{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T22:55:53Z","timestamp":1778108153119,"version":"3.51.4"},"reference-count":113,"publisher":"Springer Science and Business Media LLC","issue":"13","license":[{"start":{"date-parts":[[2024,9,2]],"date-time":"2024-09-02T00:00:00Z","timestamp":1725235200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.springernature.com\/gp\/researchers\/text-and-data-mining"},{"start":{"date-parts":[[2024,9,2]],"date-time":"2024-09-02T00:00:00Z","timestamp":1725235200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/www.springernature.com\/gp\/researchers\/text-and-data-mining"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Neuropsychopharmacol."],"published-print":{"date-parts":[[2024,12]]},"DOI":"10.1038\/s41386-024-01956-6","type":"journal-article","created":{"date-parts":[[2024,9,2]],"date-time":"2024-09-02T15:02:44Z","timestamp":1725289364000},"page":"2032-2041","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Therapeutic efficacy of the BKCa channel opener chlorzoxazone in a mouse model of Fragile X syndrome"],"prefix":"10.1038","volume":"49","author":[{"given":"Celeste","family":"Ferraguto","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marion","family":"Piquemal-Lagoueillat","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Valerie","family":"Lemaire","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ma\u00eft\u00e9 M.","family":"Moreau","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2623-2769","authenticated-orcid":false,"given":"Stefania","family":"Trazzi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Beatrice","family":"Uguagliati","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Elisabetta","family":"Ciani","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sandrine S.","family":"Bertrand","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eric","family":"Louette","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bruno","family":"Bontempi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Susanna","family":"Pietropaolo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2024,9,2]]},"reference":[{"key":"1956_CR1","doi-asserted-by":"publisher","first-page":"817","DOI":"10.1016\/0092-8674(91)90125-I","volume":"66","author":"M Pieretti","year":"1991","unstructured":"Pieretti M, Zhang FP, Fu YH, Warren ST, Oostra BA, Caskey CT, et al. Absence of expression of the FMR-1 gene in fragile X syndrome. Cell. 1991;66:817\u201322.","journal-title":"Cell"},{"key":"1956_CR2","doi-asserted-by":"publisher","first-page":"219","DOI":"10.1146\/annurev-pathol-011811-132457","volume":"7","author":"MR Santoro","year":"2012","unstructured":"Santoro MR, Bray SM, Warren ST. Molecular mechanisms of fragile X syndrome: a twenty-year perspective. Annu Rev Pathol. 2012;7:219\u201345.","journal-title":"Annu Rev Pathol"},{"key":"1956_CR3","doi-asserted-by":"publisher","first-page":"7101","DOI":"10.1073\/pnas.141145998","volume":"98","author":"WT Greenough","year":"2001","unstructured":"Greenough WT, Klintsova AY, Irwin SA, Galvez R, Bates KE, Weiler IJ. Synaptic regulation of protein synthesis and the fragile X protein. Proc Natl Acad Sci USA. 2001;98:7101\u20136.","journal-title":"Proc Natl Acad Sci USA"},{"key":"1956_CR4","doi-asserted-by":"publisher","first-page":"337","DOI":"10.1159\/000329421","volume":"33","author":"MR Tranfaglia","year":"2011","unstructured":"Tranfaglia MR. The psychiatric presentation of fragile X: evolution of the diagnosis and treatment of the psychiatric comorbidities of fragile X syndrome. Dev Neurosci. 2011;33:337\u201348.","journal-title":"Dev Neurosci"},{"key":"1956_CR5","doi-asserted-by":"publisher","first-page":"248","DOI":"10.1007\/s13311-022-01183-1","volume":"19","author":"R Aishworiya","year":"2022","unstructured":"Aishworiya R, Valica T, Hagerman R, Restrepo B. An update on psychopharmacological treatment of autism spectrum disorder. Neurotherapeutics. 2022;19:248\u201362. https:\/\/doi.org\/10.1007\/s13311-022-01183-1","journal-title":"Neurotherapeutics"},{"key":"1956_CR6","doi-asserted-by":"publisher","first-page":"225","DOI":"10.1007\/978-3-031-21054-9_10","volume":"30","author":"D Johnson","year":"2023","unstructured":"Johnson D, Clark C, Hagerman R. Targeted treatments for fragile X syndrome. Adv Neurobiol. 2023;30:225\u201353. https:\/\/doi.org\/10.1007\/978-3-031-21054-9_10","journal-title":"Adv Neurobiol"},{"key":"1956_CR7","doi-asserted-by":"publisher","first-page":"251","DOI":"10.2174\/1573396315666190625110748","volume":"15","author":"D Protic","year":"2019","unstructured":"Protic D, Salcedo-Arellano MJ, Dy JB, Potter LA, Hagerman RJ. New targeted treatments for fragile X syndrome. Curr Pediatr Rev. 2019;15:251\u20138. https:\/\/doi.org\/10.2174\/1573396315666190625110748","journal-title":"Curr Pediatr Rev"},{"key":"1956_CR8","doi-asserted-by":"publisher","first-page":"264","DOI":"10.1016\/j.nurt.2010.05.005","volume":"7","author":"LW Wang","year":"2010","unstructured":"Wang LW, Berry-Kravis E, Hagerman RJ. Fragile X: leading the way for targeted treatments in autism. Neurotherapeutics. 2010;7:264\u201374.","journal-title":"Neurotherapeutics"},{"key":"1956_CR9","doi-asserted-by":"publisher","first-page":"270","DOI":"10.3389\/fnmol.2018.00270","volume":"11","author":"TH Wen","year":"2018","unstructured":"Wen TH, Binder DK, Ethell IM, Razak KA. The perineuronal \u2018safety\u2019 net? Perineuronal net abnormalities in neurological disorders. Front Mol Neurosci. 2018;11:270 https:\/\/doi.org\/10.3389\/fnmol.2018.00270","journal-title":"Front Mol Neurosci"},{"key":"1956_CR10","doi-asserted-by":"publisher","first-page":"674","DOI":"10.1038\/nm.4335","volume":"23","author":"I Gantois","year":"2017","unstructured":"Gantois I, Khoutorsky A, Popic J, Aguilar-Valles A, Freemantle E, Cao R, et al. Metformin ameliorates core deficits in a mouse model of fragile X syndrome. Nat Med. 2017;23:674\u20137. https:\/\/doi.org\/10.1038\/nm.4335","journal-title":"Nat Med"},{"key":"1956_CR11","doi-asserted-by":"publisher","first-page":"1742","DOI":"10.1016\/j.celrep.2014.10.064","volume":"9","author":"CG Gkogkas","year":"2014","unstructured":"Gkogkas CG, Khoutorsky A, Cao R, Jafarnejad SM, Prager-Khoutorsky M, Giannakas N, et al. Pharmacogenetic inhibition of eIF4E-dependent Mmp9 mRNA translation reverses fragile X syndrome-like phenotypes. Cell Rep. 2014;9:1742\u201355. https:\/\/doi.org\/10.1016\/j.celrep.2014.10.064","journal-title":"Cell Rep"},{"key":"1956_CR12","doi-asserted-by":"publisher","first-page":"141","DOI":"10.3389\/fnbeh.2019.00141","volume":"13","author":"P Cogram","year":"2019","unstructured":"Cogram P, Deacon RMJ, Warner-Schmidt JL, von Schimmelmann MJ, Abrahams BS, During MJ. Gaboxadol normalizes behavioral abnormalities in a mouse model of fragile X syndrome. Front Behav Neurosci. 2019;13:141. https:\/\/doi.org\/10.3389\/fnbeh.2019.00141","journal-title":"Front Behav Neurosci"},{"key":"1956_CR13","doi-asserted-by":"publisher","first-page":"395","DOI":"10.1159\/000332884","volume":"33","author":"JL Olmos-Serrano","year":"2011","unstructured":"Olmos-Serrano JL, Corbin JG, Burns MP. The GABA(A) receptor agonist THIP ameliorates specific behavioral deficits in the mouse model of fragile X syndrome. Dev Neurosci. 2011;33:395\u2013403.","journal-title":"Dev Neurosci"},{"key":"1956_CR14","doi-asserted-by":"publisher","first-page":"30","DOI":"10.1016\/j.coph.2005.10.004","volume":"6","author":"KA Wafford","year":"2006","unstructured":"Wafford KA, Ebert B. Gaboxadol-a new awakening in sleep. Curr Opin Pharm. 2006;6:30\u20136. https:\/\/doi.org\/10.1016\/j.coph.2005.10.004","journal-title":"Curr Opin Pharm"},{"key":"1956_CR15","doi-asserted-by":"publisher","first-page":"55","DOI":"10.1002\/med.2610050103","volume":"5","author":"G Bartholini","year":"1985","unstructured":"Bartholini G. GABA receptor agonists: pharmacological spectrum and therapeutic actions. Med Res Rev. 1985;5:55\u201375. https:\/\/doi.org\/10.1002\/med.2610050103","journal-title":"Med Res Rev"},{"key":"1956_CR16","doi-asserted-by":"publisher","first-page":"373","DOI":"10.3389\/fphys.2014.00373","volume":"5","author":"P N\u2019Gouemo","year":"2014","unstructured":"N\u2019Gouemo P. BKCa channel dysfunction in neurological diseases. Front Physiol. 2014;5:373. https:\/\/doi.org\/10.3389\/fphys.2014.00373","journal-title":"Front Physiol"},{"key":"1956_CR17","doi-asserted-by":"publisher","first-page":"258","DOI":"10.3389\/fnmol.2018.00258","volume":"11","author":"AS Kshatri","year":"2018","unstructured":"Kshatri AS, Gonzalez-Hernandez A, Giraldez T. Physiological roles and therapeutic potential of Ca(2+) activated potassium channels in the nervous system. Front Mol Neurosci. 2018;11:258. https:\/\/doi.org\/10.3389\/fnmol.2018.00258","journal-title":"Front Mol Neurosci"},{"key":"1956_CR18","doi-asserted-by":"publisher","unstructured":"Ancaten-Gonzalez C, Segura I, Alvarado-Sanchez R, Chavez AE, Latorre R. Ca(2+)- and voltage-activated K(+) (BK) channels in the nervous system: one gene, a myriad of physiological functions. Int J Mol Sci. 2023;24. https:\/\/doi.org\/10.3390\/ijms24043407","DOI":"10.3390\/ijms24043407"},{"key":"1956_CR19","doi-asserted-by":"publisher","unstructured":"Orfali R, Albanyan N. Ca(2+)-sensitive potassium channels. Molecules. 2023;28. https:\/\/doi.org\/10.3390\/molecules28020885","DOI":"10.3390\/molecules28020885"},{"key":"1956_CR20","doi-asserted-by":"publisher","DOI":"10.3389\/fphar.2024.1373507","volume":"15","author":"F Echeverria","year":"2024","unstructured":"Echeverria F, Gonzalez-Sanabria N, Alvarado-Sanchez R, Fernandez M, Castillo K, Latorre R. Large conductance voltage-and calcium-activated K(+) (BK) channel in health and disease. Front Pharm. 2024;15:1373507 https:\/\/doi.org\/10.3389\/fphar.2024.1373507","journal-title":"Front Pharm"},{"key":"1956_CR21","doi-asserted-by":"publisher","first-page":"83","DOI":"10.1113\/JP271031","volume":"594","author":"PY Deng","year":"2016","unstructured":"Deng PY, Klyachko VA. Genetic upregulation of BK channel activity normalizes multiple synaptic and circuit defects in a mouse model of fragile X syndrome. J Physiol. 2016;594:83\u201397. https:\/\/doi.org\/10.1113\/JP271031","journal-title":"J Physiol"},{"key":"1956_CR22","doi-asserted-by":"publisher","first-page":"696","DOI":"10.1016\/j.neuron.2012.12.018","volume":"77","author":"PY Deng","year":"2013","unstructured":"Deng PY, Rotman Z, Blundon JA, Cho Y, Cui J, Cavalli V, et al. FMRP regulates neurotransmitter release and synaptic information transmission by modulating action potential duration via BK channels. Neuron. 2013;77:696\u2013711. https:\/\/doi.org\/10.1016\/j.neuron.2012.12.018","journal-title":"Neuron"},{"key":"1956_CR23","doi-asserted-by":"publisher","unstructured":"Kshatri A, Cerrada A, Gimeno R, Bartolome-Martin D, Rojas P, Giraldez T. Differential regulation of BK channels by fragile X mental retardation protein. J Gen Physiol. 2020;152. https:\/\/doi.org\/10.1085\/jgp.201912502","DOI":"10.1085\/jgp.201912502"},{"key":"1956_CR24","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-014-0124-6","volume":"9","author":"B Hebert","year":"2014","unstructured":"Hebert B, Pietropaolo S, Meme S, Laudier B, Laugeray A, Doisne N, et al. Rescue of fragile X syndrome phenotypes in Fmr1 KO mice by a BKCa channel opener molecule. Orphanet J Rare Dis. 2014;9:124.","journal-title":"Orphanet J Rare Dis"},{"key":"1956_CR25","doi-asserted-by":"publisher","first-page":"1701","DOI":"10.1038\/nn.3864","volume":"17","author":"Y Zhang","year":"2014","unstructured":"Zhang Y, Bonnan A, Bony G, Ferezou I, Pietropaolo S, Ginger M, et al. Dendritic channelopathies contribute to neocortical and sensory hyperexcitability in Fmr1(-\/y) mice. Nat Neurosci. 2014;17:1701\u20139. https:\/\/doi.org\/10.1038\/nn.3864","journal-title":"Nat Neurosci"},{"key":"1956_CR26","doi-asserted-by":"publisher","first-page":"949","DOI":"10.1073\/pnas.1423094112","volume":"112","author":"LK Myrick","year":"2015","unstructured":"Myrick LK, Deng PY, Hashimoto H, Oh YM, Cho Y, Poidevin MJ, et al. Independent role for presynaptic FMRP revealed by an FMR1 missense mutation associated with intellectual disability and seizures. Proc Natl Acad Sci USA. 2015;112:949\u201356. https:\/\/doi.org\/10.1073\/pnas.1423094112","journal-title":"Proc Natl Acad Sci USA"},{"key":"1956_CR27","doi-asserted-by":"publisher","first-page":"153","DOI":"10.1007\/s12035-008-8039-7","volume":"38","author":"ZW Wang","year":"2008","unstructured":"Wang ZW. Regulation of synaptic transmission by presynaptic CaMKII and BK channels. Mol Neurobiol. 2008;38:153\u201366. https:\/\/doi.org\/10.1007\/s12035-008-8039-7","journal-title":"Mol Neurobiol"},{"key":"1956_CR28","doi-asserted-by":"publisher","first-page":"1651","DOI":"10.1152\/jn.00058.2011","volume":"105","author":"I van Welie","year":"2011","unstructured":"van Welie I, du Lac S. Bidirectional control of BK channel open probability by CAMKII and PKC in medial vestibular nucleus neurons. J Neurophysiol. 2011;105:1651\u20139. https:\/\/doi.org\/10.1152\/jn.00058.2011","journal-title":"J Neurophysiol"},{"key":"1956_CR29","doi-asserted-by":"publisher","first-page":"247","DOI":"10.1016\/j.cell.2011.06.013","volume":"146","author":"JC Darnell","year":"2011","unstructured":"Darnell JC, Van Driesche SJ, Zhang C, Hung KY, Mele A, Fraser CE, et al. FMRP stalls ribosomal translocation on mRNAs linked to synaptic function and autism. Cell. 2011;146:247\u201361. https:\/\/doi.org\/10.1016\/j.cell.2011.06.013","journal-title":"Cell"},{"key":"1956_CR30","doi-asserted-by":"publisher","first-page":"492","DOI":"10.1038\/npp.2017.149","volume":"43","author":"MI Carreno-Munoz","year":"2018","unstructured":"Carreno-Munoz MI, Martins F, Medrano MC, Aloisi E, Pietropaolo S, Dechaud C, et al. Potential involvement of impaired BK(Ca) channel function in sensory defensiveness and some behavioral disturbances induced by unfamiliar environment in a mouse model of fragile X syndrome. Neuropsychopharmacology. 2018;43:492\u2013502. https:\/\/doi.org\/10.1038\/npp.2017.149","journal-title":"Neuropsychopharmacology"},{"key":"1956_CR31","doi-asserted-by":"publisher","first-page":"2310","DOI":"10.1111\/bcp.14040","volume":"85","author":"N Hohmann","year":"2019","unstructured":"Hohmann N, Blank A, Burhenne J, Suzuki Y, Mikus G, Haefeli WE. Simultaneous phenotyping of CYP2E1 and CYP3A using oral chlorzoxazone and midazolam microdoses. Br J Clin Pharm. 2019;85:2310\u201320. https:\/\/doi.org\/10.1111\/bcp.14040","journal-title":"Br J Clin Pharm"},{"key":"1956_CR32","unstructured":"Martindale W. The extra pharmacopoeia. 30th ed. Amer Pharmaceutical Assn; London, UK; 1993."},{"key":"1956_CR33","doi-asserted-by":"publisher","unstructured":"Ferraguto C, Bouleau Y, Peineau T, Dulon D, Pietropaolo S. Hyperacusis in the adult Fmr1-KO mouse model of fragile X syndrome: the therapeutic relevance of cochlear alterations and BKCa channels. Int J Mol Sci. 2023;24. https:\/\/doi.org\/10.3390\/ijms241411863","DOI":"10.3390\/ijms241411863"},{"key":"1956_CR34","doi-asserted-by":"publisher","DOI":"10.1038\/s41419-020-2357-8","volume":"11","author":"L Deng","year":"2020","unstructured":"Deng L, Li H, Su X, Zhang Y, Xu H, Fan L, et al. Chlorzoxazone, a small molecule drug, augments immunosuppressive capacity of mesenchymal stem cells via modulation of FOXO3 phosphorylation. Cell Death Dis. 2020;11:158 https:\/\/doi.org\/10.1038\/s41419-020-2357-8","journal-title":"Cell Death Dis"},{"key":"1956_CR35","doi-asserted-by":"publisher","DOI":"10.1016\/j.intimp.2020.106790","volume":"88","author":"Y Bai","year":"2020","unstructured":"Bai Y, Ma X. Chlorzoxazone exhibits neuroprotection against Alzheimer\u2019s disease by attenuating neuroinflammation and neurodegeneration in vitro and in vivo. Int Immunopharmacol. 2020;88:106790 https:\/\/doi.org\/10.1016\/j.intimp.2020.106790","journal-title":"Int Immunopharmacol"},{"key":"1956_CR36","doi-asserted-by":"publisher","first-page":"279","DOI":"10.3389\/fnins.2020.00279","volume":"14","author":"PA Egorova","year":"2020","unstructured":"Egorova PA, Gavrilova AV, Bezprozvanny IB. Ataxic symptoms in huntington\u2019s disease transgenic mouse model are alleviated by chlorzoxazone. Front Neurosci. 2020;14:279. https:\/\/doi.org\/10.3389\/fnins.2020.00279","journal-title":"Front Neurosci"},{"key":"1956_CR37","doi-asserted-by":"publisher","first-page":"1340","DOI":"10.1016\/j.chembiol.2012.07.013","volume":"19","author":"AW Kasumu","year":"2012","unstructured":"Kasumu AW, Hougaard C, Rode F, Jacobsen TA, Sabatier JM, Eriksen BL, et al. Selective positive modulator of calcium-activated potassium channels exerts beneficial effects in a mouse model of spinocerebellar ataxia type 2. Chem Biol. 2012;19:1340\u201353. https:\/\/doi.org\/10.1016\/j.chembiol.2012.07.013","journal-title":"Chem Biol"},{"key":"1956_CR38","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-023-39331-y","volume":"13","author":"KS Marinina","year":"2023","unstructured":"Marinina KS, Bezprozvanny IB, Egorova PA. A chlorzoxazone-folic acid combination improves cognitive affective decline in SCA2-58Q mice. Sci Rep. 2023;13:12588 https:\/\/doi.org\/10.1038\/s41598-023-39331-y","journal-title":"Sci Rep"},{"key":"1956_CR39","doi-asserted-by":"publisher","first-page":"742","DOI":"10.1007\/s12311-021-01349-1","volume":"21","author":"PA Egorova","year":"2022","unstructured":"Egorova PA, Bezprozvanny IB. Electrophysiological studies support utility of positive modulators of SK channels for the treatment of spinocerebellar ataxia type 2. Cerebellum. 2022;21:742\u20139. https:\/\/doi.org\/10.1007\/s12311-021-01349-1","journal-title":"Cerebellum"},{"key":"1956_CR40","doi-asserted-by":"publisher","first-page":"86","DOI":"10.1016\/s0006-8993(02)03730-7","volume":"959","author":"YC Liu","year":"2003","unstructured":"Liu YC, Lo YK, Wu SN. Stimulatory effects of chlorzoxazone, a centrally acting muscle relaxant, on large conductance calcium-activated potassium channels in pituitary GH3 cells. Brain Res. 2003;959:86\u201397. https:\/\/doi.org\/10.1016\/s0006-8993(02)03730-7","journal-title":"Brain Res"},{"key":"1956_CR41","doi-asserted-by":"publisher","first-page":"453","DOI":"10.4062\/biomolther.2014.058","volume":"22","author":"YH Jung","year":"2014","unstructured":"Jung YH, Hong SI, Ma SX, Hwang JY, Kim JS, Lee JH, et al. Strain differences in the chronic mild stress animal model of depression and anxiety in mice. Biomol Ther. 2014;22:453\u20139. https:\/\/doi.org\/10.4062\/biomolther.2014.058","journal-title":"Biomol Ther"},{"key":"1956_CR42","doi-asserted-by":"publisher","first-page":"75","DOI":"10.1016\/j.bbr.2013.01.002","volume":"251","author":"D Oddi","year":"2013","unstructured":"Oddi D, Crusio WE, D\u2019Amato FR, Pietropaolo S. Monogenic mouse models of social dysfunction: implications for autism. Behav Brain Res. 2013;251:75\u201384.","journal-title":"Behav Brain Res"},{"key":"1956_CR43","doi-asserted-by":"publisher","first-page":"e17073","DOI":"10.1371\/journal.pone.0017073","volume":"6","author":"S Pietropaolo","year":"2011","unstructured":"Pietropaolo S, Guilleminot A, Martin B, D\u2019Amato FR, Crusio WE. Genetic-background modulation of core and variable autistic-like symptoms in FMR1 knock-out mice. PLoS ONE. 2011;6:e17073.","journal-title":"PLoS ONE"},{"key":"1956_CR44","doi-asserted-by":"publisher","first-page":"119","DOI":"10.1016\/j.psyneuen.2014.07.002","volume":"49","author":"S Pietropaolo","year":"2014","unstructured":"Pietropaolo S, Goubran MG, Joffre C, Aubert A, Lemaire-Mayo V, Crusio WE, et al. Dietary supplementation of omega-3 fatty acids rescues fragile X phenotypes in Fmr1-Ko mice. Psychoneuroendocrinology. 2014;49:119\u201329. https:\/\/doi.org\/10.1016\/j.psyneuen.2014.07.002","journal-title":"Psychoneuroendocrinology"},{"key":"1956_CR45","doi-asserted-by":"crossref","unstructured":"Pietropaolo S, Subashi E. Mouse models of fragile X syndrome. In: Pietropaolo S, Sluyter F, Crusio WE, editors. Behavioral genetics of the mouse. Cambridge: Cambridge University Press; 2014. pp. 146\u201363.","DOI":"10.1017\/CBO9781107360556.014"},{"key":"1956_CR46","doi-asserted-by":"publisher","first-page":"549","DOI":"10.1016\/j.nbd.2005.08.019","volume":"21","author":"EJ Mientjes","year":"2006","unstructured":"Mientjes EJ, Nieuwenhuizen I, Kirkpatrick L, Zu T, Hoogeveen-Westerveld M, Severijnen L, et al. The generation of a conditional Fmr1 knock out mouse model to study Fmrp function in vivo. Neurobiol Dis. 2006;21:549\u201355.","journal-title":"Neurobiol Dis"},{"key":"1956_CR47","doi-asserted-by":"publisher","first-page":"337","DOI":"10.1111\/j.1601-183X.2004.00087.x","volume":"3","author":"QJ Yan","year":"2004","unstructured":"Yan QJ, Asafo-Adjei PK, Arnold HM, Brown RE, Bauchwitz RP. A phenotypic and molecular characterization of the fmr1-tm1Cgr fragile X mouse. Genes Brain Behav. 2004;3:337\u201359.","journal-title":"Genes Brain Behav"},{"key":"1956_CR48","doi-asserted-by":"publisher","first-page":"1584","DOI":"10.1002\/aur.1814","volume":"10","author":"J Gaudissard","year":"2017","unstructured":"Gaudissard J, Ginger M, Premoli M, Memo M, Frick A, Pietropaolo S. Behavioral abnormalities in the Fmr1-KO2 mouse model of fragile X syndrome: the relevance of early life phases. Autism Res. 2017;10:1584\u201396. https:\/\/doi.org\/10.1002\/aur.1814","journal-title":"Autism Res"},{"key":"1956_CR49","doi-asserted-by":"publisher","first-page":"1067","DOI":"10.1002\/aur.1743","volume":"10","author":"M Gauducheau","year":"2017","unstructured":"Gauducheau M, Lemaire-Mayo V, D\u2019Amato FR, Oddi D, Crusio WE, Pietropaolo S. Age-specific autistic-like behaviors in heterozygous Fmr1-KO female mice. Autism Res. 2017;10:1067\u201378. https:\/\/doi.org\/10.1002\/aur.1743","journal-title":"Autism Res"},{"key":"1956_CR50","doi-asserted-by":"publisher","DOI":"10.1016\/j.neubiorev.2022.104722","volume":"139","author":"R Kat","year":"2022","unstructured":"Kat R, Arroyo-Araujo M, de Vries RBM, Koopmans MA, de Boer SF, Kas MJH. Translational validity and methodological underreporting in animal research: a systematic review and meta-analysis of the fragile X syndrome (Fmr1 KO) rodent model. Neurosci Biobehav Rev. 2022;139:104722 https:\/\/doi.org\/10.1016\/j.neubiorev.2022.104722","journal-title":"Neurosci Biobehav Rev"},{"key":"1956_CR51","doi-asserted-by":"publisher","first-page":"e1000412","DOI":"10.1371\/journal.pbio.1000412","volume":"8","author":"C Kilkenny","year":"2010","unstructured":"Kilkenny C, Browne WJ, Cuthill IC, Emerson M, Altman DG. Improving bioscience research reporting: the ARRIVE guidelines for reporting animal research. PLoS Biol. 2010;8:e1000412 https:\/\/doi.org\/10.1371\/journal.pbio.1000412","journal-title":"PLoS Biol"},{"key":"1956_CR52","doi-asserted-by":"publisher","first-page":"1113","DOI":"10.1038\/npp.2014.291","volume":"40","author":"D Oddi","year":"2015","unstructured":"Oddi D, Subashi E, Middei S, Bellocchio L, Lemaire-Mayo V, Guzman M, et al. Early social enrichment rescues adult behavioral and brain abnormalities in a mouse model of fragile X syndrome. Neuropsychopharmacology. 2015;40:1113\u201322. https:\/\/doi.org\/10.1038\/npp.2014.291","journal-title":"Neuropsychopharmacology"},{"key":"1956_CR53","doi-asserted-by":"publisher","DOI":"10.3389\/fncel.2022.917183","volume":"16","author":"V Petroni","year":"2022","unstructured":"Petroni V, Subashi E, Premoli M, Memo M, Lemaire V, Pietropaolo S. Long-term behavioral effects of prenatal stress in the Fmr1-knock-out mouse model for fragile X syndrome. Front Cell Neurosci. 2022;16:917183. https:\/\/doi.org\/10.3389\/fncel.2022.917183","journal-title":"Front Cell Neurosci"},{"key":"1956_CR54","doi-asserted-by":"publisher","unstructured":"Premoli M, Fyke W, Bellocchio L, Lemaire V, Wolley-Roberts M, Bontempi B, et al. Early administration of the phytocannabinoid cannabidivarin prevents the neurobehavioral abnormalities associated with the Fmr1-KO mouse model of fragile X syndrome. Cells. 2023;12. https:\/\/doi.org\/10.3390\/cells12151927","DOI":"10.3390\/cells12151927"},{"key":"1956_CR55","doi-asserted-by":"publisher","first-page":"84","DOI":"10.1136\/jmg.21.2.84","volume":"21","author":"MA De Arce","year":"1984","unstructured":"De Arce MA, Kearns A. The fragile X syndrome: the patients and their chromosomes. J Med Genet. 1984;21:84\u201391. https:\/\/doi.org\/10.1136\/jmg.21.2.84","journal-title":"J Med Genet"},{"key":"1956_CR56","doi-asserted-by":"publisher","first-page":"283","DOI":"10.1002\/ajmg.1320380223","volume":"38","author":"RJ Hagerman","year":"1991","unstructured":"Hagerman RJ, Amiri K, Cronister A. Fragile X checklist. Am J Med Genet. 1991;38:283\u20137. https:\/\/doi.org\/10.1002\/ajmg.1320380223","journal-title":"Am J Med Genet"},{"key":"1956_CR57","doi-asserted-by":"publisher","first-page":"83","DOI":"10.1016\/s0301-0082(96)00021-4","volume":"50","author":"DG Herrera","year":"1996","unstructured":"Herrera DG, Robertson HA. Activation of c-fos in the brain. Prog Neurobiol. 1996;50:83\u2013107. https:\/\/doi.org\/10.1016\/s0301-0082(96)00021-4","journal-title":"Prog Neurobiol"},{"key":"1956_CR58","doi-asserted-by":"publisher","first-page":"416","DOI":"10.1038\/ng859","volume":"30","author":"J Zhang","year":"2002","unstructured":"Zhang J, Zhang D, McQuade JS, Behbehani M, Tsien JZ, Xu M. c-fos regulates neuronal excitability and survival. Nat Genet. 2002;30:416\u201320. https:\/\/doi.org\/10.1038\/ng859","journal-title":"Nat Genet"},{"key":"1956_CR59","doi-asserted-by":"publisher","first-page":"211","DOI":"10.1007\/s11689-011-9084-5","volume":"3","author":"R Hagerman","year":"2011","unstructured":"Hagerman R, Au J, Hagerman P. FMR1 premutation and full mutation molecular mechanisms related to autism. J Neurodev Disord. 2011;3:211\u201324. https:\/\/doi.org\/10.1007\/s11689-011-9084-5","journal-title":"J Neurodev Disord"},{"key":"1956_CR60","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-019-11891-6","volume":"10","author":"SA Booker","year":"2019","unstructured":"Booker SA, Domanski APF, Dando OR, Jackson AD, Isaac JTR, Hardingham GE, et al. Altered dendritic spine function and integration in a mouse model of fragile X syndrome. Nat Commun. 2019;10:4813. https:\/\/doi.org\/10.1038\/s41467-019-11891-6","journal-title":"Nat Commun"},{"key":"1956_CR61","doi-asserted-by":"publisher","first-page":"926","DOI":"10.1016\/j.devcel.2008.04.003","volume":"14","author":"JB Dictenberg","year":"2008","unstructured":"Dictenberg JB, Swanger SA, Antar LN, Singer RH, Bassell GJ. A direct role for FMRP in activity-dependent dendritic mRNA transport links filopodial-spine morphogenesis to fragile X syndrome. Dev Cell. 2008;14:926\u201339. https:\/\/doi.org\/10.1016\/j.devcel.2008.04.003","journal-title":"Dev Cell"},{"key":"1956_CR62","doi-asserted-by":"publisher","first-page":"1038","DOI":"10.1093\/cercor\/10.10.1038","volume":"10","author":"SA Irwin","year":"2000","unstructured":"Irwin SA, Galvez R, Greenough WT. Dendritic spine structural anomalies in fragile-X mental retardation syndrome. Cereb Cortex. 2000;10:1038\u201344. https:\/\/doi.org\/10.1093\/cercor\/10.10.1038","journal-title":"Cereb Cortex"},{"key":"1956_CR63","doi-asserted-by":"publisher","first-page":"492","DOI":"10.1038\/npp.2017.149","volume":"43","author":"MI Carreno-Munoz","year":"2018","unstructured":"Carreno-Munoz MI, Martins F, Medrano MC, Aloisi E, Pietropaolo S, Dechaud C, et al. Potential Involvement of Impaired BKCa Channel Function in Sensory Defensiveness and Some Behavioral Disturbances Induced by Unfamiliar Environment in a Mouse Model of fragile X Syndrome. Neuropsychopharmacology. 2018;43:492\u2013502. https:\/\/doi.org\/10.1038\/npp.2017.149","journal-title":"Neuropsychopharmacology"},{"key":"1956_CR64","first-page":"683","volume":"296","author":"Y Cao","year":"2001","unstructured":"Cao Y, Dreixler JC, Roizen JD, Roberts MT, Houamed KM. Modulation of recombinant small-conductance Ca(2+)-activated K(+) channels by the muscle relaxant chlorzoxazone and structurally related compounds. J Pharm Exp Ther. 2001;296:683\u20139.","journal-title":"J Pharm Exp Ther"},{"key":"1956_CR65","doi-asserted-by":"publisher","first-page":"28","DOI":"10.1523\/JNEUROSCI.1593-18.2018","volume":"39","author":"PY Deng","year":"2019","unstructured":"Deng PY, Carlin D, Oh YM, Myrick LK, Warren ST, Cavalli V, et al. Voltage-independent SK-channel dysfunction causes neuronal hyperexcitability in the hippocampus of Fmr1 knock-out mice. J Neurosci. 2019;39:28\u201343. https:\/\/doi.org\/10.1523\/JNEUROSCI.1593-18.2018","journal-title":"J Neurosci"},{"key":"1956_CR66","doi-asserted-by":"publisher","DOI":"10.1002\/mgg3.956","volume":"7","author":"HMB Biag","year":"2019","unstructured":"Biag HMB, Potter LA, Wilkins V, Afzal S, Rosvall A, Salcedo-Arellano MJ, et al. Metformin treatment in young children with fragile X syndrome. Mol Genet Genom Med. 2019;7:e956. https:\/\/doi.org\/10.1002\/mgg3.956","journal-title":"Mol Genet Genom Med"},{"key":"1956_CR67","doi-asserted-by":"publisher","DOI":"10.1016\/j.neubiorev.2022.104706","volume":"139","author":"V Polito","year":"2022","unstructured":"Polito V, Liknaitzky P. The emerging science of microdosing: A systematic review of research on low dose psychedelics (1955-2021) and recommendations for the field. Neurosci Biobehav Rev. 2022;139:104706. https:\/\/doi.org\/10.1016\/j.neubiorev.2022.104706","journal-title":"Neurosci Biobehav Rev"},{"key":"1956_CR68","doi-asserted-by":"publisher","first-page":"3489","DOI":"10.1113\/JP271841","volume":"594","author":"M Griguoli","year":"2016","unstructured":"Griguoli M, Sgritta M, Cherubini E. Presynaptic BK channels control transmitter release: physiological relevance and potential therapeutic implications. J Physiol. 2016;594:3489\u2013500. https:\/\/doi.org\/10.1113\/JP271841","journal-title":"J Physiol"},{"key":"1956_CR69","doi-asserted-by":"publisher","first-page":"28","DOI":"10.1177\/1073858410395322","volume":"18","author":"C Portera-Cailliau","year":"2012","unstructured":"Portera-Cailliau C. Which comes first in fragile X syndrome, dendritic spine dysgenesis or defects in circuit plasticity? Neuroscientist. 2012;18:28\u201344. https:\/\/doi.org\/10.1177\/1073858410395322","journal-title":"Neuroscientist"},{"key":"1956_CR70","doi-asserted-by":"publisher","unstructured":"Gomez R, Maglio LE, Gonzalez-Hernandez AJ, Rivero-Perez B, Bartolome-Martin D, Giraldez T. NMDA receptor-BK channel coupling regulates synaptic plasticity in the barrel cortex. Proc Natl Acad Sci USA. 2021;118. https:\/\/doi.org\/10.1073\/pnas.2107026118","DOI":"10.1073\/pnas.2107026118"},{"key":"1956_CR71","doi-asserted-by":"publisher","first-page":"41","DOI":"10.1016\/j.mce.2012.12.015","volume":"367","author":"ME Matzkin","year":"2013","unstructured":"Matzkin ME, Lauf S, Spinnler K, Rossi SP, Kohn FM, Kunz L, et al. The Ca2+-activated, large conductance K+-channel (BKCa) is a player in the LH\/hCG signaling cascade in testicular Leydig cells. Mol Cell Endocrinol. 2013;367:41\u20139. https:\/\/doi.org\/10.1016\/j.mce.2012.12.015","journal-title":"Mol Cell Endocrinol"},{"key":"1956_CR72","doi-asserted-by":"publisher","first-page":"23","DOI":"10.1007\/s007260070030","volume":"19","author":"A Saria","year":"2000","unstructured":"Saria A, Fischer HS, Humpel C, Pfattner A, Schatz DS, Schuligoi R. Margatoxin and iberiotoxin, two selective potassium channel inhibitors, induce c-fos like protein and mRNA in rat organotypic dorsal striatal slices. Amino Acids. 2000;19:23\u201331. https:\/\/doi.org\/10.1007\/s007260070030","journal-title":"Amino Acids"},{"key":"1956_CR73","doi-asserted-by":"publisher","first-page":"192","DOI":"10.1016\/j.euroneuro.2016.11.014","volume":"27","author":"JA Waller","year":"2017","unstructured":"Waller JA, Tamm JA, Abdourahman A, Pehrson AL, Li Y, Cajina M, et al. Chronic vortioxetine treatment in rodents modulates gene expression of neurodevelopmental and plasticity markers. Eur Neuropsychopharmacol. 2017;27:192\u2013203. https:\/\/doi.org\/10.1016\/j.euroneuro.2016.11.014","journal-title":"Eur Neuropsychopharmacol"},{"key":"1956_CR74","doi-asserted-by":"publisher","DOI":"10.1186\/s12868-017-0376-x","volume":"18","author":"JA Waller","year":"2017","unstructured":"Waller JA, Nygaard SH, Li Y, du Jardin KG, Tamm JA, Abdourahman A, et al. Neuroplasticity pathways and protein-interaction networks are modulated by vortioxetine in rodents. BMC Neurosci. 2017;18:56. https:\/\/doi.org\/10.1186\/s12868-017-0376-x","journal-title":"BMC Neurosci"},{"key":"1956_CR75","doi-asserted-by":"publisher","first-page":"70","DOI":"10.1016\/j.pbb.2015.05.013","volume":"135","author":"Y Li","year":"2015","unstructured":"Li Y, Abdourahman A, Tamm JA, Pehrson AL, Sanchez C, Gulinello M. Reversal of age-associated cognitive deficits is accompanied by increased plasticity-related gene expression after chronic antidepressant administration in middle-aged mice. Pharm Biochem Behav. 2015;135:70\u201382. https:\/\/doi.org\/10.1016\/j.pbb.2015.05.013","journal-title":"Pharm Biochem Behav"},{"key":"1956_CR76","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1016\/j.molbrainres.2004.08.014","volume":"131","author":"JC Lauterborn","year":"2004","unstructured":"Lauterborn JC. Stress induced changes in cortical and hypothalamic c-fos expression are altered in fragile X mutant mice. Brain Res Mol Brain Res. 2004;131:101\u20139. https:\/\/doi.org\/10.1016\/j.molbrainres.2004.08.014","journal-title":"Brain Res Mol Brain Res"},{"key":"1956_CR77","doi-asserted-by":"publisher","first-page":"2587","DOI":"10.1073\/pnas.1013855108","volume":"108","author":"DD Krueger","year":"2011","unstructured":"Krueger DD, Osterweil EK, Chen SP, Tye LD, Bear MF. Cognitive dysfunction and prefrontal synaptic abnormalities in a mouse model of fragile X syndrome. Proc Natl Acad Sci USA. 2011;108:2587\u201392. https:\/\/doi.org\/10.1073\/pnas.1013855108","journal-title":"Proc Natl Acad Sci USA"},{"key":"1956_CR78","doi-asserted-by":"publisher","DOI":"10.1186\/s13229-017-0148-6","volume":"8","author":"TD Rogers","year":"2017","unstructured":"Rogers TD, Anacker AMJ, Kerr TM, Forsberg CG, Wang J, Zhang B, et al. Effects of a social stimulus on gene expression in a mouse model of fragile X syndrome. Mol autism. 2017;8:30. https:\/\/doi.org\/10.1186\/s13229-017-0148-6","journal-title":"Mol autism"},{"key":"1956_CR79","doi-asserted-by":"publisher","first-page":"341","DOI":"10.1017\/S0016672308009415","volume":"90","author":"V Errijgers","year":"2008","unstructured":"Errijgers V, Fransen E, D\u2019Hooge R, De Deyn PP, Kooy RF. Effect of genetic background on acoustic startle response in fragile X knockout mice. Genet Res. 2008;90:341\u20135.","journal-title":"Genet Res"},{"key":"1956_CR80","doi-asserted-by":"publisher","first-page":"40","DOI":"10.1002\/aur.168","volume":"4","author":"CM Spencer","year":"2011","unstructured":"Spencer CM, Alekseyenko O, Hamilton SM, Thomas AM, Serysheva E, Yuva-Paylor LA, et al. Modifying behavioral phenotypes in Fmr1KO mice: genetic background differences reveal autistic-like responses. Autism Res. 2011;4:40\u201356.","journal-title":"Autism Res"},{"key":"1956_CR81","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1515\/revneuro.1994.5.2.171","volume":"5","author":"M Asanuma","year":"1994","unstructured":"Asanuma M, Ogawa N. Pitfalls in assessment of c-fos mRNA expression in the brain: effects of animal handling. Rev Neurosci. 1994;5:171\u20138. https:\/\/doi.org\/10.1515\/revneuro.1994.5.2.171","journal-title":"Rev Neurosci"},{"key":"1956_CR82","doi-asserted-by":"publisher","first-page":"353","DOI":"10.1111\/j.1527-3458.2002.tb00233.x","volume":"8","author":"BS Jensen","year":"2002","unstructured":"Jensen BS. BMS-204352: a potassium channel opener developed for the treatment of stroke. CNS Drug Rev. 2002;8:353\u201360.","journal-title":"CNS Drug Rev"},{"key":"1956_CR83","doi-asserted-by":"publisher","first-page":"1173","DOI":"10.1085\/jgp.201912457","volume":"151","author":"CS Bailey","year":"2019","unstructured":"Bailey CS, Moldenhauer HJ, Park SM, Keros S, Meredith AL. KCNMA1-linked channelopathy. J Gen Physiol. 2019;151:1173\u201389. https:\/\/doi.org\/10.1085\/jgp.201912457","journal-title":"J Gen Physiol"},{"key":"1956_CR84","doi-asserted-by":"publisher","first-page":"1008","DOI":"10.1016\/j.cell.2013.10.031","volume":"155","author":"NN Parikshak","year":"2013","unstructured":"Parikshak NN, Luo R, Zhang A, Won H, Lowe JK, Chandran V, et al. Integrative functional genomic analyses implicate specific molecular pathways and circuits in autism. Cell. 2013;155:1008\u201321. https:\/\/doi.org\/10.1016\/j.cell.2013.10.031","journal-title":"Cell"},{"key":"1956_CR85","doi-asserted-by":"publisher","first-page":"165","DOI":"10.1023\/A:1010747131386","volume":"31","author":"DB Bailey Jr","year":"2001","unstructured":"Bailey DB Jr, Hatton DD, Skinner M, Mesibov G. Autistic behavior, FMR1 protein, and developmental trajectories in young males with fragile X syndrome. J Autism Dev Disord. 2001;31:165\u201374.","journal-title":"J Autism Dev Disord"},{"key":"1956_CR86","doi-asserted-by":"publisher","first-page":"499","DOI":"10.1023\/A:1026048027397","volume":"28","author":"DB Bailey Jr","year":"1998","unstructured":"Bailey DB Jr, Mesibov GB, Hatton DD, Clark RD, Roberts JE, Mayhew L. Autistic behavior in young boys with fragile X syndrome. J Autism Dev Disord. 1998;28:499\u2013508.","journal-title":"J Autism Dev Disord"},{"key":"1956_CR87","doi-asserted-by":"crossref","unstructured":"Brock M, Hatton D. Distinguishing features of autism in boys with fragile X syndrome. J Intellect Disabil Res. 2010;54:894\u2013905.","DOI":"10.1111\/j.1365-2788.2010.01315.x"},{"key":"1956_CR88","doi-asserted-by":"publisher","first-page":"409","DOI":"10.1097\/00004703-200112000-00008","volume":"22","author":"SJ Rogers","year":"2001","unstructured":"Rogers SJ, Wehner DE, Hagerman R. The behavioral phenotype in fragile X: symptoms of autism in very young children with fragile X syndrome, idiopathic autism, and other developmental disorders. J Dev Behav Pediatr. 2001;22:409\u201317.","journal-title":"J Dev Behav Pediatr"},{"key":"1956_CR89","doi-asserted-by":"publisher","first-page":"336","DOI":"10.2119\/molmed.2011.00363","volume":"18","author":"C Gross","year":"2012","unstructured":"Gross C, Bassell GJ. Excess protein synthesis in FXS patient lymphoblastoid cells can be rescued with a p110beta-selective inhibitor. Mol Med. 2012;18:336\u201345. https:\/\/doi.org\/10.2119\/molmed.2011.00363","journal-title":"Mol Med"},{"key":"1956_CR90","doi-asserted-by":"publisher","first-page":"10624","DOI":"10.1523\/JNEUROSCI.0402-10.2010","volume":"30","author":"C Gross","year":"2010","unstructured":"Gross C, Nakamoto M, Yao X, Chan CB, Yim SY, Ye K, et al. Excess phosphoinositide 3-kinase subunit synthesis and activity as a novel therapeutic target in fragile X syndrome. J Neurosci. 2010;30:10624\u201338. https:\/\/doi.org\/10.1523\/JNEUROSCI.0402-10.2010","journal-title":"J Neurosci"},{"key":"1956_CR91","doi-asserted-by":"publisher","first-page":"332","DOI":"10.1111\/j.1601-183X.2012.00768.x","volume":"11","author":"CA Hoeffer","year":"2012","unstructured":"Hoeffer CA, Sanchez E, Hagerman RJ, Mu Y, Nguyen DV, Wong H, et al. Altered mTOR signaling and enhanced CYFIP2 expression levels in subjects with fragile X syndrome. Genes Brain Behav. 2012;11:332\u201341. https:\/\/doi.org\/10.1111\/j.1601-183X.2012.00768.x","journal-title":"Genes Brain Behav"},{"key":"1956_CR92","doi-asserted-by":"publisher","first-page":"694","DOI":"10.1523\/JNEUROSCI.3696-09.2010","volume":"30","author":"A Sharma","year":"2010","unstructured":"Sharma A, Hoeffer CA, Takayasu Y, Miyawaki T, McBride SM, Klann E, et al. Dysregulation of mTOR signaling in fragile X syndrome. J Neurosci. 2010;30:694\u2013702. https:\/\/doi.org\/10.1523\/JNEUROSCI.3696-09.2010","journal-title":"J Neurosci"},{"key":"1956_CR93","doi-asserted-by":"publisher","DOI":"10.3389\/fcell.2022.934662","volume":"10","author":"C Song","year":"2022","unstructured":"Song C, Broadie K. Dysregulation of BMP, Wnt, and insulin signaling in fragile X syndrome. Front Cell Dev Biol. 2022;10:934662. https:\/\/doi.org\/10.3389\/fcell.2022.934662","journal-title":"Front Cell Dev Biol"},{"key":"1956_CR94","doi-asserted-by":"publisher","first-page":"672","DOI":"10.1111\/j.1471-4159.2012.07722.x","volume":"121","author":"X Wang","year":"2012","unstructured":"Wang X, Snape M, Klann E, Stone JG, Singh A, Petersen RB, et al. Activation of the extracellular signal-regulated kinase pathway contributes to the behavioral deficit of fragile x-syndrome. J Neurochem. 2012;121:672\u20139. https:\/\/doi.org\/10.1111\/j.1471-4159.2012.07722.x","journal-title":"J Neurochem"},{"key":"1956_CR95","doi-asserted-by":"publisher","DOI":"10.1186\/s13041-020-00706-1","volume":"13","author":"CR Casingal","year":"2020","unstructured":"Casingal CR, Kikkawa T, Inada H, Sasaki Y, Osumi N. Identification of FMRP target mRNAs in the developmental brain: FMRP might coordinate Ras\/MAPK, Wnt\/beta-catenin, and mTOR signaling during corticogenesis. Mol Brain. 2020;13:167. https:\/\/doi.org\/10.1186\/s13041-020-00706-1","journal-title":"Mol Brain"},{"key":"1956_CR96","doi-asserted-by":"publisher","DOI":"10.1038\/s41419-022-05019-w","volume":"13","author":"G Pedini","year":"2022","unstructured":"Pedini G, Buccarelli M, Bianchi F, Pacini L, Cencelli G, D\u2019Alessandris QG, et al. FMRP modulates the Wnt signalling pathway in glioblastoma. Cell Death Dis. 2022;13:719. https:\/\/doi.org\/10.1038\/s41419-022-05019-w","journal-title":"Cell Death Dis"},{"key":"1956_CR97","doi-asserted-by":"publisher","first-page":"53","DOI":"10.1016\/j.nbd.2019.02.004","volume":"127","author":"JC Sears","year":"2019","unstructured":"Sears JC, Choi WJ, Broadie K. Fragile X Mental Retardation Protein positively regulates PKA anchor Rugose and PKA activity to control actin assembly in learning\/memory circuitry. Neurobiol Dis. 2019;127:53\u201364. https:\/\/doi.org\/10.1016\/j.nbd.2019.02.004","journal-title":"Neurobiol Dis"},{"key":"1956_CR98","doi-asserted-by":"publisher","first-page":"246","DOI":"10.1080\/01677063.2018.1500571","volume":"32","author":"WL Bollinger","year":"2018","unstructured":"Bollinger WL, Sial N, Dawson-Scully K. BK channels and a cGMP-dependent protein kinase (PKG) function through independent mechanisms to regulate the tolerance of synaptic transmission to acute oxidative stress at the Drosophila larval neuromuscular junction. J Neurogenet. 2018;32:246\u201355. https:\/\/doi.org\/10.1080\/01677063.2018.1500571","journal-title":"J Neurogenet"},{"key":"1956_CR99","doi-asserted-by":"publisher","first-page":"25","DOI":"10.1016\/j.jphs.2019.08.006","volume":"141","author":"MV Mitrokhin","year":"2019","unstructured":"Mitrokhin MV, Kalsin V, Kamkina O, Babkina I, Zotov A, Troitskiy VA, et al. Participation of PKG and PKA-related pathways in the IFN-gamma induced modulation of the BK(Ca) channel activity in human cardiac fibroblasts. J Pharm Sci. 2019;141:25\u201331. https:\/\/doi.org\/10.1016\/j.jphs.2019.08.006","journal-title":"J Pharm Sci"},{"key":"1956_CR100","doi-asserted-by":"publisher","first-page":"F283","DOI":"10.1152\/ajprenal.2000.279.2.F283","volume":"279","author":"SC Sansom","year":"2000","unstructured":"Sansom SC, Ma R, Carmines PK, Hall DA. Regulation of Ca(2+)-activated K(+) channels by multifunctional Ca(2+)\/calmodulin-dependent protein kinase. Am J Physiol Ren Physiol. 2000;279:F283\u20138. https:\/\/doi.org\/10.1152\/ajprenal.2000.279.2.F283","journal-title":"Am J Physiol Ren Physiol"},{"key":"1956_CR101","doi-asserted-by":"publisher","first-page":"505","DOI":"10.1016\/s0165-6147(00)01775-2","volume":"22","author":"R Schubert","year":"2001","unstructured":"Schubert R, Nelson MT. Protein kinases: tuners of the BKCa channel in smooth muscle. Trends Pharm Sci. 2001;22:505\u201312. https:\/\/doi.org\/10.1016\/s0165-6147(00)01775-2","journal-title":"Trends Pharm Sci"},{"key":"1956_CR102","doi-asserted-by":"publisher","DOI":"10.1186\/s12885-018-5027-9","volume":"18","author":"F Wang","year":"2018","unstructured":"Wang F, Chen Q, Huang G, Guo X, Li N, Li Y, et al. BKCa participates in E2 inducing endometrial adenocarcinoma by activating MEK\/ERK pathway. BMC Cancer. 2018;18:1128. https:\/\/doi.org\/10.1186\/s12885-018-5027-9","journal-title":"BMC Cancer"},{"key":"1956_CR103","doi-asserted-by":"publisher","first-page":"167","DOI":"10.3389\/fphys.2019.00167","volume":"10","author":"Y Wang","year":"2019","unstructured":"Wang Y, Tao J, Wang M, Yang L, Ning F, Xin H, et al. Mechanism of regulation of big-conductance Ca(2+)-activated K(+) channels by mTOR complex 2 in podocytes. Front Physiol. 2019;10:167 https:\/\/doi.org\/10.3389\/fphys.2019.00167","journal-title":"Front Physiol"},{"key":"1956_CR104","doi-asserted-by":"publisher","first-page":"32161","DOI":"10.1074\/jbc.M112.386359","volume":"287","author":"X Zhou","year":"2012","unstructured":"Zhou X, Wulfsen I, Korth M, McClafferty H, Lukowski R, Shipston MJ, et al. Palmitoylation and membrane association of the stress axis regulated insert (STREX) controls BK channel regulation by protein kinase C. J Biol Chem. 2012;287:32161\u201371. https:\/\/doi.org\/10.1074\/jbc.M112.386359","journal-title":"J Biol Chem"},{"key":"1956_CR105","doi-asserted-by":"publisher","first-page":"10977","DOI":"10.1523\/JNEUROSCI.1077-10.2010","volume":"30","author":"LY Chen","year":"2010","unstructured":"Chen LY, Rex CS, Babayan AH, Kramar EA, Lynch G, Gall CM, et al. Physiological activation of synaptic Rac>PAK (p-21 activated kinase) signaling is defective in a mouse model of fragile X syndrome. J Neurosci. 2010;30:10977\u201384. https:\/\/doi.org\/10.1523\/JNEUROSCI.1077-10.2010","journal-title":"J Neurosci"},{"key":"1956_CR106","doi-asserted-by":"publisher","first-page":"2297","DOI":"10.1016\/j.celrep.2015.11.013","volume":"13","author":"W Guo","year":"2015","unstructured":"Guo W, Ceolin L, Collins KA, Perroy J, Huber KM. Elevated CaMKIIalpha and hyperphosphorylation of homer mediate circuit dysfunction in a fragile X syndrome mouse model. Cell Rep. 2015;13:2297\u2013311. https:\/\/doi.org\/10.1016\/j.celrep.2015.11.013","journal-title":"Cell Rep"},{"key":"1956_CR107","doi-asserted-by":"publisher","unstructured":"Jiang A, Wang L, Lu JYD, Freeman A, Campbell C, Su P, et al. Sex differences in dopamine receptor signaling in Fmr1 knockout mice: a pilot study. Brain Sci. 2021;11. https:\/\/doi.org\/10.3390\/brainsci11111398","DOI":"10.3390\/brainsci11111398"},{"key":"1956_CR108","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-017-18598-y","volume":"8","author":"E Mok-Lin","year":"2018","unstructured":"Mok-Lin E, Ascano M Jr, Serganov A, Rosenwaks Z, Tuschl T, Williams Z. Premature recruitment of oocyte pool and increased mTOR activity in Fmr1 knockout mice and reversal of phenotype with rapamycin. Sci Rep. 2018;8:588 https:\/\/doi.org\/10.1038\/s41598-017-18598-y","journal-title":"Sci Rep"},{"key":"1956_CR109","doi-asserted-by":"publisher","first-page":"E6290","DOI":"10.1073\/pnas.1610812113","volume":"113","author":"K Sawicka","year":"2016","unstructured":"Sawicka K, Pyronneau A, Chao M, Bennett MV, Zukin RS. Elevated ERK\/p90 ribosomal S6 kinase activity underlies audiogenic seizure susceptibility in fragile X mice. Proc Natl Acad Sci USA. 2016;113:E6290\u2013E7. https:\/\/doi.org\/10.1073\/pnas.1610812113","journal-title":"Proc Natl Acad Sci USA"},{"key":"1956_CR110","doi-asserted-by":"publisher","first-page":"303","DOI":"10.1016\/0143-4160(88)90011-5","volume":"9","author":"JI Morgan","year":"1988","unstructured":"Morgan JI, Curran T. Calcium as a modulator of the immediate-early gene cascade in neurons. Cell Calcium. 1988;9:303\u201311. https:\/\/doi.org\/10.1016\/0143-4160(88)90011-5","journal-title":"Cell Calcium"},{"key":"1956_CR111","doi-asserted-by":"publisher","first-page":"113","DOI":"10.1146\/annurev-physiol-022516-034038","volume":"81","author":"V Gonzalez-Perez","year":"2019","unstructured":"Gonzalez-Perez V, Lingle CJ. Regulation of BK channels by beta and gamma subunits. Annu Rev Physiol. 2019;81:113\u201337. https:\/\/doi.org\/10.1146\/annurev-physiol-022516-034038","journal-title":"Annu Rev Physiol"},{"key":"1956_CR112","doi-asserted-by":"publisher","DOI":"10.3389\/fncel.2024.1353895","volume":"18","author":"LA Blomer","year":"2024","unstructured":"Blomer LA, Giacalone E, Abbas F, Filipis L, Tegolo D, Migliore M, et al. Kinetics and functional consequences of BK channels activation by N-type Ca(2+) channels in the dendrite of mouse neocortical layer-5 pyramidal neurons. Front Cell Neurosci. 2024;18:1353895. https:\/\/doi.org\/10.3389\/fncel.2024.1353895","journal-title":"Front Cell Neurosci"},{"key":"1956_CR113","doi-asserted-by":"publisher","first-page":"1957","DOI":"10.1113\/JP283801","volume":"601","author":"L Filipis","year":"2023","unstructured":"Filipis L, Blomer LA, Montnach J, Loussouarn G, De Waard M, Canepari M. Nav1.2 and BK channel interaction shapes the action potential in the axon initial segment. J Physiol. 2023;601:1957\u201379. https:\/\/doi.org\/10.1113\/JP283801","journal-title":"J Physiol"}],"container-title":["Neuropsychopharmacology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.nature.com\/articles\/s41386-024-01956-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.nature.com\/articles\/s41386-024-01956-6","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.nature.com\/articles\/s41386-024-01956-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,10,17]],"date-time":"2024-10-17T13:03:25Z","timestamp":1729170205000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.nature.com\/articles\/s41386-024-01956-6"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,9,2]]},"references-count":113,"journal-issue":{"issue":"13","published-print":{"date-parts":[[2024,12]]}},"alternative-id":["1956"],"URL":"https:\/\/doi.org\/10.1038\/s41386-024-01956-6","relation":{},"ISSN":["0893-133X","1740-634X"],"issn-type":[{"value":"0893-133X","type":"print"},{"value":"1740-634X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,9,2]]},"assertion":[{"value":"14 March 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"30 June 2024","order":2,"name":"revised","label":"Revised","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"23 July 2024","order":3,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"2 September 2024","order":4,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"VL, EL, and SP are inventors on the patent: \u201cMethods of treatment and\/or prevention of disorders and symptoms related to BKCa and\/or SK channelopathies\u201d (N\u00b0. EP20775228.8 and N\u00b0. US17760612) owned by CNRS. The remaining authors have nothing to disclose.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}]}}