Altered brain serotonin 5-HT < sub > 1A < /sub > receptor expression and function in juvenile Fmr1 knockout mice
We examined agonist-labeled 5-HT1AR densities in male and female Fmr1 knockout mice, a mouse model of FXS, and found no difference in whole-brain 5-HT1AR expression in adult control compared to Fmr1 knockout mice. However, juvenile Fmr1 knockout mice had significantly lower whole-brain 5-HT1AR expression than age-matched controls. Consistent with these results, juvenile Fmr1 knockout mice showed significantly reduced behavioral responses elicited by the 5-HT1AR agonist (R)-8-OH-DPAT, effects blocked by the selective 5-HT1AR antagonist, WAY-100635. Also, treatment with the selective 5-HT1AR agonist, NLX-112, dose-dependentl...
Source: Neuropharmacology - November 3, 2023 Category: Drugs & Pharmacology Authors: Tanishka S Saraf Yiming Chen Richa Tyagi Clinton E Canal Source Type: research
Altered brain serotonin 5-HT < sub > 1A < /sub > receptor expression and function in juvenile Fmr1 knockout mice
We examined agonist-labeled 5-HT1AR densities in male and female Fmr1 knockout mice, a mouse model of FXS, and found no difference in whole-brain 5-HT1AR expression in adult control compared to Fmr1 knockout mice. However, juvenile Fmr1 knockout mice had significantly lower whole-brain 5-HT1AR expression than age-matched controls. Consistent with these results, juvenile Fmr1 knockout mice showed significantly reduced behavioral responses elicited by the 5-HT1AR agonist (R)-8-OH-DPAT, effects blocked by the selective 5-HT1AR antagonist, WAY-100635. Also, treatment with the selective 5-HT1AR agonist, NLX-112, dose-dependentl...
Source: Neuropharmacology - November 3, 2023 Category: Drugs & Pharmacology Authors: Tanishka S Saraf Yiming Chen Richa Tyagi Clinton E Canal Source Type: research
Altered brain serotonin 5-HT < sub > 1A < /sub > receptor expression and function in juvenile Fmr1 knockout mice
We examined agonist-labeled 5-HT1AR densities in male and female Fmr1 knockout mice, a mouse model of FXS, and found no difference in whole-brain 5-HT1AR expression in adult control compared to Fmr1 knockout mice. However, juvenile Fmr1 knockout mice had significantly lower whole-brain 5-HT1AR expression than age-matched controls. Consistent with these results, juvenile Fmr1 knockout mice showed significantly reduced behavioral responses elicited by the 5-HT1AR agonist (R)-8-OH-DPAT, effects blocked by the selective 5-HT1AR antagonist, WAY-100635. Also, treatment with the selective 5-HT1AR agonist, NLX-112, dose-dependentl...
Source: Neuropharmacology - November 3, 2023 Category: Drugs & Pharmacology Authors: Tanishka S Saraf Yiming Chen Richa Tyagi Clinton E Canal Source Type: research
An Exploratory Study of Pragmatic Language Use Across Contexts With the Pragmatic Rating Scale-School Age Among Autistic Boys and Boys With Fragile X Syndrome Plus Autism
CONCLUSIONS: Results from this study indicate the feasibility of using the PRS-SA in a shorter conversational context than the ADOS to assess pragmatic language among autistic boys. For boys with FXS + ASD, contextual differences warrant careful consideration in future work.PMID:37844621 | DOI:10.1044/2023_JSLHR-23-00024 (Source: Journal of speech, language, and hearing research : JSLHR)
Source: Journal of speech, language, and hearing research : JSLHR - October 16, 2023 Category: Speech-Language Pathology Authors: Nell Maltman Rebecca Willer Audra Sterling Source Type: research
An Exploratory Study of Pragmatic Language Use Across Contexts With the Pragmatic Rating Scale-School Age Among Autistic Boys and Boys With Fragile X Syndrome Plus Autism
CONCLUSIONS: Results from this study indicate the feasibility of using the PRS-SA in a shorter conversational context than the ADOS to assess pragmatic language among autistic boys. For boys with FXS + ASD, contextual differences warrant careful consideration in future work.PMID:37844621 | DOI:10.1044/2023_JSLHR-23-00024 (Source: Journal of speech, language, and hearing research : JSLHR)
Source: Journal of speech, language, and hearing research : JSLHR - October 16, 2023 Category: Speech-Language Pathology Authors: Nell Maltman Rebecca Willer Audra Sterling Source Type: research
Sensory Symptoms and Signs of Hyperarousal in Individuals with Fragile X Syndrome: Findings from the FORWARD Registry and Database Multisite Study
This study was designed to increase our understanding about characteristics and the impact of sensory symptoms (SS) and signs of hyperarousal (HA) in individuals with fragile X syndrome (FXS) from childhood through early adulthood and by gender. Data derived from the Fragile X Online Registry With Accessible Research Database (FORWARD), a natural history study of FXS, were analyzed using descriptive statistics and multivariate linear and logistic regression models to examine SS and signs of HA, their impact on behavioral regulation and limitations on the subject/family. The sample (N = 933) consisted of 720 males and 213 f...
Source: Journal of Autism and Developmental Disorders - October 15, 2023 Category: Psychiatry Authors: Ave M Lachiewicz Tracy M Stackhouse Kristin Burgess Debra Burgess Howard F Andrews Tse-Hwei Choo Walter E Kaufmann Sharon A Kidd Source Type: research
Sensory Symptoms and Signs of Hyperarousal in Individuals with Fragile X Syndrome: Findings from the FORWARD Registry and Database Multisite Study
This study was designed to increase our understanding about characteristics and the impact of sensory symptoms (SS) and signs of hyperarousal (HA) in individuals with fragile X syndrome (FXS) from childhood through early adulthood and by gender. Data derived from the Fragile X Online Registry With Accessible Research Database (FORWARD), a natural history study of FXS, were analyzed using descriptive statistics and multivariate linear and logistic regression models to examine SS and signs of HA, their impact on behavioral regulation and limitations on the subject/family. The sample (N = 933) consisted of 720 males and 213 f...
Source: Journal of Autism and Developmental Disorders - October 15, 2023 Category: Psychiatry Authors: Ave M Lachiewicz Tracy M Stackhouse Kristin Burgess Debra Burgess Howard F Andrews Tse-Hwei Choo Walter E Kaufmann Sharon A Kidd Source Type: research
Serum matrix metalloproteinase-9 (MMP9) and amyloid-beta protein precursor (APP) as potential biomarkers in children with Fragile-X syndrome: A cross sectional study
CONCLUSIONS: Screening for circulating biomarkers in the absence of FXS genetic diagnosis is justified. Our study is the first to evaluate both MMP9 and APP in FXS suspected children in a clinical setting and to assess their correlation with disease presentation and severity.PMID:37797798 | DOI:10.1016/j.clinbiochem.2023.110659 (Source: Clinical Biochemistry)
Source: Clinical Biochemistry - October 5, 2023 Category: Biochemistry Authors: Mohamed H Y Bekheet Lamiaa A Mansour Rasha H Elkaffas Mona A Kamel Mohamed A Elmonem Source Type: research
Serum matrix metalloproteinase-9 (MMP9) and amyloid-beta protein precursor (APP) as potential biomarkers in children with Fragile-X syndrome: A cross sectional study
CONCLUSIONS: Screening for circulating biomarkers in the absence of FXS genetic diagnosis is justified. Our study is the first to evaluate both MMP9 and APP in FXS suspected children in a clinical setting and to assess their correlation with disease presentation and severity.PMID:37797798 | DOI:10.1016/j.clinbiochem.2023.110659 (Source: Clinical Biochemistry)
Source: Clinical Biochemistry - October 5, 2023 Category: Biochemistry Authors: Mohamed H Y Bekheet Lamiaa A Mansour Rasha H Elkaffas Mona A Kamel Mohamed A Elmonem Source Type: research
Long-Term Safety and Sustained Efficacy of ZYN002 Cannabidiol Transdermal Gel in the Treatment of Behavioral Symptoms in Children and Adolescents With Fragile X Syndrome (ZYN2-CL-017)
The objective of this presentation is to assess the safety and efficacy of ZYN002, a pharmaceutically produced transdermal cannabidiol gel in development for fragile X syndrome (FXS). (Source: Journal of the American Academy of Child and Adolescent Psychiatry)
Source: Journal of the American Academy of Child and Adolescent Psychiatry - October 1, 2023 Category: Psychiatry Authors: Nancy Tich, Anthony Thibodeau, Terri Sebree, Thomas W. Dobbins, Stephen O'Quinn Source Type: research
RNA-Binding Proteins: A Role in Neurotoxicity?
Neurotox Res. 2023 Sep 30. doi: 10.1007/s12640-023-00669-w. Online ahead of print.ABSTRACTDespite sustained efforts to treat neurodegenerative diseases, little is known at the molecular level to understand and generate novel therapeutic approaches for these malignancies. Therefore, it is not surprising that neurogenerative diseases are among the leading causes of death in the aged population. Neurons require sophisticated cellular mechanisms to maintain proper protein homeostasis. These cells are generally sensitive to loss of gene expression control at the post-transcriptional level. Post-translational control responds to...
Source: Cancer Control - September 30, 2023 Category: Cancer & Oncology Authors: Andrea Ochar án-Mercado Jaqueline Loaeza-Loaeza Yaneth Castro-Coronel Leonor C Acosta-Saavedra Luisa C Hern ández-Kelly Daniel Hern ández-Sotelo Arturo Ortega Source Type: research
EEG Microstates as Markers for Cognitive Impairments in Fragile X Syndrome
Brain Topogr. 2023 Sep 26. doi: 10.1007/s10548-023-01009-z. Online ahead of print.ABSTRACTFragile X syndrome (FXS) is one of the most common inherited causes of intellectual disabilities. While there is currently no cure for FXS, EEG is considered an important method to investigate the pathophysiology and evaluate behavioral and cognitive treatments. We conducted EEG microstate analysis to investigate resting brain dynamics in FXS participants. Resting-state recordings from 70 FXS participants and 71 chronological age-matched typically developing control (TDC) participants were used to derive microstates via modified k-mea...
Source: Brain Topography - September 26, 2023 Category: Neuroscience Authors: Yukari Takarae Anthony Zanesco Craig A Erickson Ernest V Pedapati Source Type: research
EEG Microstates as Markers for Cognitive Impairments in Fragile X Syndrome
Brain Topogr. 2023 Sep 26. doi: 10.1007/s10548-023-01009-z. Online ahead of print.ABSTRACTFragile X syndrome (FXS) is one of the most common inherited causes of intellectual disabilities. While there is currently no cure for FXS, EEG is considered an important method to investigate the pathophysiology and evaluate behavioral and cognitive treatments. We conducted EEG microstate analysis to investigate resting brain dynamics in FXS participants. Resting-state recordings from 70 FXS participants and 71 chronological age-matched typically developing control (TDC) participants were used to derive microstates via modified k-mea...
Source: Brain Topography - September 26, 2023 Category: Neuroscience Authors: Yukari Takarae Anthony Zanesco Craig A Erickson Ernest V Pedapati Source Type: research
EEG Microstates as Markers for Cognitive Impairments in Fragile X Syndrome
Brain Topogr. 2023 Sep 26. doi: 10.1007/s10548-023-01009-z. Online ahead of print.ABSTRACTFragile X syndrome (FXS) is one of the most common inherited causes of intellectual disabilities. While there is currently no cure for FXS, EEG is considered an important method to investigate the pathophysiology and evaluate behavioral and cognitive treatments. We conducted EEG microstate analysis to investigate resting brain dynamics in FXS participants. Resting-state recordings from 70 FXS participants and 71 chronological age-matched typically developing control (TDC) participants were used to derive microstates via modified k-mea...
Source: Brain Topography - September 26, 2023 Category: Neuroscience Authors: Yukari Takarae Anthony Zanesco Craig A Erickson Ernest V Pedapati Source Type: research
EEG Microstates as Markers for Cognitive Impairments in Fragile X Syndrome
Brain Topogr. 2023 Sep 26. doi: 10.1007/s10548-023-01009-z. Online ahead of print.ABSTRACTFragile X syndrome (FXS) is one of the most common inherited causes of intellectual disabilities. While there is currently no cure for FXS, EEG is considered an important method to investigate the pathophysiology and evaluate behavioral and cognitive treatments. We conducted EEG microstate analysis to investigate resting brain dynamics in FXS participants. Resting-state recordings from 70 FXS participants and 71 chronological age-matched typically developing control (TDC) participants were used to derive microstates via modified k-mea...
Source: Brain Topography - September 26, 2023 Category: Neuroscience Authors: Yukari Takarae Anthony Zanesco Craig A Erickson Ernest V Pedapati Source Type: research
