Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severity

Background: Immune dysregulation may play a role in the development of Rett syndrome (RTT), a neurodevelopmental disorder caused by mutations of the MECP2 gene. Abnormal cytokine concentrations have been documented in the serum of individuals with RTT. Measurement of salivary cytokines has been inve...

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Main Authors:	Breanne J. Byiers, Alyssa M. Merbler, Chantel C. Barney, Kristin A. Frenn, Angela Panoskaltsis-Mortari, Michael J. Ehrhardt, Timothy J. Feyma, Arthur A. Beisang, Frank Symons
Format:	Article
Language:	English
Published:	Elsevier 2020-01-01
Series:	Brain, Behavior, & Immunity - Health
Subjects:	Rett syndrome MECP2 Cytokines Saliva Biomarkers
Online Access:	http://www.sciencedirect.com/science/article/pii/S2666354619300080

id	doaj-eebacc8e255544558ee628c5441ce697
record_format	Article
spelling	doaj-eebacc8e255544558ee628c5441ce6972021-06-10T04:57:17ZengElsevierBrain, Behavior, & Immunity - Health2666-35462020-01-011100008Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severityBreanne J. Byiers0Alyssa M. Merbler1Chantel C. Barney2Kristin A. Frenn3Angela Panoskaltsis-Mortari4Michael J. Ehrhardt5Timothy J. Feyma6Arthur A. Beisang7Frank Symons8Department of Educational Psychology, 56 E River Rd, University of Minnesota, Minneapolis, MN, 55455, USA; Corresponding author. 56 E River Rd, Rm 250, Department of Educational Psychology, University of Minnesota, Minneapolis, MN, 55455, USA.Department of Educational Psychology, 56 E River Rd, University of Minnesota, Minneapolis, MN, 55455, USAGillette Children’s Specialty Healthcare, 200 University Ave E, St. Paul, Minnesota, 55101, USAGillette Children’s Specialty Healthcare, 200 University Ave E, St. Paul, Minnesota, 55101, USACancer Center and Department of Pediatrics, Division of Blood and Marrow Transplantation, 2450 Riverside Ave, University of Minnesota, Minneapolis, MN, 55454, USACancer Center and Department of Pediatrics, Division of Blood and Marrow Transplantation, 2450 Riverside Ave, University of Minnesota, Minneapolis, MN, 55454, USAGillette Children’s Specialty Healthcare, 200 University Ave E, St. Paul, Minnesota, 55101, USAGillette Children’s Specialty Healthcare, 200 University Ave E, St. Paul, Minnesota, 55101, USADepartment of Educational Psychology, 56 E River Rd, University of Minnesota, Minneapolis, MN, 55455, USABackground: Immune dysregulation may play a role in the development of Rett syndrome (RTT), a neurodevelopmental disorder caused by mutations of the MECP2 gene. Abnormal cytokine concentrations have been documented in the serum of individuals with RTT. Measurement of salivary cytokines has been investigated as a potential alternative approach to measurement in blood and serum, but it is unclear whether salivary cytokine concentrations can provide valid information about systemic immune function in neurodevelopmental disorders. The goal of this study was to evaluate the potential validity of salivary cytokines as biomarkers of immune dysregulation in RTT. Methods: Saliva samples from 16 individuals with RTT (all female; age range 2–40 years) and 16 healthy control females (age range 2–40 years) were analyzed for concentrations of 12 cytokines. Between-group differences in concentrations, and correlations with clinical severity in the RTT group were evaluated. Results: Concentrations of several salivary cytokines (IL-1β, IL-6, IL-8, IL-10, GM-CSF, TNF-α, and VEGF) were increased in RTT compared to controls. The same cytokines showed significant positive correlations with clinical severity scores. There were no differences in concentrations of IL-2, IL-4, IL-5, IL-12p70, and IFN-γ. Conclusion: The results suggest that salivary cytokines may be a possible indicator of immune dysregulation in RTT. Future research should investigate whether these results can be applied to other neurodevelopmental disorders.http://www.sciencedirect.com/science/article/pii/S2666354619300080Rett syndromeMECP2CytokinesSalivaBiomarkers
collection	DOAJ
language	English
format	Article
sources	DOAJ
author	Breanne J. Byiers Alyssa M. Merbler Chantel C. Barney Kristin A. Frenn Angela Panoskaltsis-Mortari Michael J. Ehrhardt Timothy J. Feyma Arthur A. Beisang Frank Symons
spellingShingle	Breanne J. Byiers Alyssa M. Merbler Chantel C. Barney Kristin A. Frenn Angela Panoskaltsis-Mortari Michael J. Ehrhardt Timothy J. Feyma Arthur A. Beisang Frank Symons Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severity Brain, Behavior, & Immunity - Health Rett syndrome MECP2 Cytokines Saliva Biomarkers
author_facet	Breanne J. Byiers Alyssa M. Merbler Chantel C. Barney Kristin A. Frenn Angela Panoskaltsis-Mortari Michael J. Ehrhardt Timothy J. Feyma Arthur A. Beisang Frank Symons
author_sort	Breanne J. Byiers
title	Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severity
title_short	Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severity
title_full	Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severity
title_fullStr	Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severity
title_full_unstemmed	Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severity
title_sort	evidence of altered salivary cytokine concentrations in rett syndrome and associations with clinical severity
publisher	Elsevier
series	Brain, Behavior, & Immunity - Health
issn	2666-3546
publishDate	2020-01-01
description	Background: Immune dysregulation may play a role in the development of Rett syndrome (RTT), a neurodevelopmental disorder caused by mutations of the MECP2 gene. Abnormal cytokine concentrations have been documented in the serum of individuals with RTT. Measurement of salivary cytokines has been investigated as a potential alternative approach to measurement in blood and serum, but it is unclear whether salivary cytokine concentrations can provide valid information about systemic immune function in neurodevelopmental disorders. The goal of this study was to evaluate the potential validity of salivary cytokines as biomarkers of immune dysregulation in RTT. Methods: Saliva samples from 16 individuals with RTT (all female; age range 2–40 years) and 16 healthy control females (age range 2–40 years) were analyzed for concentrations of 12 cytokines. Between-group differences in concentrations, and correlations with clinical severity in the RTT group were evaluated. Results: Concentrations of several salivary cytokines (IL-1β, IL-6, IL-8, IL-10, GM-CSF, TNF-α, and VEGF) were increased in RTT compared to controls. The same cytokines showed significant positive correlations with clinical severity scores. There were no differences in concentrations of IL-2, IL-4, IL-5, IL-12p70, and IFN-γ. Conclusion: The results suggest that salivary cytokines may be a possible indicator of immune dysregulation in RTT. Future research should investigate whether these results can be applied to other neurodevelopmental disorders.
topic	Rett syndrome MECP2 Cytokines Saliva Biomarkers
url	http://www.sciencedirect.com/science/article/pii/S2666354619300080
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Evidence of altered salivary cytokine concentrations in Rett syndrome and associations with clinical severity

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