{"database":"biostudies-literature","file_versions":[],"scores":null,"additional":{"submitter":["Nelson L"],"funding":["NIMH NIH HHS"],"pagination":["1667476"],"full_dataset_link":["https://www.ebi.ac.uk/biostudies/studies/S-EPMC12657441"],"repository":["biostudies-literature"],"omics_type":["Unknown"],"volume":["19"],"pubmed_abstract":["<h4>Introduction</h4>Fragile X Syndrome (FXS), the most common genetic cause of intellectual disability and autism spectrum disorder (ASD), results from silencing of the <i>FMR1</i> gene and consequent loss of Fragile X Messenger Ribonucleoprotein (FMRP). FMRP deficiency disrupts neural development, leading to behavioral and motor deficits associated with striatal dysfunction. Although structural and functional abnormalities in striatal projection neurons (SPNs) have been observed in adult <i>Fmr1</i> knockout mice (<i>Fmr1-/y</i>), their developmental onset and contribution to early FXS pathophysiology remain unknown.<h4>Methods</h4>We examined the postnatal maturation of SPNs in the dorsomedial striatum (DMS) of <i>Fmr1-/y</i> mice, assessing glutamatergic synaptic inputs and intrinsic e"],"journal":["Frontiers in cellular neuroscience"],"pubmed_title":["Delayed onset of striatal projection neuron hyperexcitability in &lt;i&gt;Fmr1-/y&lt;/i&gt; mice."],"pmcid":["PMC12657441"],"funding_grant_id":["T32 MH016804","R01 MH124695","R21 MH132015"],"pubmed_authors":["Matarazzo M","Shih YC","Peixoto RT","Janecek M","Nelson L"],"additional_accession":[]},"is_claimable":false,"name":"Delayed onset of striatal projection neuron hyperexcitability in &lt;i&gt;Fmr1-/y&lt;/i&gt; mice.","description":"<h4>Introduction</h4>Fragile X Syndrome (FXS), the most common genetic cause of intellectual disability and autism spectrum disorder (ASD), results from silencing of the <i>FMR1</i> gene and consequent loss of Fragile X Messenger Ribonucleoprotein (FMRP). FMRP deficiency disrupts neural development, leading to behavioral and motor deficits associated with striatal dysfunction. Although structural and functional abnormalities in striatal projection neurons (SPNs) have been observed in adult <i>Fmr1</i> knockout mice (<i>Fmr1-/y</i>), their developmental onset and contribution to early FXS pathophysiology remain unknown.<h4>Methods</h4>We examined the postnatal maturation of SPNs in the dorsomedial striatum (DMS) of <i>Fmr1-/y</i> mice, assessing glutamatergic synaptic inputs and intrinsic e","dates":{"release":"2025-01-01T00:00:00Z","publication":"2025","modification":"2026-06-07T03:15:54.726Z","creation":"2026-06-07T03:06:55.738Z"},"accession":"S-EPMC12657441","cross_references":{"pubmed":["41324081"],"doi":["10.3389/fncel.2025.1667476"]}}