{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,16]],"date-time":"2026-10-16T06:21:57Z","timestamp":1792131717935,"version":"4.4.0"},"reference-count":93,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,3,16]],"date-time":"2023-03-16T00:00:00Z","timestamp":1678924800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Mol. Neurosci."],"abstract":"<jats:p>Autism is characterized by atypical social communication and stereotyped behaviors. Mutations in the gene encoding the synaptic scaffolding protein SHANK3 are detected in 1\u20132% of patients with autism and intellectual disability, but the mechanisms underpinning the symptoms remain largely unknown. Here, we characterized the behavior of <jats:italic>Shank3<\/jats:italic><jats:sup>\u039411\/\u039411<\/jats:sup> mice from 3 to 12 months of age. We observed decreased locomotor activity, increased stereotyped self-grooming and modification of socio-sexual interaction compared to wild-type littermates. We then used RNAseq on four brain regions of the same animals to identify differentially expressed genes (DEGs). DEGs were identified mainly in the striatum and were associated with synaptic transmission (e.g., <jats:italic>Grm2, Dlgap1<\/jats:italic>), G-protein-signaling pathways (e.g., <jats:italic>Gnal<\/jats:italic>, <jats:italic>Prkcg1<\/jats:italic>, <jats:italic>and Camk2g<\/jats:italic>), as well as excitation\/inhibition balance (e.g., <jats:italic>Gad2<\/jats:italic>). Downregulated and upregulated genes were enriched in the gene clusters of medium-sized spiny neurons expressing the dopamine 1 (D1-MSN) and the dopamine 2 receptor (D2-MSN), respectively. Several DEGs (<jats:italic>Cnr1<\/jats:italic>, <jats:italic>Gnal<\/jats:italic>, <jats:italic>Gad2<\/jats:italic>, <jats:italic>and Drd4<\/jats:italic>) were reported as striosome markers. By studying the distribution of the glutamate decarboxylase GAD65, encoded by <jats:italic>Gad2<\/jats:italic>, we showed that the striosome compartment of <jats:italic>Shank3<\/jats:italic><jats:sup>\u039411\/\u039411<\/jats:sup> mice was enlarged and displayed much higher expression of GAD65 compared to wild-type mice. Altogether, these results indicate altered gene expression in the striatum of <jats:italic>Shank3<\/jats:italic>-deficient mice and strongly suggest, for the first time, that the excessive self-grooming of these mice is related to an imbalance in the striatal striosome and matrix compartments.<\/jats:p>","DOI":"10.3389\/fnmol.2023.1139118","type":"journal-article","created":{"date-parts":[[2023,3,16]],"date-time":"2023-03-16T06:56:16Z","timestamp":1678949776000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":23,"title":["Excessive self-grooming, gene dysregulation and imbalance between the striosome and matrix compartments in the striatum of Shank3 mutant mice"],"prefix":"10.3389","volume":"16","author":[{"given":"Allain-Thibeault","family":"Ferhat","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisabeth","family":"Verpy","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne","family":"Biton","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beno\u00eet","family":"Forget","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fabrice","family":"De Chaumont","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Florian","family":"Mueller","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne-Marie","family":"Le Sourd","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sabrina","family":"Coqueran","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julien","family":"Schmitt","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christelle","family":"Rochefort","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laure","family":"Rondi-Reig","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aziliz","family":"Leboucher","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne","family":"Boland","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bertrand","family":"Fin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean-Fran\u00e7ois","family":"Deleuze","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tobias M.","family":"Boeckers","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elodie","family":"Ey","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Bourgeron","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2023,3,16]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"414","DOI":"10.1093\/bioinformatics\/btw623","article-title":"Combining multiple tools outperforms individual methods in gene set enrichment analyses","volume":"33","author":"Alhamdoosh","year":"2017","journal-title":"Bioinformatics"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"76","DOI":"10.1016\/j.fob.2012.04.003","article-title":"GeneSetDB: a comprehensive meta-database, statistical and visualisation framework for gene set analysis","volume":"2","author":"Araki","year":"2012","journal-title":"FEBS Open Bio"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1002\/syn.21955","article-title":"VTA DA neuron excitatory synapses in Shank3\u0394ex4\u20139 mouse line","volume":"71","author":"Bariselli","year":"2017","journal-title":"Synapse"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"926","DOI":"10.1038\/nn.4319","article-title":"SHANK3 controls maturation of social reward circuits in the VTA","volume":"19","author":"Bariselli","year":"2016","journal-title":"Nat. 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