Metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder.

Since 2012, studies in mice, rats, and humans have suggested that abnormalities in purinergic signaling may be a final common pathway for many genetic and environmental causes of autism spectrum disorder (ASD). The current study in mice was conducted to characterize the bioenergetic, metabolomic, br...

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Main Authors: Zarazuela Zolkipli-Cunningham, Jane C Naviaux, Tomohiro Nakayama, Charlotte M Hirsch, Jonathan M Monk, Kefeng Li, Lin Wang, Thuy P Le, Simone Meinardi, Donald R Blake, Robert K Naviaux
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Language:English
Published: Public Library of Science (PLoS) 2021-01-01
Series:PLoS ONE
Online Access:https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0248771&type=printable
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author Zarazuela Zolkipli-Cunningham
Jane C Naviaux
Tomohiro Nakayama
Charlotte M Hirsch
Jonathan M Monk
Kefeng Li
Lin Wang
Thuy P Le
Simone Meinardi
Donald R Blake
Robert K Naviaux
author_facet Zarazuela Zolkipli-Cunningham
Jane C Naviaux
Tomohiro Nakayama
Charlotte M Hirsch
Jonathan M Monk
Kefeng Li
Lin Wang
Thuy P Le
Simone Meinardi
Donald R Blake
Robert K Naviaux
author_sort Zarazuela Zolkipli-Cunningham
collection DOAJ
description Since 2012, studies in mice, rats, and humans have suggested that abnormalities in purinergic signaling may be a final common pathway for many genetic and environmental causes of autism spectrum disorder (ASD). The current study in mice was conducted to characterize the bioenergetic, metabolomic, breathomic, and behavioral features of acute hyperpurinergia triggered by systemic injection of the purinergic agonist and danger signal, extracellular ATP (eATP). Responses were studied in C57BL/6J mice in the maternal immune activation (MIA) model and controls. Basal metabolic rates and locomotor activity were measured in CLAMS cages. Plasma metabolomics measured 401 metabolites. Breathomics measured 98 volatile organic compounds. Intraperitoneal eATP dropped basal metabolic rate measured by whole body oxygen consumption by 74% ± 6% (mean ± SEM) and rectal temperature by 6.2˚ ± 0.3˚C in 30 minutes. Over 200 metabolites from 37 different biochemical pathways where changed. Breathomics showed an increase in exhaled carbon monoxide, dimethylsulfide, and isoprene. Metabolomics revealed an acute increase in lactate, citrate, purines, urea, dopamine, eicosanoids, microbiome metabolites, oxidized glutathione, thiamine, niacinamide, and pyridoxic acid, and decreased folate-methylation-1-carbon intermediates, amino acids, short and medium chain acyl-carnitines, phospholipids, ceramides, sphingomyelins, cholesterol, bile acids, and vitamin D similar to some children with ASD. MIA animals were hypersensitive to postnatal exposure to eATP or poly(IC), which produced a rebound increase in body temperature that lasted several weeks before returning to baseline. Acute hyperpurinergia produced metabolic and behavioral changes in mice. The behaviors and metabolic changes produced by ATP injection were associated with mitochondrial functional changes that were profound but reversible.
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spelling doaj-art-dcd56786bb624e8fb59639e27974fd182025-08-20T02:17:50ZengPublic Library of Science (PLoS)PLoS ONE1932-62032021-01-01163e024877110.1371/journal.pone.0248771Metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder.Zarazuela Zolkipli-CunninghamJane C NaviauxTomohiro NakayamaCharlotte M HirschJonathan M MonkKefeng LiLin WangThuy P LeSimone MeinardiDonald R BlakeRobert K NaviauxSince 2012, studies in mice, rats, and humans have suggested that abnormalities in purinergic signaling may be a final common pathway for many genetic and environmental causes of autism spectrum disorder (ASD). The current study in mice was conducted to characterize the bioenergetic, metabolomic, breathomic, and behavioral features of acute hyperpurinergia triggered by systemic injection of the purinergic agonist and danger signal, extracellular ATP (eATP). Responses were studied in C57BL/6J mice in the maternal immune activation (MIA) model and controls. Basal metabolic rates and locomotor activity were measured in CLAMS cages. Plasma metabolomics measured 401 metabolites. Breathomics measured 98 volatile organic compounds. Intraperitoneal eATP dropped basal metabolic rate measured by whole body oxygen consumption by 74% ± 6% (mean ± SEM) and rectal temperature by 6.2˚ ± 0.3˚C in 30 minutes. Over 200 metabolites from 37 different biochemical pathways where changed. Breathomics showed an increase in exhaled carbon monoxide, dimethylsulfide, and isoprene. Metabolomics revealed an acute increase in lactate, citrate, purines, urea, dopamine, eicosanoids, microbiome metabolites, oxidized glutathione, thiamine, niacinamide, and pyridoxic acid, and decreased folate-methylation-1-carbon intermediates, amino acids, short and medium chain acyl-carnitines, phospholipids, ceramides, sphingomyelins, cholesterol, bile acids, and vitamin D similar to some children with ASD. MIA animals were hypersensitive to postnatal exposure to eATP or poly(IC), which produced a rebound increase in body temperature that lasted several weeks before returning to baseline. Acute hyperpurinergia produced metabolic and behavioral changes in mice. The behaviors and metabolic changes produced by ATP injection were associated with mitochondrial functional changes that were profound but reversible.https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0248771&type=printable
spellingShingle Zarazuela Zolkipli-Cunningham
Jane C Naviaux
Tomohiro Nakayama
Charlotte M Hirsch
Jonathan M Monk
Kefeng Li
Lin Wang
Thuy P Le
Simone Meinardi
Donald R Blake
Robert K Naviaux
Metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder.
PLoS ONE
title Metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder.
title_full Metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder.
title_fullStr Metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder.
title_full_unstemmed Metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder.
title_short Metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder.
title_sort metabolic and behavioral features of acute hyperpurinergia and the maternal immune activation mouse model of autism spectrum disorder
url https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0248771&type=printable
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