrenal hyperplasia
  • HC
  • Hydrocortisone
  • OAH
  • Observed adult height
  • PAH
  • Predicted adult height
  • SV
  • SW
  • Salt-wasting
  • Simple-virilizing
  • βHC
  • βhydrocortisone

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Impact of hydrocortisone on adult height in congenital adrenal hyperplasia - The Minnesota cohort. / Sarafoglou, Kyriakie; Addo, O. Yaw; Turcotte, Lucie; Otten, Noelle; Wickremasinghe, Andrea; Pittock, Siobhan; Kyllo, Jennifer; Lteif, Aida N.; Himes, John H.; Miller, Bradley S.

In: Journal of Pediatrics, Vol. 164, No. 5, 01.01.2014.

Research output: Contribution to journalArticle

Sarafoglou, Kyriakie ; Addo, O. Yaw ; Turcotte, Lucie ; Otten, Noelle ; Wickremasinghe, Andrea ; Pittock, Siobhan ; Kyllo, Jennifer ; Lteif, Aida N. ; Himes, John H. ; Miller, Bradley S. / Impact of hydrocortisone on adult height in congenital adrenal hyperplasia - The Minnesota cohort. In: Journal of Pediatrics. 2014 ; Vol. 164, No. 5.
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AU - Otten, Noelle

AU - Wickremasinghe, Andrea

AU - Pittock, Siobhan

AU - Kyllo, Jennifer

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AU - Himes, John H.

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N2 - Objective To estimate the impact of the average daily dose of hydrocortisone (HC) on the amount of growth attained in children with congenital adrenal hyperplasia (CAH). The effect of glucocorticoid therapy on adult height (AH) in children with CAH has yet to be elucidated. Study design Triple-logistic models estimating components of growth and maturation were fitted to longitudinal records of 104 patients with classic CAH from 3 pediatric medical centers in Minnesota between 1955 and 2012. A total of 3664 clinical encounters were examined. Random-effects longitudinal models with time-related covariates were used to estimate the effect of HC therapy on linear growth. Results The predicted AH z-score (-0.7) was similar between the sexes and among CAH subtypes. The mean growth period HC dose was 18.9 ± 5.6 mg/m 2/day. In the final regression model, HC dose was negatively associated with predicted AH, with each mg/m2/day increase in average growth period HC dose predicting a 0.37-cm decrease in AH (P <.004). Conclusion This study has quantified the fractional reduction in predicted final AH with an incremental increase in HC dose. These findings have important clinical implications in the decision making balance between HC replacement dose and adrenal androgen suppression in children with CAH.

AB - Objective To estimate the impact of the average daily dose of hydrocortisone (HC) on the amount of growth attained in children with congenital adrenal hyperplasia (CAH). The effect of glucocorticoid therapy on adult height (AH) in children with CAH has yet to be elucidated. Study design Triple-logistic models estimating components of growth and maturation were fitted to longitudinal records of 104 patients with classic CAH from 3 pediatric medical centers in Minnesota between 1955 and 2012. A total of 3664 clinical encounters were examined. Random-effects longitudinal models with time-related covariates were used to estimate the effect of HC therapy on linear growth. Results The predicted AH z-score (-0.7) was similar between the sexes and among CAH subtypes. The mean growth period HC dose was 18.9 ± 5.6 mg/m 2/day. In the final regression model, HC dose was negatively associated with predicted AH, with each mg/m2/day increase in average growth period HC dose predicting a 0.37-cm decrease in AH (P <.004). Conclusion This study has quantified the fractional reduction in predicted final AH with an incremental increase in HC dose. These findings have important clinical implications in the decision making balance between HC replacement dose and adrenal androgen suppression in children with CAH.

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