Auditory brainstem response in infants and children with autism spectrum disorder: A meta‐analysis of wave V / Oren MIRON in Autism Research, 11-2 (February 2018)  

Public ISBD [article]  inAutism Research > 11-2 (February 2018) . - p.355-363 Titre : Auditory brainstem response in infants and children with autism spectrum disorder: A meta‐analysis of wave V Type de document : texte imprimé Auteurs : Oren MIRON, Auteur ; Andrew L. BEAM, Auteur ; Isaac S. KOHANE, Auteur Année de publication : 2018 Article en page(s) : p.355-363 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Infants with autism spectrum disorder (ASD) were recently found to have prolonged auditory brainstem response (ABR); however, at older ages, findings are contradictory. We compared ABR differences between participants with ASD and controls with respect to age using a meta‐analysis. Data sources included MEDLINE, EMBASE, Web of Science, Google Scholar, HOLLIS, and ScienceDirect from their inception to June 2016. The 25 studies that were included had a total of 1349 participants (727 participants with ASD and 622 controls) and an age range of 0–40 years. Prolongation of the absolute latency of wave V in ASD had a significant negative correlation with age (R2 = 0.23; P = 0.01). The 22 studies below age 18 years showed a significantly prolonged wave V in ASD (Standard Mean Difference = 0.6 [95% CI, 0.5–0.8]; P < 0.001). The 3 studies above 18 years of age showed a significantly shorter wave V in ASD (SMD = −0.6 [95% CI, −1.0 to −0.2]; P = 0.004). Prolonged ABR was consistent in infants and children with ASD, suggesting it can serve as an ASD biomarker at infancy. As the ABR is routinely used to screen infants for hearing impairment, the opportunity for replication studies is extensive. Autism Res 2018, 11: 355–363. © 2017 The Authors Autism Research published by International Society for Autism Research and Wiley Periodicals, Inc. Lay Summary Our analysis of previous studies showed that infants and children with autism spectrum disorder (ASD) have a slower brain response to sound, while adults have a faster brain response to sound. This suggests that slower brain response in infants may predict ASD risk. Brain response to sound is routinely tested on newborns to screen hearing impairment, which has created large data sets to afford replication of these results. En ligne : https://doi.org/10.1002/aur.1886 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=334 [article] Auditory brainstem response in infants and children with autism spectrum disorder: A meta‐analysis of wave V [texte imprimé] / Oren MIRON, Auteur ; Andrew L. BEAM, Auteur ; Isaac S. KOHANE, Auteur . - 2018 . - p.355-363. Langues : Anglais (eng) in Autism Research > 11-2 (February 2018) . - p.355-363 Index. décimale : PER Périodiques Résumé : Infants with autism spectrum disorder (ASD) were recently found to have prolonged auditory brainstem response (ABR); however, at older ages, findings are contradictory. We compared ABR differences between participants with ASD and controls with respect to age using a meta‐analysis. Data sources included MEDLINE, EMBASE, Web of Science, Google Scholar, HOLLIS, and ScienceDirect from their inception to June 2016. The 25 studies that were included had a total of 1349 participants (727 participants with ASD and 622 controls) and an age range of 0–40 years. Prolongation of the absolute latency of wave V in ASD had a significant negative correlation with age (R2 = 0.23; P = 0.01). The 22 studies below age 18 years showed a significantly prolonged wave V in ASD (Standard Mean Difference = 0.6 [95% CI, 0.5–0.8]; P < 0.001). The 3 studies above 18 years of age showed a significantly shorter wave V in ASD (SMD = −0.6 [95% CI, −1.0 to −0.2]; P = 0.004). Prolonged ABR was consistent in infants and children with ASD, suggesting it can serve as an ASD biomarker at infancy. As the ABR is routinely used to screen infants for hearing impairment, the opportunity for replication studies is extensive. Autism Res 2018, 11: 355–363. © 2017 The Authors Autism Research published by International Society for Autism Research and Wiley Periodicals, Inc. Lay Summary Our analysis of previous studies showed that infants and children with autism spectrum disorder (ASD) have a slower brain response to sound, while adults have a faster brain response to sound. This suggests that slower brain response in infants may predict ASD risk. Brain response to sound is routinely tested on newborns to screen hearing impairment, which has created large data sets to afford replication of these results. En ligne : https://doi.org/10.1002/aur.1886 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=334

Divergent dysregulation of gene expression in murine models of fragile X syndrome and tuberous sclerosis / Sek KONG in Molecular Autism, (February 2014)  

Public ISBD [article]  inMolecular Autism > (February 2014) Titre : Divergent dysregulation of gene expression in murine models of fragile X syndrome and tuberous sclerosis Type de document : texte imprimé Auteurs : Sek KONG, Auteur ; Mustafa SAHIN, Auteur ; Christin COLLINS, Auteur ; Mary WERTZ, Auteur ; Malcolm CAMPBELL, Auteur ; Jarrett LEECH, Auteur ; Dilja D. KRUEGER, Auteur ; Mark F. BEAR, Auteur ; Louis KUNKEL, Auteur ; Isaac S. KOHANE, Auteur Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Fragile X syndrome and tuberous sclerosis are genetic syndromes that both have a high rate of comorbidity with autism spectrum disorder (ASD). Several lines of evidence suggest that these two monogenic disorders may converge at a molecular level through the dysfunction of activity-dependent synaptic plasticity. To explore the characteristics of transcriptomic changes in these monogenic disorders, we profiled genome-wide gene expression levels in cerebellum and blood from murine models of fragile X syndrome and tuberous sclerosis. Differentially expressed genes and enriched pathways were distinct for the two murine models examined, with the exception of immune response-related pathways. In the cerebellum of the Fmr1 knockout (Fmr1-KO) model, the neuroactive ligand receptor interaction pathway and gene sets associated with synaptic plasticity such as long-term potentiation, gap junction, and axon guidance were the most significantly perturbed pathways. The phosphatidylinositol signaling pathway was significantly dysregulated in both cerebellum and blood of Fmr1-KO mice. In Tsc2 heterozygous (+/) mice, immune system-related pathways, genes encoding ribosomal proteins, and glycolipid metabolism pathways were significantly changed in both tissues. Our data suggest that distinct molecular pathways may be involved in ASD with known but different genetic causes and that blood gene expression profiles of Fmr1-KO and Tsc2+/ mice mirror some, but not all, of the perturbed molecular pathways in the brain. En ligne : http://dx.doi.org/10.1186/2040-2392-5-16 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=227 [article] Divergent dysregulation of gene expression in murine models of fragile X syndrome and tuberous sclerosis [texte imprimé] / Sek KONG, Auteur ; Mustafa SAHIN, Auteur ; Christin COLLINS, Auteur ; Mary WERTZ, Auteur ; Malcolm CAMPBELL, Auteur ; Jarrett LEECH, Auteur ; Dilja D. KRUEGER, Auteur ; Mark F. BEAR, Auteur ; Louis KUNKEL, Auteur ; Isaac S. KOHANE, Auteur. Langues : Anglais (eng) in Molecular Autism > (February 2014) Index. décimale : PER Périodiques Résumé : Fragile X syndrome and tuberous sclerosis are genetic syndromes that both have a high rate of comorbidity with autism spectrum disorder (ASD). Several lines of evidence suggest that these two monogenic disorders may converge at a molecular level through the dysfunction of activity-dependent synaptic plasticity. To explore the characteristics of transcriptomic changes in these monogenic disorders, we profiled genome-wide gene expression levels in cerebellum and blood from murine models of fragile X syndrome and tuberous sclerosis. Differentially expressed genes and enriched pathways were distinct for the two murine models examined, with the exception of immune response-related pathways. In the cerebellum of the Fmr1 knockout (Fmr1-KO) model, the neuroactive ligand receptor interaction pathway and gene sets associated with synaptic plasticity such as long-term potentiation, gap junction, and axon guidance were the most significantly perturbed pathways. The phosphatidylinositol signaling pathway was significantly dysregulated in both cerebellum and blood of Fmr1-KO mice. In Tsc2 heterozygous (+/) mice, immune system-related pathways, genes encoding ribosomal proteins, and glycolipid metabolism pathways were significantly changed in both tissues. Our data suggest that distinct molecular pathways may be involved in ASD with known but different genetic causes and that blood gene expression profiles of Fmr1-KO and Tsc2+/ mice mirror some, but not all, of the perturbed molecular pathways in the brain. En ligne : http://dx.doi.org/10.1186/2040-2392-5-16 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=227

Gene expression analysis in Fmr1KO mice identifies an immunological signature in brain tissue and mGluR5-related signaling in primary neuronal cultures / Daria PRILUTSKY in Molecular Autism, (December 2015)  

Public ISBD [article]  inMolecular Autism > (December 2015) . - p.1-14 Titre : Gene expression analysis in Fmr1KO mice identifies an immunological signature in brain tissue and mGluR5-related signaling in primary neuronal cultures Type de document : texte imprimé Auteurs : Daria PRILUTSKY, Auteur ; Alvin T. KHO, Auteur ; Nathan P. PALMER, Auteur ; Asha L. BHAKAR, Auteur ; Niklas SMEDEMARK-MARGULIES, Auteur ; Sek Won KONG, Auteur ; David M. MARGULIES, Auteur ; Mark F. BEAR, Auteur ; Isaac S. KOHANE, Auteur Article en page(s) : p.1-14 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS) is a neurodevelopmental disorder whose biochemical manifestations involve dysregulation of mGluR5-dependent pathways, which are widely modeled using cultured neurons. In vitro phenotypes in cultured neurons using standard morphological, functional, and chemical approaches have demonstrated considerable variability. Here, we study transcriptomes obtained in situ in the intact brain tissues of a murine model of FXS to see how they reflect the in vitro state. En ligne : http://dx.doi.org/10.1186/s13229-015-0061-9 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=277 [article] Gene expression analysis in Fmr1KO mice identifies an immunological signature in brain tissue and mGluR5-related signaling in primary neuronal cultures [texte imprimé] / Daria PRILUTSKY, Auteur ; Alvin T. KHO, Auteur ; Nathan P. PALMER, Auteur ; Asha L. BHAKAR, Auteur ; Niklas SMEDEMARK-MARGULIES, Auteur ; Sek Won KONG, Auteur ; David M. MARGULIES, Auteur ; Mark F. BEAR, Auteur ; Isaac S. KOHANE, Auteur . - p.1-14. Langues : Anglais (eng) in Molecular Autism > (December 2015) . - p.1-14 Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS) is a neurodevelopmental disorder whose biochemical manifestations involve dysregulation of mGluR5-dependent pathways, which are widely modeled using cultured neurons. In vitro phenotypes in cultured neurons using standard morphological, functional, and chemical approaches have demonstrated considerable variability. Here, we study transcriptomes obtained in situ in the intact brain tissues of a murine model of FXS to see how they reflect the in vitro state. En ligne : http://dx.doi.org/10.1186/s13229-015-0061-9 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=277

Prolonged Auditory Brainstem Response in Universal Hearing Screening of Newborns with Autism Spectrum Disorder / Oren MIRON in Autism Research, 14-1 (January 2021)  

Public ISBD [article]  inAutism Research > 14-1 (January 2021) . - p.46-52 Titre : Prolonged Auditory Brainstem Response in Universal Hearing Screening of Newborns with Autism Spectrum Disorder Type de document : texte imprimé Auteurs : Oren MIRON, Auteur ; Rafael E. DELGADO, Auteur ; Christine F. DELGADO, Auteur ; Elizabeth A. SIMPSON, Auteur ; Kun-Hsing YU, Auteur ; Anibal GUTIERREZ, Auteur ; Guangyu ZENG, Auteur ; Jillian N. GERSTENBERGER, Auteur ; Isaac S. KOHANE, Auteur Article en page(s) : p.46-52 Langues : Anglais (eng) Mots-clés : auditory  biomarker  children  event-related potential  infants Index. décimale : PER Périodiques Résumé : Previous studies report prolonged auditory brainstem response (ABR) in children and adults with autism spectrum disorder (ASD). Despite its promise as a biomarker, it is unclear whether healthy newborns who later develop ASD also show ABR abnormalities. In the current study, we extracted ABR data on 139,154 newborns from their Universal Newborn Hearing Screening, including 321 newborns who were later diagnosed with ASD. We found that the ASD newborns had significant prolongations of their ABR phase and V-negative latency compared with the non-ASD newborns. Newborns in the ASD group also exhibited greater variance in their latencies compared to previous studies in older ASD samples, likely due in part to the low intensity of the ABR stimulus. These findings suggest that newborns display neurophysiological variation associated with ASD at birth. Future studies with higher-intensity stimulus ABRs may allow more accurate predictions of ASD risk, which could augment the universal ABR test that currently screens millions of newborns worldwide. LAY SUMMARY: Children with autism spectrum disorder (ASD) have slow brain responses to sounds. We examined these brain responses from newborns' hearing tests and found that newborns who were later diagnosed with autism also had slower brain responses to sounds. Future studies might use these findings to better predict autism risk, with a hearing test that is already used on millions of newborns worldwide. En ligne : http://dx.doi.org/10.1002/aur.2422 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=441 [article] Prolonged Auditory Brainstem Response in Universal Hearing Screening of Newborns with Autism Spectrum Disorder [texte imprimé] / Oren MIRON, Auteur ; Rafael E. DELGADO, Auteur ; Christine F. DELGADO, Auteur ; Elizabeth A. SIMPSON, Auteur ; Kun-Hsing YU, Auteur ; Anibal GUTIERREZ, Auteur ; Guangyu ZENG, Auteur ; Jillian N. GERSTENBERGER, Auteur ; Isaac S. KOHANE, Auteur . - p.46-52. Langues : Anglais (eng) in Autism Research > 14-1 (January 2021) . - p.46-52 Mots-clés : auditory  biomarker  children  event-related potential  infants Index. décimale : PER Périodiques Résumé : Previous studies report prolonged auditory brainstem response (ABR) in children and adults with autism spectrum disorder (ASD). Despite its promise as a biomarker, it is unclear whether healthy newborns who later develop ASD also show ABR abnormalities. In the current study, we extracted ABR data on 139,154 newborns from their Universal Newborn Hearing Screening, including 321 newborns who were later diagnosed with ASD. We found that the ASD newborns had significant prolongations of their ABR phase and V-negative latency compared with the non-ASD newborns. Newborns in the ASD group also exhibited greater variance in their latencies compared to previous studies in older ASD samples, likely due in part to the low intensity of the ABR stimulus. These findings suggest that newborns display neurophysiological variation associated with ASD at birth. Future studies with higher-intensity stimulus ABRs may allow more accurate predictions of ASD risk, which could augment the universal ABR test that currently screens millions of newborns worldwide. LAY SUMMARY: Children with autism spectrum disorder (ASD) have slow brain responses to sounds. We examined these brain responses from newborns' hearing tests and found that newborns who were later diagnosed with autism also had slower brain responses to sounds. Future studies might use these findings to better predict autism risk, with a hearing test that is already used on millions of newborns worldwide. En ligne : http://dx.doi.org/10.1002/aur.2422 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=441

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