Short Report: Lack of Diurnal Variation in Salivary Cortisol Is Linked to Sleep Disturbances and Heightened Anxiety in Adolescents with Williams Syndrome

Overview

Journal Behav Sci (Basel)

Date 2023 Mar 28

PMID 36975245

Authors

Jessica Hayton

Atiqah Azhari

Gianluca Esposito

Ray Iles

Michaella Chadiarakos

Giulio Gabrieli

Dagmara Dimitriou

Stephen Mangar

Affiliations

Soon will be listed here.

Abstract

Objective: The aim of the current study was to examine the potential relationship between sleep patterns, cortisol levels, and anxiety profiles in adolescents with Williams Syndrome (WS) compared to typically developing adolescents.

Method: Thirteen adolescents with WS and thirteen TD adolescents (age range 12-18 years) were recruited. Participants were provided with a "testing kit", containing instructions for collecting data through a sleep diary, MotionWare actigraphy, the Childhood Sleep Habits Questionnaire (CSHQ), and the Spence Children's Anxiety Scale, and a salivary cortisol collection kit.

Results: Adolescents in the WS group did not show diurnal variation in salivary cortisol. Significantly higher scores were reported for two CSHQ subsections, night wakings and parasomnias, in the WS group. Regarding the actigraphy, only significantly longer sleep latency was observed in the WS group. In comparison to the TD group, the WS group had significantly higher anxiety. As expected, the TD group showed typical diurnal variation in cortisol, whereas the WS group showed a flattened cortisol profile throughout the day.

Conclusions: From the developmental perspective, this study provides new data supporting the conclusion that sleep problems are not transient but continue to persist into adolescence in WS. Future studies ought to consider examining the role of cortisol and its interplay with anxiety levels and sleep problems across the lifespan in individuals with WS.

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References

Annaz D, Hill C, Ashworth A, Holley S, Karmiloff-Smith A . Characterisation of sleep problems in children with Williams syndrome. Res Dev Disabil. 2010; 32(1):164-9. DOI: 10.1016/j.ridd.2010.09.008. View

Fredriksen K, Rhodes J, Reddy R, Way N . Sleepless in Chicago: tracking the effects of adolescent sleep loss during the middle school years. Child Dev. 2004; 75(1):84-95. DOI: 10.1111/j.1467-8624.2004.00655.x. View

Axelsson E, Hill C, Sadeh A, Dimitriou D . Sleep problems and language development in toddlers with Williams syndrome. Res Dev Disabil. 2013; 34(11):3988-96. DOI: 10.1016/j.ridd.2013.08.018. View

Spence S . A measure of anxiety symptoms among children. Behav Res Ther. 1998; 36(5):545-66. DOI: 10.1016/s0005-7967(98)00034-5. View

Woodruff-Borden J, Kistler D, Henderson D, Crawford N, Mervis C . Longitudinal course of anxiety in children and adolescents with Williams syndrome. Am J Med Genet C Semin Med Genet. 2010; 154C(2):277-90. PMC: 2914498. DOI: 10.1002/ajmg.c.30259. View

Shelton A, Malow B . Neurodevelopmental Disorders Commonly Presenting with Sleep Disturbances. Neurotherapeutics. 2021; 18(1):156-169. PMC: 8116361. DOI: 10.1007/s13311-020-00982-8. View

Stone A, Schwartz J, Smyth J, Kirschbaum C, Cohen S, Hellhammer D . Individual differences in the diurnal cycle of salivary free cortisol: a replication of flattened cycles for some individuals. Psychoneuroendocrinology. 2001; 26(3):295-306. DOI: 10.1016/s0306-4530(00)00057-3. View

Maume D . Social ties and adolescent sleep disruption. J Health Soc Behav. 2013; 54(4):498-515. DOI: 10.1177/0022146513498512. View

Lewin D, Rosen R, England S, Dahl R . Preliminary evidence of behavioral and cognitive sequelae of obstructive sleep apnea in children. Sleep Med. 2003; 3(1):5-13. DOI: 10.1016/s1389-9457(01)00070-3. View

10.

Markovich A, Gendron M, Corkum P . Validating the Children's Sleep Habits Questionnaire Against Polysomnography and Actigraphy in School-Aged Children. Front Psychiatry. 2015; 5:188. PMC: 4285019. DOI: 10.3389/fpsyt.2014.00188. View

11.

Capaldi Ii V, Handwerger K, Richardson E, Stroud L . Associations between sleep and cortisol responses to stress in children and adolescents: a pilot study. Behav Sleep Med. 2005; 3(4):177-92. DOI: 10.1207/s15402010bsm0304_1. View

12.

Royston R, Oliver C, Howlin P, Waite J . Anxiety characteristics in individuals with Williams syndrome. J Appl Res Intellect Disabil. 2021; 34(4):1098-1107. DOI: 10.1111/jar.12864. View

13.

Leyfer O, Woodruff-Borden J, Klein-Tasman B, Fricke J, Mervis C . Prevalence of psychiatric disorders in 4 to 16-year-olds with Williams syndrome. Am J Med Genet B Neuropsychiatr Genet. 2006; 141B(6):615-22. PMC: 2561212. DOI: 10.1002/ajmg.b.30344. View

14.

Carskadon M . Sleep in adolescents: the perfect storm. Pediatr Clin North Am. 2011; 58(3):637-47. PMC: 3130594. DOI: 10.1016/j.pcl.2011.03.003. View

15.

Sniecinska-Cooper A, Iles R, Butler S, Jones H, Bayford R, Dimitriou D . Abnormal secretion of melatonin and cortisol in relation to sleep disturbances in children with Williams syndrome. Sleep Med. 2014; 16(1):94-100. DOI: 10.1016/j.sleep.2014.09.003. View

16.

Mason T, Arens R, Sharman J, Bintliff-Janisak B, Schultz B, Walters A . Sleep in children with Williams Syndrome. Sleep Med. 2011; 12(9):892-7. PMC: 3210863. DOI: 10.1016/j.sleep.2011.05.003. View

17.

DSouza D, DSouza H, Horvath K, Plunkett K, Karmiloff-Smith A . Sleep is atypical across neurodevelopmental disorders in infants and toddlers: A cross-syndrome study. Res Dev Disabil. 2019; 97:103549. DOI: 10.1016/j.ridd.2019.103549. View

18.

Alfano C, Ginsburg G, Kingery J . Sleep-related problems among children and adolescents with anxiety disorders. J Am Acad Child Adolesc Psychiatry. 2007; 46(2):224-32. DOI: 10.1097/01.chi.0000242233.06011.8e. View

19.

Archbold K, Giordani B, Ruzicka D, Chervin R . Cognitive executive dysfunction in children with mild sleep-disordered breathing. Biol Res Nurs. 2004; 5(3):168-76. DOI: 10.1177/1099800403260261. View

20.

Ashworth A, Hill C, Karmiloff-Smith A, Dimitriou D . Sleep enhances memory consolidation in children. J Sleep Res. 2013; 23(3):302-8. DOI: 10.1111/jsr.12119. View