Associations Among Diurnal Salivary Cortisol Patterns, Medication Use, and Behavioral Phenotype Features in a Community Sample of Rett Syndrome

Am J Intellect Dev Disabil. 2020 Sep 1;125(5):353-368. doi: 10.1352/1944-7558-125.5.353.

Abstract

Rett syndrome (RTT) is a severe neurodevelopmental disorder resulting from mutations of the MECP2 gene. Hyperactivity of the hypothalamic-pituitary-adrenal (HPA) axis and abnormal stress responses have been observed in animal models of RTT, but little is known about HPA axis function among individuals with RTT. Diurnal salivary cortisol patterns from 30 females with RTT were examined in relation to mutation type, medication use, and features of the RTT behavioral phenotype. Cortisol patterns were significantly related to mutation severity, anticonvulsant medication status, and bruxism (tooth grinding). This study provides preliminary support for the hypothesis that RTT may be at risk for outcomes associated with aberrant HPA axis function, and that this risk may be mediated by mutation type.

Keywords: HPA axis; Rett syndrome; salivary cortisol; stress physiology.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Anticonvulsants / therapeutic use*
  • Circadian Rhythm / physiology
  • Female
  • Humans
  • Hydrocortisone
  • Hypothalamo-Hypophyseal System / metabolism*
  • Methyl-CpG-Binding Protein 2 / genetics
  • Phenotype
  • Rett Syndrome / drug therapy*
  • Rett Syndrome / genetics
  • Rett Syndrome / metabolism*
  • Rett Syndrome / physiopathology*
  • Saliva

Substances

  • Anticonvulsants
  • MECP2 protein, human
  • Methyl-CpG-Binding Protein 2
  • Hydrocortisone