ESPE Abstracts (2023) 97 P1-211

ESPE2023 Poster Category 1 Adrenals and HPA Axis (40 abstracts)

Salivary 11-oxygenated 19-carbon steroids in children with congenital adrenal hyperplasia and Addison's disease compared to healthy children

Julie Park 1,2 , Daniel Hawcutt 1,2 , Alena Shantsila 2,3 , Orla Bright 2 , Silothabo Dliso 1 , Gregory Lip 3,2 & Joanne Blair 1,2


1Alder Hey Children's NHS Foundation Trust, Liverpool, United Kingdom. 2University of Liverpool, Liverpool, United Kingdom. 3Liverpool Centre for Cardiovascular Science, Liverpool, United Kingdom


Background: 11-oxygenated 19-carbon (11oxC19) steroids, 11ketotestosterone (11KT) and 11βhydroxyandrostenedione(11OHA4) are adrenally derived steroids that rise in congenital adrenal hyperplasia (CAH). Increased 11oxC19 concentrations are associated with markers of poor control of CAH. To date, 11oxC19 concentrations have not been measured in patients with Addison’s disease (AD).

Methods: Children with primary adrenal insufficiency provided saliva samples at 9am, then 2-hourly throughout the day using Salivettes®. Children did not eat, drink or brush their teeth an hour prior to sampling. Salivary cortisol, cortisone, 17OHP, T, A4, 11KT and 11OHA4 were analysed. Results were compared to healthy controls matched for age and sex.

Results: 26 (15M), aged 2–18 years [21 with CAH (hydrocortisone dose 5.6-15.7mg/m2/day), 4 with AD (hydrocortisone dose 10.3-25mg/m2/day)] participated. One child, aged 2 years, could not provide samples. Salivary cortisol showed hydrocortisone contamination. Median salivary cortisone, in all groups, did not differ from healthy children. Salivary 17OHP was significantly higher in CAH compared to AD, P<0.001.

Table 1: Table showing salivary steroid hormone concentrations compared to healthy controls matched for age and sex
Salivary steroid CAH (n=21) Addison’s disease (n=4)
Cortisol High* High*
Cortisone No difference (NS) No difference (NS)
T Increased (P=0.03) Slightly increased (NS)
A4 Increased (P=0.03) Increased (P=0.03)
11KT Increased (P=0.03) Decreased (P=0.03)
11OHA4 Increased (P<0.01) No difference (NS)
* Likely contaminated with hydrocortisone NS: non-significant on Wilcoxon matched pairs signed rank test Of 24 samples 11KT was undetectable in 2 children with AD. No healthy children had undetectable concentrations of 11KT. Salivary 11OHA4 concentrations were similar (undetectable in 20/24(83.3%) samples in AD and 16/25(64%) samples in healthy children).

Discussion: Saliva samples are preferable for home monitoring of patients with CAH and AD, and multiple hormones can be measured on single samples. Salivary cortisol is unreliable in patients treated taking hydrocortisone, while salivary cortisone is more reliable. Salivary 11oxC19 steroids, T and A4 are elevated in CAH. The place of 11oxC19 steroids in monitoring of children with CAH, and optimal range for good control, are yet to be determined. Children with AD have significantly lower salivary 11KT and slightly increased salivary T compared to healthy children. Increased salivary T would be unexpected in AD as the HPG axis is not thought to be affected. Both T and 11KT are potent activators of the androgen receptor. It is possible that loss of negative feedback from 11KT may lead to increased free salivary testosterone.

Volume 97

61st Annual ESPE (ESPE 2023)

The Hague, Netherlands
21 Sep 2023 - 23 Sep 2023

European Society for Paediatric Endocrinology 

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