{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T23:11:57Z","timestamp":1791241917318,"version":"4.1.0"},"publisher-location":"Cham","reference-count":145,"publisher":"Springer International Publishing","isbn-type":[{"value":"9783031210532","type":"print"},{"value":"9783031210549","type":"electronic"}],"license":[{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.springernature.com\/gp\/researchers\/text-and-data-mining"},{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"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":[],"published-print":{"date-parts":[[2023]]},"DOI":"10.1007\/978-3-031-21054-9_10","type":"book-chapter","created":{"date-parts":[[2023,3,16]],"date-time":"2023-03-16T13:02:55Z","timestamp":1678971775000},"page":"225-253","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["Targeted Treatments for Fragile X Syndrome"],"prefix":"10.1007","author":[{"given":"Devon","family":"Johnson","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Courtney","family":"Clark","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Randi","family":"Hagerman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2023,3,17]]},"reference":[{"key":"10_CR1","doi-asserted-by":"publisher","first-page":"31","DOI":"10.1002\/mrdd.20006","volume":"10","author":"DZ Loesch","year":"2004","unstructured":"Loesch DZ, Huggins RM, Hagerman RJ. Phenotypic variation and FMRP levels in fragile X. Ment Retard Dev Disabil Res Rev. 2004;10:31\u201341.","journal-title":"Ment Retard Dev Disabil Res Rev"},{"key":"10_CR2","doi-asserted-by":"publisher","first-page":"2816","DOI":"10.1007\/s10803-015-2443-4","volume":"45","author":"AJ Thurman","year":"2015","unstructured":"Thurman AJ, McDuffie A, Kover ST, Hagerman RJ, Abbeduto L. Autism symptomatology in boys with fragile X syndrome: a cross sectional developmental trajectories comparison with nonsyndromic autism spectrum disorder. J Autism Dev Disord. 2015;45:2816\u201332.","journal-title":"J Autism Dev Disord"},{"key":"10_CR3","doi-asserted-by":"publisher","first-page":"227","DOI":"10.3389\/fpsyg.2012.00227","volume":"3","author":"K Cornish","year":"2012","unstructured":"Cornish K, Steele A, Monteiro CRC, Karmiloff-Smith A, Scerif G. Attention deficits predict phenotypic outcomes in syndrome-specific and domain-specific ways. Front Psychol. 2012;3:227.","journal-title":"Front Psychol"},{"key":"10_CR4","doi-asserted-by":"publisher","first-page":"57","DOI":"10.1007\/s11689-010-9067-y","volume":"3","author":"L Cordeiro","year":"2011","unstructured":"Cordeiro L, Ballinger E, Hagerman R, Hessl D. Clinical assessment of DSM-IV anxiety disorders in fragile X syndrome: prevalence and characterization. J Neurodev Disord. 2011;3:57\u201367.","journal-title":"J Neurodev Disord"},{"key":"10_CR5","doi-asserted-by":"publisher","first-page":"961","DOI":"10.1007\/s10803-007-0470-5","volume":"38","author":"KL Hess","year":"2008","unstructured":"Hess KL, Morrier MJ, Heflin LJ, Ivey ML. Autism treatment survey: services received by children with autism spectrum disorders in public school classrooms. J Autism Dev Disord. 2008;38:961\u201371.","journal-title":"J Autism Dev Disord"},{"key":"10_CR6","doi-asserted-by":"publisher","first-page":"17065","DOI":"10.1038\/nrdp.2017.65","volume":"3","author":"RJ Hagerman","year":"2017","unstructured":"Hagerman RJ, et al. Fragile X syndrome. Nat Rev Dis Prim. 2017;3:17065.","journal-title":"Nat Rev Dis Prim"},{"key":"10_CR7","doi-asserted-by":"publisher","first-page":"100","DOI":"10.1186\/gm401","volume":"4","author":"F Tassone","year":"2012","unstructured":"Tassone F, et al. FMR1 CGG allele size and prevalence ascertained through newborn screening in the United States. Genome Med. 2012;4:100.","journal-title":"Genome Med"},{"key":"10_CR8","doi-asserted-by":"publisher","first-page":"136","DOI":"10.1159\/000320025","volume":"1","author":"A Schneider","year":"2010","unstructured":"Schneider A. MRNET Conference. Mol Syndromol. 2010;1:136\u201357.","journal-title":"Mol Syndromol"},{"key":"10_CR9","doi-asserted-by":"publisher","first-page":"3228","DOI":"10.1093\/hmg\/ddu032","volume":"23","author":"AL Ludwig","year":"2014","unstructured":"Ludwig AL, et al. CNS expression of murine fragile X protein (FMRP) as a function of CGG-repeat size. Hum Mol Genet. 2014;23:3228\u201338.","journal-title":"Hum Mol Genet"},{"key":"10_CR10","first-page":"23","volume":"78","author":"The Dutch-Belgian Fragile X Consorthium et al","year":"1994","unstructured":"The Dutch-Belgian Fragile X Consorthium et al. Fmr1 knockout mice: a model to study fragile X mental retardation. Cell. 1994;78:23\u201333.","journal-title":"Cell"},{"key":"10_CR11","doi-asserted-by":"publisher","first-page":"111","DOI":"10.1159\/000072845","volume":"100","author":"CE Bakker","year":"2003","unstructured":"Bakker CE, Oostra BA. Understanding fragile X syndrome: insights from animal models. Cytogenet Genome Res. 2003;100:111\u201323.","journal-title":"Cytogenet Genome Res"},{"key":"10_CR12","doi-asserted-by":"publisher","first-page":"5196","DOI":"10.1523\/JNEUROSCI.17-13-05196.1997","volume":"17","author":"Y-M Lu","year":"1997","unstructured":"Lu Y-M, et al. Mice lacking metabotropic glutamate receptor 5 show impaired learning and reduced CA1 Long-term potentiation (LTP) but normal CA3 LTP. J Neurosci. 1997;17:5196\u2013205.","journal-title":"J Neurosci"},{"key":"10_CR13","doi-asserted-by":"publisher","first-page":"8536","DOI":"10.1128\/MCB.20.22.8536-8547.2000","volume":"20","author":"L Wan","year":"2000","unstructured":"Wan L, Dockendorff TC, Jongens TA, Dreyfuss G. Characterization of dFMR1, a Drosophila melanogaster homolog of the fragile X mental retardation protein. Mol Cell Biol. 2000;20:8536\u201347.","journal-title":"Mol Cell Biol"},{"key":"10_CR14","doi-asserted-by":"publisher","first-page":"1","DOI":"10.29245\/2572-9411\/2020\/1.1190","volume":"5","author":"P Kulkarni","year":"2020","unstructured":"Kulkarni P, Sevilimedu A. The known unknowns: missing pieces in in vivo models of fragile X syndrome. J Rare Dis Res Treat. 2020;5:1\u20139.","journal-title":"J Rare Dis Res Treat"},{"key":"10_CR15","doi-asserted-by":"publisher","first-page":"448","DOI":"10.1159\/000078218","volume":"105","author":"V Errijgers","year":"2004","unstructured":"Errijgers V, Kooy RF. Genetic modifiers in mice: the example of the fragile X mouse model. Cytogenet Genome Res. 2004;105:448\u201354.","journal-title":"Cytogenet Genome Res"},{"key":"10_CR16","doi-asserted-by":"publisher","first-page":"973","DOI":"10.1016\/S0896-6273(02)00724-9","volume":"34","author":"TC Dockendorff","year":"2002","unstructured":"Dockendorff TC, et al. Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest. Neuron. 2002;34:973\u201384.","journal-title":"Neuron"},{"key":"10_CR17","doi-asserted-by":"publisher","first-page":"22","DOI":"10.1016\/j.brainres.2014.11.023","volume":"1596","author":"S Gholizadeh","year":"2015","unstructured":"Gholizadeh S, Halder SK, Hampson DR. Expression of fragile X mental retardation protein in neurons and glia of the developing and adult mouse brain. Brain Res. 2015;1596:22\u201330.","journal-title":"Brain Res"},{"key":"10_CR18","doi-asserted-by":"publisher","first-page":"280","DOI":"10.1038\/nrd.2017.221","volume":"17","author":"EM Berry-Kravis","year":"2018","unstructured":"Berry-Kravis EM, et al. Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome. Nat Rev Drug Discov. 2018;17:280\u201399.","journal-title":"Nat Rev Drug Discov"},{"key":"10_CR19","doi-asserted-by":"publisher","first-page":"275","DOI":"10.1038\/s41583-021-00445-9","volume":"22","author":"P-Y Deng","year":"2021","unstructured":"Deng P-Y, Klyachko VA. Channelopathies in fragile X syndrome. Nat Rev Neurosci. 2021;22:275\u201389.","journal-title":"Nat Rev Neurosci"},{"key":"10_CR20","doi-asserted-by":"publisher","first-page":"7746","DOI":"10.1073\/pnas.122205699","volume":"99","author":"KM Huber","year":"2002","unstructured":"Huber KM, Gallagher SM, Warren ST, Bear MF. Altered synaptic plasticity in a mouse model of fragile X mental retardation. Proc Natl Acad Sci. 2002;99:7746\u201350.","journal-title":"Proc Natl Acad Sci"},{"key":"10_CR21","doi-asserted-by":"publisher","first-page":"955","DOI":"10.1016\/j.neuron.2007.12.001","volume":"56","author":"G D\u00f6len","year":"2007","unstructured":"D\u00f6len G, et al. Correction of fragile X syndrome in mice. Neuron. 2007;56:955\u201362.","journal-title":"Neuron"},{"key":"10_CR22","doi-asserted-by":"publisher","first-page":"152","DOI":"10.1016\/j.brainresbull.2011.08.001","volume":"86","author":"JA Burket","year":"2011","unstructured":"Burket JA, Herndon AL, Winebarger EE, Jacome LF, Deutsch SI. Complex effects of mGluR5 antagonism on sociability and stereotypic behaviors in mice: possible implications for the pharmacotherapy of autism spectrum disorders. Brain Res Bull. 2011;86:152\u20138.","journal-title":"Brain Res Bull"},{"issue":"47\u201358","key":"10_CR23","doi-asserted-by":"publisher","first-page":"47","DOI":"10.1007\/s00213-011-2375-4","volume":"219","author":"AM Thomas","year":"2012","unstructured":"Thomas AM, Bui N, Perkins JR, Yuva-Paylor LA, Paylor R. Group I metabotropic glutamate receptor antagonists alter select behaviors in a mouse model for fragile X syndrome. Psychopharmacology. 2012;219(47\u201358):47.","journal-title":"Psychopharmacology"},{"key":"10_CR24","doi-asserted-by":"publisher","first-page":"753","DOI":"10.1016\/j.neuron.2005.01.038","volume":"45","author":"SMJ McBride","year":"2005","unstructured":"McBride SMJ, et al. Pharmacological rescue of synaptic plasticity, courtship behavior, and mushroom body defects in a drosophila model of fragile X syndrome. Neuron. 2005;45:753\u201364.","journal-title":"Neuron"},{"key":"10_CR25","doi-asserted-by":"publisher","first-page":"109805","DOI":"10.1016\/j.celrep.2021.109805","volume":"37","author":"G Fernandes","year":"2021","unstructured":"Fernandes G, et al. Correction of amygdalar dysfunction in a rat model of fragile X syndrome. Cell Rep. 2021;37:109805.","journal-title":"Cell Rep"},{"key":"10_CR26","doi-asserted-by":"publisher","first-page":"12867","DOI":"10.1038\/ncomms12867","volume":"7","author":"SA Wolfe","year":"2016","unstructured":"Wolfe SA, et al. FMRP regulates an ethanol-dependent shift in GABABR function and expression with rapid antidepressant properties. Nat Commun. 2016;7:12867.","journal-title":"Nat Commun"},{"key":"10_CR27","doi-asserted-by":"publisher","first-page":"2985","DOI":"10.4161\/15384101.2014.989114","volume":"14","author":"S Braat","year":"2015","unstructured":"Braat S, et al. The GABA A receptor is an FMRP target with therapeutic potential in fragile X syndrome. Cell Cycle. 2015;14:2985\u201395.","journal-title":"Cell Cycle"},{"key":"10_CR28","doi-asserted-by":"publisher","first-page":"48","DOI":"10.1016\/j.neuropharm.2014.06.028","volume":"88","author":"S Braat","year":"2015","unstructured":"Braat S, Kooy RF. Insights into GABAAergic system deficits in fragile X syndrome lead to clinical trials. Neuropharmacology. 2015;88:48\u201354.","journal-title":"Neuropharmacology"},{"key":"10_CR29","doi-asserted-by":"publisher","first-page":"612685","DOI":"10.3389\/fnmol.2020.612685","volume":"13","author":"ST Reyes","year":"2020","unstructured":"Reyes ST, et al. GABA measurement in a neonatal fragile X syndrome mouse model using 1H-magnetic resonance spectroscopy and mass spectrometry. Front Mol Neurosci. 2020;13:612685.","journal-title":"Front Mol Neurosci"},{"key":"10_CR30","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0131486","volume":"10","author":"C D\u2019Hulst","year":"2015","unstructured":"D\u2019Hulst C, et al. Positron emission tomography (PET) quantification of GABAA receptors in the brain of fragile X patients. PLoS One. 2015;10:e0131486.","journal-title":"PLoS One"},{"key":"10_CR31","doi-asserted-by":"publisher","first-page":"10468","DOI":"10.1038\/srep10468","volume":"5","author":"W Zhang","year":"2015","unstructured":"Zhang W, et al. GABAB receptor upregulates fragile X mental retardation protein expression in neurons. Sci Rep. 2015;5:10468.","journal-title":"Sci Rep"},{"key":"10_CR32","doi-asserted-by":"publisher","first-page":"88","DOI":"10.3389\/fnmol.2020.00088","volume":"13","author":"I Kramvis","year":"2020","unstructured":"Kramvis I, et al. Dysregulated prefrontal cortex inhibition in prepubescent and adolescent fragile X mouse model. Front Mol Neurosci. 2020;13:88.","journal-title":"Front Mol Neurosci"},{"key":"10_CR33","doi-asserted-by":"publisher","first-page":"227","DOI":"10.1016\/j.neulet.2006.11.062","volume":"412","author":"L Selby","year":"2007","unstructured":"Selby L, Zhang C, Sun Q-Q. Major defects in neocortical GABAergic inhibitory circuits in mice lacking the fragile X mental retardation protein. Neurosci Lett. 2007;412:227\u201332.","journal-title":"Neurosci Lett"},{"key":"10_CR34","doi-asserted-by":"publisher","first-page":"963","DOI":"10.1016\/j.biopsych.2007.09.008","volume":"63","author":"D Centonze","year":"2008","unstructured":"Centonze D, et al. Abnormal striatal GABA transmission in the mouse model for the fragile X syndrome. Biol Psychiatry. 2008;63:963\u201373.","journal-title":"Biol Psychiatry"},{"key":"10_CR35","doi-asserted-by":"publisher","first-page":"244","DOI":"10.1016\/j.bbr.2012.01.031","volume":"229","author":"I Heulens","year":"2012","unstructured":"Heulens I, D\u2019Hulst C, Van Dam D, De Deyn PP, Kooy RF. Pharmacological treatment of fragile X syndrome with GABAergic drugs in a knockout mouse model. Behav Brain Res. 2012;229:244\u20139.","journal-title":"Behav Brain Res"},{"key":"10_CR36","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 GABAA 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":"10_CR37","doi-asserted-by":"publisher","first-page":"15","DOI":"10.3389\/fnmol.2019.00015","volume":"12","author":"A Modgil","year":"2019","unstructured":"Modgil A, et al. Neuroactive steroids reverse tonic inhibitory deficits in fragile X syndrome mouse model. Front Mol Neurosci. 2019;12:15.","journal-title":"Front Mol Neurosci"},{"key":"10_CR38","doi-asserted-by":"publisher","first-page":"256","DOI":"10.1038\/nchembio.78","volume":"4","author":"S Chang","year":"2008","unstructured":"Chang S, et al. Identification of small molecules rescuing fragile X syndrome phenotypes in Drosophila. Nat Chem Biol. 2008;4:256\u201363.","journal-title":"Nat Chem Biol"},{"key":"10_CR39","doi-asserted-by":"publisher","unstructured":"Khalsa JH. Medical and health consequences of Marijuana. In: Marijuana and the Cannabinoids. Humana Press, pp. 237\u2013252. https:\/\/doi.org\/10.1007\/978-1-59259-947-9_10.","DOI":"10.1007\/978-1-59259-947-9_10"},{"key":"10_CR40","doi-asserted-by":"publisher","first-page":"1218","DOI":"10.3390\/genes12081218","volume":"12","author":"W Fyke","year":"2021","unstructured":"Fyke W, Velinov M. FMR1 and autism, an intriguing connection revisited. Genes (Basel). 2021;12:1218.","journal-title":"Genes (Basel)"},{"key":"10_CR41","doi-asserted-by":"publisher","first-page":"RC188","DOI":"10.1523\/JNEUROSCI.21-24-j0003.2001","volume":"21","author":"N Varma","year":"2001","unstructured":"Varma N, Carlson GC, Ledent C, Alger BE. Metabotropic glutamate receptors drive the endocannabinoid system in hippocampus. J Neurosci. 2001;21:RC188.","journal-title":"J Neurosci"},{"key":"10_CR42","doi-asserted-by":"publisher","first-page":"1508","DOI":"10.3389\/fphar.2018.01508","volume":"9","author":"D Garc\u00eda-Rinc\u00f3n","year":"2019","unstructured":"Garc\u00eda-Rinc\u00f3n D, et al. Contribution of altered endocannabinoid system to overactive mTORC1 signaling in focal cortical dysplasia. Front Pharmacol. 2019;9:1508.","journal-title":"Front Pharmacol"},{"key":"10_CR43","doi-asserted-by":"publisher","first-page":"603","DOI":"10.1038\/nm.3127","volume":"19","author":"A Busquets-Garcia","year":"2013","unstructured":"Busquets-Garcia A, et al. Targeting the endocannabinoid system in the treatment of fragile X syndrome. Nat Med. 2013;19:603\u20137.","journal-title":"Nat Med"},{"key":"10_CR44","doi-asserted-by":"publisher","first-page":"1500","DOI":"10.1038\/npp.2010.19","volume":"35","author":"M Maccarrone","year":"2010","unstructured":"Maccarrone M, et al. Abnormal mGlu 5 receptor\/endocannabinoid coupling in mice lacking FMRP and BC1 RNA. Neuropsychopharmacology. 2010;35:1500\u20139.","journal-title":"Neuropsychopharmacology"},{"key":"10_CR45","doi-asserted-by":"publisher","first-page":"1080","DOI":"10.1038\/ncomms2045","volume":"3","author":"K-M Jung","year":"2012","unstructured":"Jung K-M, et al. Uncoupling of the endocannabinoid signalling complex in a mouse model of fragile X syndrome. Nat Commun. 2012;3:1080.","journal-title":"Nat Commun"},{"key":"10_CR46","doi-asserted-by":"publisher","first-page":"47","DOI":"10.1186\/s11689-021-09394-x","volume":"13","author":"PS Pirbhoy","year":"2021","unstructured":"Pirbhoy PS, et al. Increased 2-arachidonoyl-sn-glycerol levels normalize cortical responses to sound and improve behaviors in Fmr1 KO mice. J Neurodev Disord. 2021;13:47.","journal-title":"J Neurodev Disord"},{"key":"10_CR47","doi-asserted-by":"publisher","first-page":"15616","DOI":"10.1523\/JNEUROSCI.3888-10.2010","volume":"30","author":"EK Osterweil","year":"2010","unstructured":"Osterweil EK, Krueger DD, Reinhold K, Bear MF. Hypersensitivity to mGluR5 and ERK1\/2 leads to excessive protein synthesis in the hippocampus of a mouse model of fragile X syndrome. J Neurosci. 2010;30:15616\u201327.","journal-title":"J Neurosci"},{"key":"10_CR48","doi-asserted-by":"publisher","first-page":"452","DOI":"10.3389\/fnmol.2017.00452","volume":"10","author":"RM Sar\u00e9","year":"2018","unstructured":"Sar\u00e9 RM, et al. Negative effects of chronic rapamycin treatment on behavior in a mouse model of fragile X syndrome. Front Mol Neurosci. 2018;10:452.","journal-title":"Front Mol Neurosci"},{"key":"10_CR49","doi-asserted-by":"publisher","first-page":"eaao0498","DOI":"10.1126\/scitranslmed.aao0498","volume":"11","author":"A Asiminas","year":"2019","unstructured":"Asiminas A, et al. Sustained correction of associative learning deficits after brief, early treatment in a rat model of fragile X syndrome. Sci Transl Med. 2019;11:eaao0498.","journal-title":"Sci Transl Med"},{"key":"10_CR50","doi-asserted-by":"publisher","first-page":"12","DOI":"10.1186\/s11689-020-09315-4","volume":"12","author":"AJ Thurman","year":"2020","unstructured":"Thurman AJ, et al. Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome. J Neurodev Disord. 2020;12:12.","journal-title":"J Neurodev Disord"},{"key":"10_CR51","doi-asserted-by":"publisher","first-page":"45","DOI":"10.1016\/j.eplepsyres.2012.09.018","volume":"104","author":"A Konopka","year":"2013","unstructured":"Konopka A, et al. Matrix metalloproteinase-9 (MMP-9) in human intractable epilepsy caused by focal cortical dysplasia. Epilepsy Res. 2013;104:45\u201358.","journal-title":"Epilepsy Res"},{"key":"10_CR52","doi-asserted-by":"publisher","first-page":"262","DOI":"10.1111\/cge.13469","volume":"95","author":"JA Ram\u00edrez-Cheyne","year":"2019","unstructured":"Ram\u00edrez-Cheyne JA, et al. Fragile X syndrome and connective tissue dysregulation. Clin Genet. 2019;95:262\u20137.","journal-title":"Clin Genet"},{"key":"10_CR53","doi-asserted-by":"publisher","first-page":"9867","DOI":"10.1523\/JNEUROSCI.1162-14.2014","volume":"34","author":"H Sidhu","year":"2014","unstructured":"Sidhu H, Dansie LE, Hickmott PW, Ethell DW, Ethell IM. Genetic removal of matrix metalloproteinase 9 rescues the symptoms of fragile X syndrome in a mouse model. J Neurosci. 2014;34:9867\u201379.","journal-title":"J Neurosci"},{"key":"10_CR54","doi-asserted-by":"publisher","first-page":"94","DOI":"10.1136\/jmg.2008.061796","volume":"46","author":"TV Bilousova","year":"2008","unstructured":"Bilousova TV, et al. Minocycline promotes dendritic spine maturation and improves behavioural performance in the fragile X mouse model. J Med Genet. 2008;46:94\u2013102.","journal-title":"J Med Genet"},{"key":"10_CR55","doi-asserted-by":"publisher","first-page":"41","DOI":"10.1002\/(SICI)1097-4547(19980101)51:1<41::AID-JNR4>3.0.CO;2-L","volume":"51","author":"E Berry-Kravis","year":"1998","unstructured":"Berry-Kravis E, Ciurlionis R. Overexpression of fragile X gene (FMR-1) transcripts increases cAMP production in neural cells. J Neurosci Res. 1998;51:41\u20138.","journal-title":"J Neurosci Res"},{"key":"10_CR56","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0000931","volume":"2","author":"DJ Kelley","year":"2007","unstructured":"Kelley DJ, et al. The cyclic AMP cascade is altered in the fragile X nervous system. PLoS One. 2007;2:e931.","journal-title":"PLoS One"},{"key":"10_CR57","doi-asserted-by":"publisher","first-page":"22","DOI":"10.1002\/ana.410310105","volume":"31","author":"E Berry-Kravis","year":"1992","unstructured":"Berry-Kravis E, Huttenlocher PR. Cyclic AMP metabolism in fragile X syndrome. Ann Neurol. 1992;31:22\u20136.","journal-title":"Ann Neurol"},{"key":"10_CR58","doi-asserted-by":"publisher","first-page":"638","DOI":"10.1203\/00006450-199511000-00002","volume":"38","author":"E Berry-Kravis","year":"1995","unstructured":"Berry-Kravis E, Hicar M, Ciurlionis R. Reduced cyclic AMP production in fragile X syndrome: cytogenetic and molecular correlations. Pediatr Res. 1995;38:638\u201343.","journal-title":"Pediatr Res"},{"key":"10_CR59","doi-asserted-by":"publisher","first-page":"396","DOI":"10.1523\/JNEUROSCI.1356-12.2015","volume":"35","author":"CH Choi","year":"2015","unstructured":"Choi CH, et al. PDE-4 inhibition rescues aberrant synaptic plasticity in drosophila and mouse models of fragile X syndrome. J Neurosci. 2015;35:396\u2013408.","journal-title":"J Neurosci"},{"key":"10_CR60","doi-asserted-by":"publisher","first-page":"265","DOI":"10.1007\/s13311-020-00968-6","volume":"18","author":"MJ Salcedo-Arellano","year":"2021","unstructured":"Salcedo-Arellano MJ, et al. Overlapping molecular pathways leading to autism Spectrum disorders, fragile X syndrome, and targeted treatments. Neurotherapeutics. 2021;18:265\u201383.","journal-title":"Neurotherapeutics"},{"key":"10_CR61","doi-asserted-by":"publisher","first-page":"10","DOI":"10.1186\/2040-2392-3-10","volume":"3","author":"HO Kalkman","year":"2012","unstructured":"Kalkman HO. A review of the evidence for the canonical Wnt pathway in autism spectrum disorders. Mol Autism. 2012;3:10.","journal-title":"Mol Autism"},{"key":"10_CR62","doi-asserted-by":"publisher","first-page":"198","DOI":"10.1016\/j.biopsych.2013.08.003","volume":"75","author":"AV Franklin","year":"2014","unstructured":"Franklin AV, et al. Glycogen synthase Kinase-3 inhibitors reverse deficits in long-term potentiation and cognition in fragile X mice. Biol Psychiatry. 2014;75:198\u2013206.","journal-title":"Biol Psychiatry"},{"key":"10_CR63","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0009706","volume":"5","author":"MA Mines","year":"2010","unstructured":"Mines MA, Yuskaitis CJ, King MK, Beurel E, Jope RS. GSK3 influences social preference and anxiety-related behaviors during social interaction in a mouse model of fragile X syndrome and autism. PLoS One. 2010;5:e9706.","journal-title":"PLoS One"},{"key":"10_CR64","doi-asserted-by":"publisher","first-page":"632","DOI":"10.1016\/j.bcp.2009.09.023","volume":"79","author":"CJ Yuskaitis","year":"2010","unstructured":"Yuskaitis CJ, et al. Lithium ameliorates altered glycogen synthase kinase-3 and behavior in a mouse model of fragile X syndrome. Biochem Pharmacol. 2010;79:632\u201346.","journal-title":"Biochem Pharmacol"},{"key":"10_CR65","doi-asserted-by":"publisher","first-page":"619","DOI":"10.1007\/s10571-020-00882-7","volume":"41","author":"J Baranova","year":"2021","unstructured":"Baranova J, et al. Autism spectrum disorder: signaling pathways and prospective therapeutic targets. Cell Mol Neurobiol. 2021;41:619\u201349.","journal-title":"Cell Mol Neurobiol"},{"key":"10_CR66","doi-asserted-by":"publisher","first-page":"1398","DOI":"10.1016\/j.cellsig.2012.03.001","volume":"24","author":"MA Mines","year":"2012","unstructured":"Mines MA, Jope RS. Brain region differences in regulation of Akt and GSK3 by chronic stimulant administration in mice. Cell Signal. 2012;24:1398\u2013405.","journal-title":"Cell Signal"},{"key":"10_CR67","doi-asserted-by":"publisher","first-page":"83","DOI":"10.1113\/JP271031","volume":"594","author":"P-Y Deng","year":"2016","unstructured":"Deng P-Y, 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.","journal-title":"J Physiol"},{"key":"10_CR68","doi-asserted-by":"publisher","first-page":"124","DOI":"10.1186\/s13023-014-0124-6","volume":"9","author":"B H\u00e9bert","year":"2014","unstructured":"H\u00e9bert B, et al. Rescue of fragile X syndrome phenotypes in Fmr1KO mice by a BKCa channel opener molecule. Orphanet J Rare Dis. 2014;9:124.","journal-title":"Orphanet J Rare Dis"},{"key":"10_CR69","doi-asserted-by":"publisher","first-page":"42","DOI":"10.1002\/mrdd.20007","volume":"10","author":"E Berry-Kravis","year":"2004","unstructured":"Berry-Kravis E, Potanos K. Psychopharmacology in fragile X syndrome?Present and future. Ment Retard Dev Disabil Res Rev. 2004;10:42\u20138.","journal-title":"Ment Retard Dev Disabil Res Rev"},{"key":"10_CR70","doi-asserted-by":"publisher","first-page":"2275","DOI":"10.1002\/ajmg.a.31388","volume":"140A","author":"K Sullivan","year":"2006","unstructured":"Sullivan K, et al. ADHD symptoms in children with FXS. Am J Med Genet Part A. 2006;140A:2275\u201388.","journal-title":"Am J Med Genet Part A"},{"key":"10_CR71","doi-asserted-by":"publisher","first-page":"744","DOI":"10.1542\/peds.95.5.744","volume":"95","author":"TL Baumgardner","year":"1995","unstructured":"Baumgardner TL, Reiss AL, Freund LS, Abrams MT. Specification of the neurobehavioral phenotype in males with fragile X syndrome. Pediatrics. 1995;95:744\u201352.","journal-title":"Pediatrics"},{"key":"10_CR72","doi-asserted-by":"publisher","first-page":"175","DOI":"10.1007\/s11689-011-9085-4","volume":"3","author":"JE Roberts","year":"2011","unstructured":"Roberts JE, et al. Treatment effects of stimulant medication in young boys with fragile X syndrome. J Neurodev Disord. 2011;3:175\u201384.","journal-title":"J Neurodev Disord"},{"key":"10_CR73","doi-asserted-by":"publisher","first-page":"377","DOI":"10.1002\/ajmg.1320300138","volume":"30","author":"RJ Hagerman","year":"1988","unstructured":"Hagerman RJ, Murphy MA, Wittenberger MD. A controlled trial of stimulant medication in children with the fragile X syndrome. Am J Med Genet. 1988;30:377\u201392.","journal-title":"Am J Med Genet"},{"key":"10_CR74","doi-asserted-by":"publisher","first-page":"168","DOI":"10.1002\/jemt.10067","volume":"57","author":"RJ Hagerman","year":"2002","unstructured":"Hagerman RJ, et al. Influence of stimulants on electrodermal studies in fragile X syndrome. Microsc Res Tech. 2002;57:168\u201373.","journal-title":"Microsc Res Tech"},{"key":"10_CR75","first-page":"336","volume":"8","author":"RJ Hagerman","year":"1995","unstructured":"Hagerman RJ, Riddle JE, Roberts LS, Breese K, Fulton M. Survey of the efficacy of clonidine in fragile X syndrome. Dev Brain Dysfunct. 1995;8:336\u201344.","journal-title":"Dev Brain Dysfunct"},{"key":"10_CR76","doi-asserted-by":"publisher","first-page":"1067","DOI":"10.1176\/appi.ajp.158.7.1067","volume":"158","author":"L Scahill","year":"2001","unstructured":"Scahill L, et al. A placebo-controlled study of Guanfacine in the treatment of children with tic disorders and attention deficit hyperactivity disorder. Am J Psychiatry. 2001;158:1067\u201374.","journal-title":"Am J Psychiatry"},{"key":"10_CR77","doi-asserted-by":"publisher","first-page":"875","DOI":"10.1007\/s00787-017-0962-6","volume":"26","author":"A Joseph","year":"2017","unstructured":"Joseph A, et al. Comparative efficacy and safety of attention-deficit\/hyperactivity disorder pharmacotherapies, including guanfacine extended release: a mixed treatment comparison. Eur Child Adolesc Psychiatry. 2017;26:875\u201397.","journal-title":"Eur Child Adolesc Psychiatry"},{"key":"10_CR78","doi-asserted-by":"publisher","first-page":"378","DOI":"10.1542\/peds.2008-0317","volume":"123","author":"RJ Hagerman","year":"2009","unstructured":"Hagerman RJ, et al. Advances in the treatment of fragile X syndrome. Pediatrics. 2009;123:378\u201390.","journal-title":"Pediatrics"},{"key":"10_CR79","doi-asserted-by":"publisher","first-page":"23","DOI":"10.1007\/s10882-008-9123-7","volume":"21","author":"MG Valdovinos","year":"2009","unstructured":"Valdovinos MG, Parsa RA, Alexander ML. Results of a nation-wide survey evaluating psychotropic medication use in fragile X syndrome. J Dev Phys Disabil. 2009;21:23\u201337.","journal-title":"J Dev Phys Disabil"},{"key":"10_CR80","doi-asserted-by":"publisher","first-page":"110","DOI":"10.5582\/irdr.2014.01027","volume":"3","author":"AC Hanson","year":"2014","unstructured":"Hanson AC, Hagerman RJ. Serotonin dysregulation in fragile X syndrome: implications for treatment. Intractable Rare Dis Res. 2014;3:110\u20137.","journal-title":"Intractable Rare Dis Res"},{"key":"10_CR81","doi-asserted-by":"publisher","first-page":"287","DOI":"10.1002\/1531-8249(199903)45:3<287::AID-ANA3>3.0.CO;2-9","volume":"45","author":"DC Chugani","year":"1999","unstructured":"Chugani DC, et al. Developmental changes in brain serotonin synthesis capacity in autistic and nonautistic children. Ann Neurol. 1999;45:287\u201395.","journal-title":"Ann Neurol"},{"key":"10_CR82","doi-asserted-by":"publisher","first-page":"571","DOI":"10.1002\/aur.1317","volume":"6","author":"A Oblak","year":"2013","unstructured":"Oblak A, Gibbs TT, Blatt GJ. Reduced serotonin receptor subtypes in a limbic and a neocortical region in autism. Autism Res. 2013;6:571\u201383.","journal-title":"Autism Res"},{"key":"10_CR83","doi-asserted-by":"publisher","first-page":"16","DOI":"10.1186\/2040-2392-4-16","volume":"4","author":"L Boccuto","year":"2013","unstructured":"Boccuto L, et al. Decreased tryptophan metabolism in patients with autism spectrum disorders. Mol Autism. 2013;4:16.","journal-title":"Mol Autism"},{"key":"10_CR84","doi-asserted-by":"publisher","first-page":"993","DOI":"10.1001\/archpsyc.1996.01830110029004","volume":"53","author":"C McDougle","year":"1996","unstructured":"McDougle C. Effects of tryptophan depletion in drug-free adults with autistic disorder. Arch Gen Psychiatry. 1996;53:993.","journal-title":"Arch Gen Psychiatry"},{"key":"10_CR85","doi-asserted-by":"publisher","first-page":"20338","DOI":"10.1038\/srep20338","volume":"6","author":"N-N Song","year":"2016","unstructured":"Song N-N, et al. Reducing central serotonin in adulthood promotes hippocampal neurogenesis. Sci Rep. 2016;6:20338.","journal-title":"Sci Rep"},{"key":"10_CR86","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0172270","volume":"12","author":"C Zhou","year":"2017","unstructured":"Zhou C, et al. Meta-analyses of comparative efficacy of antidepressant medications on peripheral BDNF concentration in patients with depression. PLoS One. 2017;12:e0172270.","journal-title":"PLoS One"},{"key":"10_CR87","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1155\/2012\/280813","volume":"2012","author":"TI Winarni","year":"2012","unstructured":"Winarni TI, Schneider A, Borodyanskara M, Hagerman RJ. Early intervention combined with targeted treatment promotes cognitive and behavioral improvements in young children with fragile X syndrome. Case Rep Genet. 2012;2012:1\u20134.","journal-title":"Case Rep Genet"},{"key":"10_CR88","doi-asserted-by":"publisher","first-page":"619","DOI":"10.1097\/DBP.0000000000000334","volume":"37","author":"L Greiss Hess","year":"2016","unstructured":"Greiss Hess L, et al. A randomized, double-blind, placebo-controlled trial of low-dose sertraline in young children with fragile X syndrome. J Dev Behav Pediatr. 2016;37:619\u201328.","journal-title":"J Dev Behav Pediatr"},{"key":"10_CR89","doi-asserted-by":"publisher","first-page":"810","DOI":"10.3389\/fpsyt.2019.00810","volume":"10","author":"LA Potter","year":"2019","unstructured":"Potter LA, et al. A randomized controlled trial of sertraline in young children with autism spectrum disorder. Front Psych. 2019;10:810.","journal-title":"Front Psych"},{"key":"10_CR90","doi-asserted-by":"publisher","first-page":"581429","DOI":"10.3389\/fneur.2020.581429","volume":"11","author":"A Rajaratnam","year":"2020","unstructured":"Rajaratnam A, et al. Review of autism profiles and response to sertraline in fragile X syndrome-associated autism vs. non-syndromic autism; next steps for targeted treatment. Front Neurol. 2020;11:581429.","journal-title":"Front Neurol"},{"key":"10_CR91","first-page":"143","volume":"4","author":"RN Amaria","year":"2001","unstructured":"Amaria RN, Billeisen LL, Hagerman RJ. Medication use in fragile X syndrome. Ment Heal Asp Dev Disabil. 2001;4:143\u20137.","journal-title":"Ment Heal Asp Dev Disabil"},{"key":"10_CR92","first-page":"155","volume":"7","author":"RJ Hagerman","year":"1994","unstructured":"Hagerman RJ, et al. A survey of fluoxetine therapy in fragile X syndrome. Dev Brain Dysfunct. 1994;7:155\u201364.","journal-title":"Dev Brain Dysfunct"},{"key":"10_CR93","doi-asserted-by":"publisher","first-page":"314","DOI":"10.1056\/NEJMoa013171","volume":"347","author":"JT McCracken","year":"2002","unstructured":"McCracken JT, et al. Risperidone in children with autism and serious behavioral problems. N Engl J Med. 2002;347:314\u201321.","journal-title":"N Engl J Med"},{"key":"10_CR94","doi-asserted-by":"publisher","first-page":"e634","DOI":"10.1542\/peds.2003-0264-F","volume":"114","author":"S Shea","year":"2004","unstructured":"Shea S, et al. Risperidone in the treatment of disruptive behavioral symptoms in children with autistic and other pervasive developmental disorders. Pediatrics. 2004;114:e634\u201341.","journal-title":"Pediatrics"},{"key":"10_CR95","doi-asserted-by":"publisher","first-page":"450","DOI":"10.1177\/08830738060210060801","volume":"21","author":"R Nagaraj","year":"2006","unstructured":"Nagaraj R, Singhi P, Malhi P. Risperidone in children with autism: randomized, placebo-controlled, double-blind study. J Child Neurol. 2006;21:450\u20135.","journal-title":"J Child Neurol"},{"key":"10_CR96","doi-asserted-by":"publisher","first-page":"85","DOI":"10.1007\/s00213-011-2194-7","volume":"216","author":"CA Erickson","year":"2011","unstructured":"Erickson CA, et al. A prospective open-label study of aripiprazole in fragile X syndrome. Psychopharmacology. 2011;216:85\u201390.","journal-title":"Psychopharmacology"},{"key":"10_CR97","doi-asserted-by":"publisher","first-page":"274","DOI":"10.1089\/cap.2017.0057","volume":"28","author":"KC Dominick","year":"2018","unstructured":"Dominick KC, et al. Risperidone treatment for irritability in fragile X syndrome. J Child Adolesc Psychopharmacol. 2018;28:274\u20138.","journal-title":"J Child Adolesc Psychopharmacol"},{"key":"10_CR98","doi-asserted-by":"publisher","first-page":"127","DOI":"10.1038\/s42003-020-0833-4","volume":"3","author":"Q Ding","year":"2020","unstructured":"Ding Q, et al. Transcriptome signature analysis repurposes trifluoperazine for the treatment of fragile X syndrome in mouse model. Commun Biol. 2020;3:127.","journal-title":"Commun Biol"},{"key":"10_CR99","first-page":"578","volume":"8","author":"L Wang","year":"2015","unstructured":"Wang L, Liu Y, Li S, Long Z-Y, Wu Y-M. Wnt signaling pathway participates in valproic acid-induced neuronal differentiation of neural stem cells. Int J Clin Exp Pathol. 2015;8:578\u201385.","journal-title":"Int J Clin Exp Pathol"},{"key":"10_CR100","doi-asserted-by":"publisher","first-page":"759","DOI":"10.1002\/2211-5463.12220","volume":"7","author":"M-Y Xia","year":"2017","unstructured":"Xia M-Y, et al. Activation of Wnt\/\u03b2-catenin signaling by lithium chloride attenuates <scp>d<\/scp> \u2212galactose-induced neurodegeneration in the auditory cortex of a rat model of aging. FEBS Open Bio. 2017;7:759\u201376.","journal-title":"FEBS Open Bio"},{"key":"10_CR101","doi-asserted-by":"publisher","first-page":"1145","DOI":"10.1016\/j.nbd.2011.12.037","volume":"45","author":"Z-H Liu","year":"2012","unstructured":"Liu Z-H, Huang T, Smith CB. Lithium reverses increased rates of cerebral protein synthesis in a mouse model of fragile X syndrome. Neurobiol Dis. 2012;45:1145\u201352.","journal-title":"Neurobiol Dis"},{"key":"10_CR102","doi-asserted-by":"publisher","first-page":"293","DOI":"10.1097\/DBP.0b013e31817dc447","volume":"29","author":"E Berry-Kravis","year":"2008","unstructured":"Berry-Kravis E, et al. Open-label treatment trial of lithium to target the underlying defect in fragile X syndrome. J Dev Behav Pediatr. 2008;29:293\u2013302.","journal-title":"J Dev Behav Pediatr"},{"key":"10_CR103","doi-asserted-by":"publisher","first-page":"266","DOI":"10.1136\/jmg.2008.063701","volume":"46","author":"E Berry-Kravis","year":"2009","unstructured":"Berry-Kravis E, et al. A pilot open label, single dose trial of fenobam in adults with fragile X syndrome. J Med Genet. 2009;46:266\u201371.","journal-title":"J Med Genet"},{"key":"10_CR104","doi-asserted-by":"publisher","first-page":"16970","DOI":"10.1038\/s41598-018-34978-4","volume":"8","author":"R Hagerman","year":"2018","unstructured":"Hagerman R, et al. Mavoglurant in fragile X syndrome: results of two open-label, extension trials in adults and adolescents. Sci Rep. 2018;8:16970.","journal-title":"Sci Rep"},{"key":"10_CR105","doi-asserted-by":"publisher","first-page":"321ra5","DOI":"10.1126\/scitranslmed.aab4109","volume":"8","author":"E Berry-Kravis","year":"2016","unstructured":"Berry-Kravis E, et al. Mavoglurant in fragile X syndrome: results of two randomized, double-blind, placebo-controlled trials. Sci Transl Med. 2016;8:321ra5.","journal-title":"Sci Transl Med"},{"key":"10_CR106","doi-asserted-by":"publisher","first-page":"503","DOI":"10.1038\/npp.2017.177","volume":"43","author":"EA Youssef","year":"2018","unstructured":"Youssef EA, et al. Effect of the mGluR5-NAM basimglurant on behavior in adolescents and adults with fragile X syndrome in a randomized, double-blind, placebo-controlled trial: FragXis phase 2 results. Neuropsychopharmacology. 2018;43:503\u201312.","journal-title":"Neuropsychopharmacology"},{"key":"10_CR107","doi-asserted-by":"publisher","first-page":"18","DOI":"10.1124\/mol.109.056127","volume":"76","author":"LKK Pacey","year":"2009","unstructured":"Pacey LKK, Heximer SP, Hampson DR. Increased GABA B receptor-mediated signaling reduces the susceptibility of fragile X knockout mice to audiogenic seizures. Mol Pharmacol. 2009;76:18\u201324.","journal-title":"Mol Pharmacol"},{"key":"10_CR108","doi-asserted-by":"publisher","first-page":"152ra127","DOI":"10.1126\/scitranslmed.3004214","volume":"4","author":"EM Berry-Kravis","year":"2012","unstructured":"Berry-Kravis EM, et al. Effects of STX209 (arbaclofen) on neurobehavioral function in children and adults with fragile X syndrome: a randomized, controlled, phase 2 trial. Sci Transl Med. 2012;4:152ra127.","journal-title":"Sci Transl Med"},{"key":"10_CR109","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1186\/s11689-016-9181-6","volume":"9","author":"E Berry-Kravis","year":"2017","unstructured":"Berry-Kravis E, et al. Arbaclofen in fragile X syndrome: results of phase 3 trials. J Neurodev Disord. 2017;9:3.","journal-title":"J Neurodev Disord"},{"key":"10_CR110","doi-asserted-by":"publisher","first-page":"26","DOI":"10.1186\/s11689-017-9207-8","volume":"9","author":"A Ligsay","year":"2017","unstructured":"Ligsay A, et al. A randomized double-blind, placebo-controlled trial of ganaxolone in children and adolescents with fragile X syndrome. J Neurodev Disord. 2017;9:26.","journal-title":"J Neurodev Disord"},{"key":"10_CR111","doi-asserted-by":"publisher","first-page":"2762","DOI":"10.3389\/fphar.2021.757825","volume":"12","author":"DB Budimirovic","year":"2021","unstructured":"Budimirovic DB, et al. Gaboxadol in fragile X syndrome: a 12-week randomized, double-blind, parallel-group, phase 2a study. Front Pharmacol. 2021;12:2762.","journal-title":"Front Pharmacol"},{"key":"10_CR112","doi-asserted-by":"publisher","first-page":"85","DOI":"10.1016\/j.jns.2008.12.003","volume":"278","author":"MJ Bickerdike","year":"2009","unstructured":"Bickerdike MJ, et al. NNZ-2566: a Gly\u2013Pro\u2013Glu analogue with neuroprotective efficacy in a rat model of acute focal stroke. J Neurol Sci. 2009;278:85\u201390.","journal-title":"J Neurol Sci"},{"key":"10_CR113","doi-asserted-by":"publisher","first-page":"30","DOI":"10.1016\/j.pediatrneurol.2020.04.019","volume":"110","author":"E Berry-Kravis","year":"2020","unstructured":"Berry-Kravis E, et al. A double-blind, randomized, placebo-controlled clinical study of trofinetide in the treatment of fragile X syndrome. Pediatr Neurol. 2020;110:30\u201341.","journal-title":"Pediatr Neurol"},{"key":"10_CR114","doi-asserted-by":"publisher","first-page":"746","DOI":"10.1016\/j.ajhg.2012.03.004","volume":"90","author":"H Lee","year":"2012","unstructured":"Lee H, et al. Exome sequencing identifies PDE4D mutations in acrodysostosis. Am J Hum Genet. 2012;90:746\u201351.","journal-title":"Am J Hum Genet"},{"key":"10_CR115","doi-asserted-by":"publisher","first-page":"1311","DOI":"10.1021\/acschemneuro.0c00077","volume":"11","author":"Y Wakabayashi","year":"2020","unstructured":"Wakabayashi Y, et al. Discovery, radiolabeling, and evaluation of subtype-selective inhibitors for positron emission tomography imaging of brain phosphodiesterase-4D. ACS Chem Neurosci. 2020;11:1311\u201323.","journal-title":"ACS Chem Neurosci"},{"key":"10_CR116","doi-asserted-by":"publisher","first-page":"862","DOI":"10.1038\/s41591-021-01321-w","volume":"27","author":"EM Berry-Kravis","year":"2021","unstructured":"Berry-Kravis EM, et al. Inhibition of phosphodiesterase-4D in adults with fragile X syndrome: a randomized, placebo-controlled, phase 2 clinical trial. Nat Med. 2021;27:862\u201370.","journal-title":"Nat Med"},{"key":"10_CR117","doi-asserted-by":"publisher","first-page":"1897","DOI":"10.1002\/ajmg.a.36023","volume":"161","author":"M Dziembowska","year":"2013","unstructured":"Dziembowska M, et al. High MMP-9 activity levels in fragile X syndrome are lowered by minocycline. Am J Med Genet Part A. 2013;161:1897\u2013903.","journal-title":"Am J Med Genet Part A"},{"key":"10_CR118","doi-asserted-by":"publisher","first-page":"91","DOI":"10.1186\/1471-2377-10-91","volume":"10","author":"C Paribello","year":"2010","unstructured":"Paribello C, et al. Open-label add-on treatment trial of minocycline in fragile X syndrome. BMC Neurol. 2010;10:91.","journal-title":"BMC Neurol"},{"key":"10_CR119","doi-asserted-by":"publisher","first-page":"147","DOI":"10.1097\/DBP.0b013e318287cd17","volume":"34","author":"MJS Leigh","year":"2013","unstructured":"Leigh MJS, et al. A randomized double-blind, placebo-controlled trial of minocycline in children and adolescents with fragile X syndrome. J Dev Behav Pediatr. 2013;34:147\u201355.","journal-title":"J Dev Behav Pediatr"},{"key":"10_CR120","doi-asserted-by":"publisher","first-page":"e956","DOI":"10.1002\/mgg3.956","volume":"7","author":"HMB Biag","year":"2019","unstructured":"Biag HMB, et al. Metformin treatment in young children with fragile X syndrome. Mol Genet Genomic Med. 2019;7:e956.","journal-title":"Mol Genet Genomic Med"},{"key":"10_CR121","doi-asserted-by":"publisher","first-page":"216","DOI":"10.1111\/cge.13039","volume":"93","author":"ABC Dy","year":"2018","unstructured":"Dy ABC, et al. Metformin as targeted treatment in fragile X syndrome. Clin Genet. 2018;93:216\u201322.","journal-title":"Clin Genet"},{"key":"10_CR122","doi-asserted-by":"publisher","first-page":"1145","DOI":"10.3389\/fneur.2019.01145","volume":"10","author":"P Fleury-Teixeira","year":"2019","unstructured":"Fleury-Teixeira P, Caixeta FV, Ramires da Silva LC, Brasil-Neto JP, Malcher-Lopes R. Effects of CBD-enriched cannabis sativa extract on autism spectrum disorder symptoms: an observational study of 18 participants undergoing compassionate use. Front Neurol. 2019;10:1145.","journal-title":"Front Neurol"},{"key":"10_CR123","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1089\/can.2018.0053","volume":"4","author":"N Tartaglia","year":"2019","unstructured":"Tartaglia N, Bonn-Miller M, Hagerman R. Treatment of fragile X syndrome with Cannabidiol: a case series study and brief review of the literature. Cannabis Cannabinoid Res. 2019;4:3\u20139.","journal-title":"Cannabis Cannabinoid Res"},{"key":"10_CR124","doi-asserted-by":"publisher","first-page":"16","DOI":"10.1186\/s11689-019-9277-x","volume":"11","author":"H Heussler","year":"2019","unstructured":"Heussler H, et al. A phase 1\/2, open-label assessment of the safety, tolerability, and efficacy of transdermal cannabidiol (ZYN002) for the treatment of pediatric fragile X syndrome. J Neurodev Disord. 2019;11:16.","journal-title":"J Neurodev Disord"},{"key":"10_CR125","first-page":"S258","volume":"60","author":"J Palumbo","year":"2021","unstructured":"Palumbo J, et al. Reconnect (ZYN2-CL-033): design of a phase 3 trial of ZYN002 Cannabidiol transdermal gel in children and adolescents with fragile x syndrome based upon learnings from connect-FX (ZYN2-CL-016). J Am Acad Child Adolesc Psychiatry. 2021;60:S258.","journal-title":"J Am Acad Child Adolesc Psychiatry"},{"key":"10_CR126","doi-asserted-by":"publisher","first-page":"629","DOI":"10.3390\/brainsci10090629","volume":"10","author":"S Luu","year":"2020","unstructured":"Luu S, et al. Response to placebo in fragile X syndrome clinical trials: an initial analysis. Brain Sci. 2020;10:629.","journal-title":"Brain Sci"},{"key":"10_CR127","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0133316","volume":"10","author":"A Curie","year":"2015","unstructured":"Curie A, et al. Placebo responses in genetically determined intellectual disability: a meta-analysis. PLoS One. 2015;10:e0133316.","journal-title":"PLoS One"},{"key":"10_CR128","doi-asserted-by":"publisher","first-page":"7","DOI":"10.1515\/tnsci-2017-0002","volume":"8","author":"PQ Duy","year":"2017","unstructured":"Duy PQ, Budimirovic DB. Fragile X syndrome: lessons learned from the most translated neurodevelopmental disorder in clinical trials. Transl Neurosci. 2017;8:7\u20138.","journal-title":"Transl Neurosci"},{"key":"10_CR129","doi-asserted-by":"publisher","first-page":"49","DOI":"10.1016\/j.euroneuro.2021.02.020","volume":"48","author":"C D\u00edaz-Caneja","year":"2021","unstructured":"D\u00edaz-Caneja C, et al. A white paper on a neurodevelopmental framework for drug discovery in autism and other neurodevelopmental disorders. Eur Neuropsychopharmacol. 2021;48:49\u201388.","journal-title":"Eur Neuropsychopharmacol"},{"key":"10_CR130","doi-asserted-by":"publisher","first-page":"264","DOI":"10.3389\/fnhum.2012.00264","volume":"6","author":"IS Knoth","year":"2012","unstructured":"Knoth IS, Lipp\u00e9 S. Event-related potential alterations in fragile X syndrome. Front Hum Neurosci. 2012;6:264.","journal-title":"Front Hum Neurosci"},{"key":"10_CR131","doi-asserted-by":"publisher","first-page":"11","DOI":"10.1186\/s11689-017-9191-z","volume":"9","author":"J Wang","year":"2017","unstructured":"Wang J, et al. A resting EEG study of neocortical hyperexcitability and altered functional connectivity in fragile X syndrome. J Neurodev Disord. 2017;9:11.","journal-title":"J Neurodev Disord"},{"key":"10_CR132","doi-asserted-by":"publisher","first-page":"104794","DOI":"10.1016\/j.nbd.2020.104794","volume":"138","author":"CR Jonak","year":"2020","unstructured":"Jonak CR, Lovelace JW, Ethell IM, Razak KA, Binder DK. Multielectrode array analysis of EEG biomarkers in a mouse model of fragile X syndrome. Neurobiol Dis. 2020;138:104794.","journal-title":"Neurobiol Dis"},{"key":"10_CR133","doi-asserted-by":"publisher","first-page":"18799","DOI":"10.1038\/s41598-020-75893-x","volume":"10","author":"N Kozono","year":"2020","unstructured":"Kozono N, Okamura A, Honda S, Matsumoto M, Mihara T. Gamma power abnormalities in a Fmr1-targeted transgenic rat model of fragile X syndrome. Sci Rep. 2020;10:18799.","journal-title":"Sci Rep"},{"key":"10_CR134","doi-asserted-by":"publisher","first-page":"133","DOI":"10.1016\/j.nec.2010.11.001","volume":"22","author":"GH Glover","year":"2011","unstructured":"Glover GH. Overview of functional magnetic resonance imaging. Neurosurg Clin N Am. 2011;22:133\u20139.","journal-title":"Neurosurg Clin N Am"},{"key":"10_CR135","doi-asserted-by":"publisher","first-page":"15897","DOI":"10.1038\/s41598-021-94967-y","volume":"11","author":"M Mody","year":"2021","unstructured":"Mody M, et al. In vivo imaging of mGlu5 receptor expression in humans with fragile X syndrome towards development of a potential biomarker. Sci Rep. 2021;11:15897.","journal-title":"Sci Rep"},{"key":"10_CR136","doi-asserted-by":"publisher","first-page":"14","DOI":"10.1186\/s11689-017-9193-x","volume":"9","author":"DB Budimirovic","year":"2017","unstructured":"Budimirovic DB, et al. Updated report on tools to measure outcomes of clinical trials in fragile X syndrome. J Neurodev Disord. 2017;9:14.","journal-title":"J Neurodev Disord"},{"key":"10_CR137","doi-asserted-by":"publisher","first-page":"35","DOI":"10.1186\/s11689-016-9167-4","volume":"8","author":"D Hessl","year":"2016","unstructured":"Hessl D, et al. The NIH toolbox cognitive battery for intellectual disabilities: three preliminary studies and future directions. J Neurodev Disord. 2016;8:35.","journal-title":"J Neurodev Disord"},{"key":"10_CR138","doi-asserted-by":"publisher","first-page":"245","DOI":"10.1097\/DBP.0b013e31828742fc","volume":"34","author":"E Berry-Kravis","year":"2013","unstructured":"Berry-Kravis E, et al. Development of an expressive language sampling procedure in fragile X syndrome. J Dev Behav Pediatr. 2013;34:245\u201351.","journal-title":"J Dev Behav Pediatr"},{"key":"10_CR139","doi-asserted-by":"publisher","first-page":"10","DOI":"10.1186\/s11689-020-09313-6","volume":"12","author":"L Abbeduto","year":"2020","unstructured":"Abbeduto L, et al. Expressive language sampling as a source of outcome measures for treatment studies in fragile X syndrome: feasibility, practice effects, test-retest reliability, and construct validity. J Neurodev Disord. 2020;12:10.","journal-title":"J Neurodev Disord"},{"key":"10_CR140","doi-asserted-by":"publisher","first-page":"1431","DOI":"10.1007\/s10803-010-1005-z","volume":"40","author":"JL Taylor","year":"2010","unstructured":"Taylor JL, Seltzer MM. Changes in the autism behavioral phenotype during the transition to adulthood. J Autism Dev Disord. 2010;40:1431\u201346.","journal-title":"J Autism Dev Disord"},{"key":"10_CR141","doi-asserted-by":"publisher","first-page":"97","DOI":"10.3810\/pgm.2010.09.2206","volume":"122","author":"TE Wilens","year":"2010","unstructured":"Wilens TE, Spencer TJ. Understanding attention-deficit\/hyperactivity disorder from childhood to adulthood. Postgrad Med. 2010;122:97\u2013109.","journal-title":"Postgrad Med"},{"key":"10_CR142","doi-asserted-by":"publisher","first-page":"47","DOI":"10.1186\/s11689-016-9179-0","volume":"8","author":"M Lee","year":"2016","unstructured":"Lee M, Martin GE, Berry-Kravis E, Losh M. A developmental, longitudinal investigation of autism phenotypic profiles in fragile X syndrome. J Neurodev Disord. 2016;8:47.","journal-title":"J Neurodev Disord"},{"key":"10_CR143","doi-asserted-by":"publisher","first-page":"13","DOI":"10.1186\/s11689-020-09310-9","volume":"12","author":"KA Razak","year":"2020","unstructured":"Razak KA, Dominick KC, Erickson CA. Developmental studies in fragile X syndrome. J Neurodev Disord. 2020;12:13.","journal-title":"J Neurodev Disord"},{"key":"10_CR144","doi-asserted-by":"publisher","first-page":"718953","DOI":"10.3389\/fpsyt.2021.718953","volume":"12","author":"DC Stoppel","year":"2021","unstructured":"Stoppel DC, McCamphill PK, Senter RK, Heynen AJ, Bear MF. mGluR5 negative modulators for fragile X: treatment resistance and persistence. Front Psych. 2021;12:718953.","journal-title":"Front Psych"},{"key":"10_CR145","doi-asserted-by":"publisher","first-page":"300","DOI":"10.3389\/fnmol.2018.00300","volume":"11","author":"L Wang","year":"2018","unstructured":"Wang L, Kempton JB, Brigande JV. Gene therapy in mouse models of deafness and balance dysfunction. Front Mol Neurosci. 2018;11:300.","journal-title":"Front Mol Neurosci"}],"container-title":["Advances in Neurobiology","Drug Development in Psychiatry"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/978-3-031-21054-9_10","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,3,16]],"date-time":"2023-03-16T13:06:48Z","timestamp":1678972008000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/978-3-031-21054-9_10"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023]]},"ISBN":["9783031210532","9783031210549"],"references-count":145,"URL":"https:\/\/doi.org\/10.1007\/978-3-031-21054-9_10","relation":{},"ISSN":["2190-5215","2190-5223"],"issn-type":[{"value":"2190-5215","type":"print"},{"value":"2190-5223","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023]]},"assertion":[{"value":"17 March 2023","order":1,"name":"first_online","label":"First Online","group":{"name":"ChapterHistory","label":"Chapter History"}}]}}