Arbaclofen in fragile X syndrome: results of phase 3 trials
Open accessAbstract
Arbaclofen improved multiple abnormal phenotypes in animal models of fragile X syndrome (FXS) and showed promising results in a phase 2 clinical study. The objective of the study is to determine safety and efficacy of arbaclofen for social avoidance in FXS. Two phase 3 placebo-controlled trials were conducted, a flexible dose trial in subjects age 12–50 (209FX301, adolescent/adult study) and a fixed dose trial in subjects age 5–11 (209FX302, child study). The primary endpoint for both trials was the Social Avoidance subscale of the Aberrant Behavior Checklist-Community Edition, FXS-specific (ABC-C FX ). Secondary outcomes included other ABC-C FX subscale scores, Clinical Global Impression-Improvement (CGI-I), Clinical Global Impression-Severity (CGI-S), and Vineland Adaptive Behavior Scales, Second Edition (Vineland-II) Socialization domain score. A total 119 of 125 randomized subjects completed the adolescent/adult study ( n = 57 arbaclofen, 62 placebo) and 159/172 completed the child study (arbaclofen 5 BID n = 38; 10 BID n = 39; 10 TID n = 38; placebo n = 44). There were no serious adverse events (AEs); the most common AEs included somatic (headache, vomiting, nausea), neurobehavioral (irritability/agitation, anxiety, hyperactivity), decreased appetite, and infectious conditions, many of which were also common on placebo. In the combined studies, there were 13 discontinuations ( n = 12 arbaclofen, 1 placebo) due to AEs (all neurobehavioral). The adolescent/adult study did not show benefit for arbaclofen over placebo for any measure. In the child study, the highest dose group showed benefit over placebo on the ABC-C FX Irritability subscale ( p = 0.03) and Parenting Stress Index (PSI, p = 0.03) and trends toward benefit on the ABC-C FX Social Avoidance and Hyperactivity subscales (both p < 0.1) and CGI-I ( p = 0.119). Effect size in the highest dose group was similar to effect sizes for FDA-approved serotonin reuptake inhibitors (SSRIs). Arbaclofen did not meet the primary outcome of improved social avoidance in FXS in either study. Data from secondary measures in the child study suggests younger patients may derive benefit, but additional studies with a larger cohort on higher doses would be required to confirm this finding. The reported studies illustrate the challenges but represent a significant step forward in translating targeted treatments from preclinical models to clinical trials in humans with FXS.
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In the arbaclofen phase III study, children receiving the highest dose improved over placebo on the secondary ABC-CFX Irritability subscale and the Parenting Stress Index, both with P = 0.03.
“In the phase III study, children receiving the highest dose of arbaclofen also showed improvement over placebo on the secondary end points of the ABC-CFX Irritability subscale (P = 0.03) and the Parenting Stress Index (P = 0.03).”
From Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome (2018) · cites this paper as [72]
In the paediatric arbaclofen phase III study, the primary endpoint, ABC-CFX Social Avoidance, narrowly missed significant improvement with P = 0.08.
“In the phase III study carried out in children, the primary end point (ABC-CFX Social Avoidance subscale) narrowly missed significant improvement (P = 0.08)72.”
From Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome (2018) · cites this paper as [72]
The arbaclofen phase III trial was limited by incomplete enrolment due to sponsor financial issues, fixed rather than flexible dosing, and possible symptom inflation by families to meet inclusion criteria.
“This trial was limited by a lack of full enrolment due to financial issues for the sponsor, the use of fixed rather than flexible dosing and possible inflation of symptoms by families in order to meet inclusion criteria72.”
From Drug development for neurodevelopmental disorders: lessons learned from fragile X syndrome (2018) · cites this paper as [72]
Pediatric trials of arbaclofen in FXS did not reach significance for the primary outcome measure.
“However, the adult studies of FXS did not demonstrate efficacy and the pediatric trials did not reach significance for the primary outcome measure, but did show limited improvements in secondary measures including the Parenting Stress Index because of lowered irritability in the children [132].”
From An Update on Psychopharmacological Treatment of Autism Spectrum Disorder (2022) · cites this paper as [132]
Adult studies of FXS with arbaclofen did not demonstrate efficacy.
“However, the adult studies of FXS did not demonstrate efficacy and the pediatric trials did not reach significance for the primary outcome measure, but did show limited improvements in secondary measures including the Parenting Stress Index because of lowered irritability in the children [132].”
From An Update on Psychopharmacological Treatment of Autism Spectrum Disorder (2022) · cites this paper as [132]
Pediatric trials of arbaclofen in FXS showed limited improvements in secondary measures including the Parenting Stress Index because of lowered irritability in the children.
“However, the adult studies of FXS did not demonstrate efficacy and the pediatric trials did not reach significance for the primary outcome measure, but did show limited improvements in secondary measures including the Parenting Stress Index because of lowered irritability in the children [132].”
From An Update on Psychopharmacological Treatment of Autism Spectrum Disorder (2022) · cites this paper as [132]
The promising mouse studies led to human trials in FXS that initially showed improvements in those with ASD plus FXS leading to phase 3 trials in FXS.
“The promising mouse studies led to human trials in FXS [131] that initially showed improvements in those with ASD plus FXS leading to phase 3 trials in FXS [132].”
From An Update on Psychopharmacological Treatment of Autism Spectrum Disorder (2022) · cites this paper as [132]
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Cites
(1976) · Medical Entomology and Zoology · Cited by 8,899 · see citing works
Owens, J. A., Spirito, A., & McGuinn, M. (2000) · Sleep · doi:10.1093/sleep/23.8.1d · Cited by 2,535 · see citing works
Selective publication of antidepressant trials and its influence on apparent efficacy
Turner, E. H., Matthews, A. M., Linardatos, E., Tell, R. A., & Rosenthal, R. (2008) · New England Journal of Medicine · doi:10.1056/nejmsa065779 · Cited by 2,435 · see citing works
Risperidone in Children with Autism and Serious Behavioral Problems
McCracken, J. T., McGough, J., Shah, B., Cronin, P., Hong, D., Aman, M. G., Arnold, L. E., Lindsay, R., Nash, P., Hollway, J., et al. (2002) · New England Journal of Medicine · doi:10.1056/nejmoa013171 · Cited by 1,574 · see citing works
Size of Treatment Effects and Their Importance to Clinical Research and Practice
KRAEMER, H. C., & Kupfer, D. J. (2005) · Biological Psychiatry · doi:10.1016/j.biopsych.2005.09.014 · Cited by 850 · see citing works
Devys, D., Lutz, Y., Rouyer, N., Bellocq, J., & Mandel, J. (1993) · Nature Genetics · doi:10.1038/ng0893-335 · Cited by 781 · see citing works
Advances in the Treatment of Fragile X Syndrome
Hagerman, R. J., Berry-Kravis, E., Kaufmann, W. E., Ono, M. Y., Tartaglia, N., Lachiewicz, A., Kronk, R., Delahunty, C., Hessl, D., Visootsak, J., et al. (2009) · Pediatrics · doi:10.1542/peds.2008-0317 · Cited by 596 · see citing works
Self-citationPsychometric characteristics of the aberrant behavior checklist.
Aman, M. G., Singh, N. N., Stewart, A. W., & Field, C. J. (1985) · PubMed · Cited by 458 · see citing works
Epidemiology of fragile X syndrome: A systematic review and meta‐analysis
Hunter, J. E., Rivero‐Arias, O., Angelov, A., Kim, E., Fotheringham, I., & Leal, J. (2014) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.36511 · Cited by 445 · see citing works
Autism Profiles of Males With Fragile X Syndrome
Harris, S. W., Hessl, D., Goodlin-Jones, B., Ferranti, J., Bacalman, S., Barbato, I., Tassone, F., Hagerman, P. J., Herman, K., & Hagerman, R. J. (2008) · American Journal on Mental Retardation · doi:10.1352/2008.113:427-438 · Cited by 396 · see citing works
Self-citationThe Pathophysiology of Fragile X (and What It Teaches Us about Synapses)
Bhakar, A. L., Dölen, G., & Bear, M. F. (2012) · Annual Review of Neuroscience · doi:10.1146/annurev-neuro-060909-153138 · Cited by 370 · see citing works
Self-citationBerry-Kravis, E. M., Hessl, D., Rathmell, B., Zarevics, P., Cherubini, M., Walton-Bowen, K., Mu, Y., Nguyen, D. V., Gonzalez-Heydrich, J., Wang, P. P., et al. (2012) · Science Translational Medicine · doi:10.1126/scitranslmed.3004214 · Cited by 328 · see citing works
Self-citationOlmos-Serrano, J. L., Paluszkiewicz, S. M., Martin, B. S., Kaufmann, W. E., Corbin, J. G., & Huntsman, M. M. (2010) · Journal of Neuroscience · doi:10.1523/jneurosci.1714-10.2010 · Cited by 309 · see citing works
Self-citationBerry-Kravis, E., Des Portes, V., Hagerman, R., Jacquemont, S., Charles, P., Visootsak, J., Brinkman, M., Rerat, K., Koumaras, B., Zhu, L., et al. (2016) · Science Translational Medicine · doi:10.1126/scitranslmed.aab4109 · Cited by 270 · see citing works
Self-citationIdentification of small molecules rescuing fragile X syndrome phenotypes in Drosophila
Chang, S., Bray, S. M., Li, Z., Zarnescu, D. C., He, C., Jin, P., & Warren, S. T. (2008) · Nature Chemical Biology · doi:10.1038/nchembio.78 · Cited by 254 · see citing works
Henderson, C., Wijetunge, L., Kinoshita, M. N., Shumway, M., Hammond, R. S., Postma, F. R., Brynczka, C., Rush, R., Thomas, A., Paylor, R., et al. (2012) · Science Translational Medicine · doi:10.1126/scitranslmed.3004218 · Cited by 249 · see citing works
Self-citationARNOLD, L. E., VITIELLO, B., MCDOUGLE, C., SCAHILL, L., SHAH, B., GONZALEZ, N. M., CHUANG, S., DAVIES, M., HOLLWAY, J., AMAN, M. G., et al. (2003) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/00004583-200312000-00011 · Cited by 218 · see citing works
Sansone, S. M., Widaman, K. F., Hall, S. S., Reiss, A. L., Lightbody, A. A., Kaufmann, W. E., Berry‐Kravis, E., Lachiewicz, A. M., Brown, E. C., & Hessl, D. R. (2011) · Journal of Autism and Developmental Disorders · doi:10.1007/s10803-011-1370-2 · Cited by 177 · see citing works
Self-citationThe GABAA receptor: a novel target for treatment of fragile X?
D’Hulst, C., & Kooy, R. F. (2007) · Trends in Neurosciences · doi:10.1016/j.tins.2007.06.003 · Cited by 166 · see citing works
Updated report on tools to measure outcomes of clinical trials in fragile X syndrome
Budimirovic, D. B., Berry‐Kravis, E., Erickson, C. A., Hall, S. S., Hessl, D. R., Reiss, A. L., King, M. K., Abbeduto, L., & Kaufmann, W. E. (2017) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-017-9193-x · Cited by 157 · see citing works
Self-citationBerry-Kravis, E., Krause, S. E., Block, S. S., Guter, S., Wuu, J., Leurgans, S., Decle, P., Potanos, K., Cook, E., Salt, J., et al. (2006) · Journal of Child and Adolescent Psychopharmacology · doi:10.1089/cap.2006.16.525 · Cited by 155 · see citing works
Self-citationEfficacy of antidepressants in juvenile depression: meta-analysis
Tsapakis, E.-M., Soldani, F., Tondo, L., & Baldessarini, R. J. (2008) · The British Journal of Psychiatry · doi:10.1192/bjp.bp.106.031088 · Cited by 150 · see citing works
Suppression of LTP-like plasticity in human motor cortex by the GABAB receptor agonist baclofen
McDonnell, M. N., Orekhov, Y., & Ziemann, U. (2007) · Experimental Brain Research · doi:10.1007/s00221-006-0849-0 · Cited by 147 · see citing works
Pacey, L. K. K., Heximer, S. P., & Hampson, D. R. (2009) · Molecular Pharmacology · doi:10.1124/mol.109.056127 · Cited by 133 · see citing works
Hessl, D., Sansone, S. M., Berry-Kravis, E., Riley, K., Widaman, K. F., Abbeduto, L., Schneider, A., Coleman, J., Oaklander, D., Rhodes, K. C., et al. (2016) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-016-9167-4 · Cited by 126 · see citing works
Self-citationThe behavioral phenotype of FMR1 mutations
Boyle, L., & Kaufmann, W. E. (2010) · American Journal of Medical Genetics Part C Seminars in Medical Genetics · doi:10.1002/ajmg.c.30277 · Cited by 118 · see citing works
Self-citationThe FMRP regulon: from targets to disease convergence
Fernández, E., Rajan, N., & Bagni, C. (2013) · Frontiers in Neuroscience · doi:10.3389/fnins.2013.00191 · Cited by 111 · see citing works
Budimirovic, D. B., Bukelis, I., Cox, C., Gray, R. M., Tierney, E., & Kaufmann, W. E. (2006) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.31405 · Cited by 109 · see citing works
Self-citationExploring the Adult Life of Men and Women With Fragile X Syndrome: Results From a National Survey
Hartley, S. L., Seltzer, M. M., Raspa, M., Olmsted, M. G., Bishop, E. E., & Bailey, D. B. (2011) · American Journal on Intellectual and Developmental Disabilities · doi:10.1352/1944-7558-116.1.16 · Cited by 100 · see citing works
Zaidman‐Zait, A., Mirenda, P., Zumbo, B. D., Georgiades, S., Szatmári, P., Bryson, S. E., Fombonne, E. J., Roberts, W., Smith, I. M., Vaillancourt, T., et al. (2011) · Autism Research · doi:10.1002/aur.213 · Cited by 77 · see citing works
Health and Economic Consequences of Fragile X Syndrome for Caregivers
Bailey, D. B., Raspa, M., Bishop, E. E., Mitra, D., Martin, S. A., Wheeler, A., & Sacco, P. (2012) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/dbp.0b013e318272dcbc · Cited by 76 · see citing works
Reliability of Eye Tracking and Pupillometry Measures in Individuals with Fragile X Syndrome
Farzin, F., Huang, F. S., Hervey, C. M., Berry‐Kravis, E., & Hessl, D. R. (2011) · Journal of Autism and Developmental Disorders · doi:10.1007/s10803-011-1176-2 · Cited by 69 · see citing works
Self-citationQin, M., Huang, T., Kader, M., Krych, L., Xia, Z., Burlin, T., Zeidler, Z., Zhao, T., & Smith, C. B. (2015) · International Journal of Neuropsychopharmacology · doi:10.1093/ijnp/pyv034 · Cited by 62 · see citing works
Knox, A. T., Schneider, A., Abucayan, F., Hervey, C. M., Tran, C., Hessl, D. R., & Berry‐Kravis, E. (2012) · Journal of Neurodevelopmental Disorders · doi:10.1186/1866-1955-4-2 · Cited by 58 · see citing works
Self-citationClinic-Based Retrospective Analysis of Psychopharmacology for Behavior in Fragile X Syndrome
Berry‐Kravis, E., Sumis, A. M., Hervey, C., & Mathur, S. (2012) · International Journal of Pediatrics · doi:10.1155/2012/843016 · Cited by 54 · see citing works
Self-citationDevelopment of an Expressive Language Sampling Procedure in Fragile X Syndrome
Berry‐Kravis, E., Doll, E. R., Sterling, A. M., Kover, S. T., Schroeder, S. M., Mathur, S., & Abbeduto, L. (2013) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/dbp.0b013e31828742fc · Cited by 53 · see citing works
Self-citationEfficacy of antidepressants in juvenile depression: meta-analysis
Tsapakis, E. M., Soldani, F., Tondo, L., & Baldessarini, R. J. (2008) · Annals of General Psychiatry · doi:10.1186/1744-859x-7-s1-s350 · Cited by 34 · see citing works
Metabolic and Pharmacokinetic Differentiation of STX209 and Racemic Baclofen in Humans
Sánchez-Ponce, R., Wang, L.-Q., Lu, W., von Hehn, J., Cherubini, M., & Rush, R. (2012) · Metabolites · doi:10.3390/metabo2030596 · Cited by 30 · see citing works
Self-citationRusso‐Ponsaran, N. M., Yesensky, J. A., Hessl, D. R., & Berry‐Kravis, E. (2014) · American Journal on Intellectual and Developmental Disabilities · doi:10.1352/1944-7558-119.1.1 · Cited by 17 · see citing works
Self-citation
Flagged first, then by citations
Research works printed but not linked: 5
- [17]Qin M, Huang T, Kader M, Krych L, Xia Z, Burlin T, Zeidler Z, Zhao T, Smith CB. R-Baclofen Reverses a Social Behavior Deficit and Elevated Protein Synthesis in a Mouse Model of Fragile X Syndrome. Int J Neuropsychopharmacol. 2015;18(9). PMC4576516 25820841
- [21]Sparrow S Cicchetti D Balla D Vineland adaptive behavior scales, second edition (Vineland II). Survey interview form/caregiver rating form 2005 Livonia Pearson Assessments
- [40]Sansone SM Berry-Kravis E Crestodina C McLennan Y Hagerman R Hessl D Sensitivity of an eye tracking paradigm as an outcome measure in a fragile X syndrome targeted treatment clinical trial 49th Annual Gatlinburg conference on research and theory in intellectual and developmental disabilities; San Diego 2016
- [42]De Sonia A Visootsak J Smith M Budimirovic D Tartaglia N Welnik N Analysis of arbaclofen responses in fragile X syndrome 14th International Fragile X Conference; Orange County, CA 2014
- [45]Berry-Kravis E, Erickson CA, Kolevzon A, N T, Hagerman R, Hatti S, Frazier J, Sanders K, Challman T, Needelman H, et al: The treatment of fragile X syndrome with Trofinetide (NNZ-2566). In Proceedings of the 15th international fragile X conference; San Antonio. 2016
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Hagerman, R. J., Berry-Kravis, E., Hazlett, H. C., Bailey, D. B., Moine, H., Kooy, R. F., Tassone, F., Gantois, I., Sonenberg, N., Mandel, J. L., et al. (2017) · Nature Reviews Disease Primers · doi:10.1038/nrdp.2017.65 · Cited by 838 · see citing works
Self-citationDrug development for neurodevelopmental disorders: lessons learned from fragile X syndrome
Berry-Kravis, E. M., Lindemann, L., Jønch, A. E., Apostol, G., Bear, M. F., Carpenter, R. L., Crawley, J. N., Curie, A., Des Portes, V., Hossain, F., et al. (2018) · Nature Reviews Drug Discovery · doi:10.1038/nrd.2017.221 · Cited by 336 · see citing works
Self-citationAn Update on Psychopharmacological Treatment of Autism Spectrum Disorder
Aishworiya, R., Valica, T., Hagerman, R., & Restrepo, B. (2022) · Neurotherapeutics · doi:10.1007/s13311-022-01183-1 · Cited by 233 · see citing works
Self-citationAn update on pharmacotherapy for autism spectrum disorder in children and adolescents
Ji, N. Y., & Findling, R. L. (2015) · Current Opinion in Psychiatry · doi:10.1097/yco.0000000000000132 · Cited by 217 · see citing works
An update on pharmacotherapy of autism spectrum disorder in children and adolescents
Goel, R., Hong, J. S., Findling, R. L., & Ji, N. Y. (2018) · International Review of Psychiatry · doi:10.1080/09540261.2018.1458706 · Cited by 191 · see citing works
Updated report on tools to measure outcomes of clinical trials in fragile X syndrome
Budimirovic, D. B., Berry‐Kravis, E., Erickson, C. A., Hall, S. S., Hessl, D. R., Reiss, A. L., King, M. K., Abbeduto, L., & Kaufmann, W. E. (2017) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-017-9193-x · Cited by 157 · see citing works
Self-citationHessl, D., Sansone, S. M., Berry-Kravis, E., Riley, K., Widaman, K. F., Abbeduto, L., Schneider, A., Coleman, J., Oaklander, D., Rhodes, K. C., et al. (2016) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-016-9167-4 · Cited by 126 · see citing works
Self-citationBerry-Kravis, E. M., Harnett, M. D., Reines, S. A., Reese, M. A., Ethridge, L. E., Outterson, A. H., Michalak, C., Furman, J., & Gurney, M. E. (2021) · Nature Medicine · doi:10.1038/s41591-021-01321-w · Cited by 131 · see citing works
Self-citationGreen, J., & Garg, S. (2018) · Journal of Child Psychology and Psychiatry · doi:10.1111/jcpp.12892 · Cited by 127 · see citing works
Greiss Hess, L., Fitzpatrick, S. E., Nguyen, D. V., Chen, Y., Gaul, K. N., Schneider, A., Lemons Chitwood, K., Eldeeb, M. A. A. A., Polussa, J., Hessl, D., et al. (2016) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/dbp.0000000000000334 · Cited by 122 · see citing works
Self-citationHeussler, H., Cohen, J., Silove, N., Tich, N., Bonn-Miller, M. O., Du, W., O’Neill, C., & Sebree, T. (2019) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-019-9277-x · Cited by 106 · see citing works
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Self-citationTherapeutic Strategies in Fragile X Syndrome: From Bench to Bedside and Back
Gross, C., Hoffmann, A., Bassell, G. J., & Berry-Kravis, E. M. (2015) · Neurotherapeutics · doi:10.1007/s13311-015-0355-9 · Cited by 105 · see citing works
Self-citationStoppel, L. J., Kazdoba, T. M., Schaffler, M. D., Preza, A. R., Heynen, A., Crawley, J. N., & Bear, M. F. (2018) · Neuropsychopharmacology · doi:10.1038/npp.2017.236 · Cited by 92 · see citing works
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Self-citationPlacebo‐controlled crossover assessment of mecasermin for the treatment of Rett syndrome
O’Leary, H. M., Kaufmann, W. E., Barnes, K. V., Rakesh, K., Kapur, K., Tarquinio, D. C., Cantwell, N. G., Roche, K. J., Rose, S. A., Walco, A. C., et al. (2018) · Annals of Clinical and Translational Neurology · doi:10.1002/acn3.533 · Cited by 83 · see citing works
Self-citationLopatina, O. L., Malinovskaya, N. A., Komleva, Y. K., Gorina, Y. V., Shuvaev, A. N., Olovyannikova, R. Y., Belozor, O. S., Belova, O. A., Higashida, H., & Salmina, A. B. (2019) · Reviews in the Neurosciences · doi:10.1515/revneuro-2019-0014 · Cited by 74 · see citing works
Budimirovic, D. B., Schlageter, A. M., Filipovic-Sadic, S., Protic, D. D., Bram, E. E., Mahone, E. M., Nicholson, K., Culp, K., Javanmardi, K., Kemppainen, J., et al. (2020) · Brain Sciences · doi:10.3390/brainsci10100694 · Cited by 80 · see citing works
Self-citationBonnycastle, K., Davenport, E. C., & Cousin, M. A. (2020) · Journal of Neurochemistry · doi:10.1111/jnc.15035 · Cited by 74 · see citing works
Asiminas, A., Jackson, A. D., Louros, S. R., Till, S. M., Spano, T., Dando, O., Bear, M. F., Chattarji, S., Hardingham, G. E., Osterweil, E. K., et al. (2019) · Science Translational Medicine · doi:10.1126/scitranslmed.aao0498 · Cited by 78 · see citing works
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Self-citationNeural circuits regulating prosocial behaviors
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Self-citationFragile X syndrome and fragile X-associated tremor ataxia syndrome
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Self-citationRevisiting the excitation/inhibition imbalance hypothesis of ASD through a clinical lens
Port, R. G., Oberman, L. M., & Roberts, T. P. L. (2019) · British Journal of Radiology · doi:10.1259/bjr.20180944 · Cited by 60 · see citing works
Farmer, C., Swineford, L., Swedo, S. E., & Thurm, A. (2018) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-017-9222-9 · Cited by 69 · see citing works
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Precision Medicine Care in ADHD: The Case for Neural Excitation and Inhibition
Mamiya, P. C., Arnett, A. B., & Stein, M. A. (2021) · Brain Sciences · doi:10.3390/brainsci11010091 · Cited by 63 · see citing works
Díaz‐Caneja, C. M., State, M. W., Hagerman, R. J., Jacquemont, S., Marín, Ó., Bagni, C., Umbricht, D., Simonoff, E. A., Andres-Trelles, F., Kaale, A., et al. (2021) · European Neuropsychopharmacology · doi:10.1016/j.euroneuro.2021.02.020 · Cited by 54 · see citing works
Self-citationLamy, M., & Erickson, C. A. (2018) · Current problems in pediatric and adolescent health care · doi:10.1016/j.cppeds.2018.08.015 · Cited by 58 · see citing works
Berg, A. T., Palac, H., Wilkening, G. N., Zelko, F. A. J., & Schust, L. F. (2020) · Epilepsia · doi:10.1111/epi.16750 · Cited by 55 · see citing works
Neurobiology of ARID1B haploinsufficiency related to neurodevelopmental and psychiatric disorders
Moffat, J. J., Smith, A. L., Jung, E., Ka, M., & Kim, W.-Y. (2021) · Molecular Psychiatry · doi:10.1038/s41380-021-01060-x · Cited by 52 · see citing works
Berg, A. T., Gaebler-Spira, D. J., Wilkening, G. N., Zelko, F. A. J., Knupp, K. G., Dixon‐Salazar, T., Villas, N., Meskis, M. A., Harwell, V., Thompson, T., et al. (2020) · Epilepsy & Behavior · doi:10.1016/j.yebeh.2020.107287 · Cited by 53 · see citing works
Hyperexcitability and homeostasis in fragile X Syndrome.
Liu, X., Kumar, V., Tsai, N.-P., & Auerbach, B. D. (2022) · Frontiers in Molecular Neuroscience · doi:10.3389/fnmol.2021.805929 · Cited by 53 · see citing works
Fragile X syndrome and fragile X-associated disorders
Rajaratnam, A., Shergill, J., Salcedo‐Arellano, M. J., Saldarriaga, W., Duan, X., & Hagerman, R. J. (2017) · F1000Research · doi:10.12688/f1000research.11885.1 · Cited by 52 · see citing works
Self-citationInterneuron Dysfunction and Inhibitory Deficits in Autism and Fragile X Syndrome
Nomura, T. (2021) · Cells · doi:10.3390/cells10102610 · Cited by 48 · see citing works
Bennett (2023) · doi:10.1097/yco.0000000000000775 · Cited by 51 · see citing works
Huang, Q., Pereira, A. C., Velthuis, H., Wong, N. M. L., Ellis, C. L., Ponteduro, F. M., Dimitrov, M., Kowalewski, Ł., Lythgoe, D. J., Rotaru, D., et al. (2022) · Science Translational Medicine · doi:10.1126/scitranslmed.abg7859 · Cited by 45 · see citing works
Novel treatments for autism spectrum disorder based on genomics and systems biology
Baribeau, D. A., & Anagnostou, E. A. (2021) · Pharmacology & Therapeutics · doi:10.1016/j.pharmthera.2021.107939 · Cited by 50 · see citing works
Treatment of Neurogenetic Developmental Conditions: From 2016 into the Future
van Karnebeek, C., Bowden, K., & Berry‐Kravis, E. (2016) · Pediatric Neurology · doi:10.1016/j.pediatrneurol.2016.07.010 · Cited by 43 · see citing works
Self-citationGABAB Receptor Chemistry and Pharmacology: Agonists, Antagonists, and Allosteric Modulators
Nieto, A., Bailey, T., Kaczanowska, K., & McDonald, P. H. (2021) · Current topics in behavioral neurosciences · doi:10.1007/7854_2021_232 · Cited by 43 · see citing works
Keeping the Balance: GABAB Receptors in the Developing Brain and Beyond
Bassetti, D. (2022) · Brain Sciences · doi:10.3390/brainsci12040419 · Cited by 43 · see citing works
Response to Placebo in Fragile X Syndrome Clinical Trials: An Initial Analysis
Luu, S., Province, H. S., Berry‐Kravis, E., Hagerman, R. J., Hessl, D. R., Vaidya, D. M., Lozano, R., Rosselot, H., Erickson, C. A., Kaufmann, W. E., et al. (2020) · Brain Sciences · doi:10.3390/brainsci10090629 · Cited by 40 · see citing works
Self-citationRepurposing available drugs for neurodevelopmental disorders: The fragile X experience
Tranfaglia, M. R., Thibodeaux, C., Mason, D. J., Brown, D., Roberts, I., Smith, R. J., Guilliams, T., & Cogram, P. (2018) · Neuropharmacology · doi:10.1016/j.neuropharm.2018.05.004 · Cited by 39 · see citing works
Hyperexcitability and impaired intracortical inhibition in patients with fragile-X syndrome
Morin‐Parent, F., Champigny, C. M., Lacroix, A., Corbin, F., & Lepage, J. (2019) · Translational Psychiatry · doi:10.1038/s41398-019-0650-z · Cited by 40 · see citing works
Bertelli, M. O., Azeem, M. W., Underwood, L., Scattoni, M. L., Persico, A. M., Ricciardello, A., Sappok, T., Bergmann, T. J., Keller, R., Bianco, A., et al. (2022) · Springer eBooks · doi:10.1007/978-3-319-95720-3_16 · Cited by 41 · see citing works
Review of targeted treatments in fragile X syndrome
Ligsay, A., & Hagerman, R. J. (2016) · Intractable & Rare Diseases Research · doi:10.5582/irdr.2016.01045 · Cited by 37 · see citing works
Self-citationLamy, M., Pedapati, E. V., Dominick, K. C., Wink, L. K., & Erickson, C. A. (2020) · Pediatric Drugs · doi:10.1007/s40272-020-00408-0 · Cited by 36 · see citing works
EEG as a translational biomarker and outcome measure in fragile X syndrome
Kenny, A., Wright, D., & Stanfield, A. C. (2022) · Translational Psychiatry · doi:10.1038/s41398-022-01796-2 · Cited by 33 · see citing works
Molecular Biomarkers in Fragile X Syndrome
Zafarullah, M., & Tassone, F. (2019) · Brain Sciences · doi:10.3390/brainsci9050096 · Cited by 36 · see citing works
Dysregulated Prefrontal Cortex Inhibition in Prepubescent and Adolescent Fragile X Mouse Model
Kramvis, I., van Westen, R., Lammertse, H. C. A., Riga, D., Heistek, T. S., Loebel, A., Spijker, S., Mansvelder, H. D., & Meredith, R. M. (2020) · Frontiers in Molecular Neuroscience · doi:10.3389/fnmol.2020.00088 · Cited by 33 · see citing works
Arnett, A. B., Wang, T., Eichler, E. E., & Bernier, R. (2021) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-021-09371-4 · Cited by 32 · see citing works
Möhrle, D., Wang, W., Whitehead, S. N., & Schmid, S. (2021) · Frontiers in Integrative Neuroscience · doi:10.3389/fnint.2021.710593 · Cited by 31 · see citing works
Auditory hypersensitivity and processing deficits in a rat model of fragile X syndrome
Auerbach, B. D., Manohar, S., Radziwon, K. E., & Salvi, R. (2021) · Neurobiology of Disease · doi:10.1016/j.nbd.2021.105541 · Cited by 32 · see citing works
Baclofen as an adjuvant therapy for autism: a randomized, double-blind, placebo-controlled trial
Mahdavinasab, S.-M., Saghazadeh, A., Motamed-Gorji, N., Vaseghi, S., Mohammadi, M.-R., Alikhani, R., & Akhondzadeh, S. (2019) · European Child & Adolescent Psychiatry · doi:10.1007/s00787-019-01333-5 · Cited by 32 · see citing works
Salcedo‐Arellano, M. J., Cabal-Herrera, A. M., Punatar, R., Clark, C. J., Romney, C. A., & Hagerman, R. J. (2020) · Neurotherapeutics · doi:10.1007/s13311-020-00968-6 · Cited by 30 · see citing works
Self-citationDecades of Progress in the Psychopharmacology of Autism Spectrum Disorder
Henneberry, E. A., Lamy, M., Dominick, K. C., & Erickson, C. A. (2021) · Journal of Autism and Developmental Disorders · doi:10.1007/s10803-021-05237-9 · Cited by 31 · see citing works
Therapeutic strategies for autism: targeting three levels of the central dogma of molecular biology
Hong, D., & Iakoucheva, L. M. (2023) · Translational Psychiatry · doi:10.1038/s41398-023-02356-y · Cited by 31 · see citing works
Modelling fragile X syndrome in the laboratory setting: A behavioral perspective
Melancia, F., & Trezza, V. (2018) · Behavioural Brain Research · doi:10.1016/j.bbr.2018.04.042 · Cited by 29 · see citing works
Bird, L. M., Ochoa‐Lubinoff, C., Tan, W., Heimer, G., Melmed, R. D., Rakhit, A., Visootsak, J., During, M. J., Holcroft, C., Burdine, R. D., et al. (2020) · Neurology · doi:10.1212/wnl.0000000000011409 · Cited by 28 · see citing works
Self-citationGABAergic abnormalities in the fragile X syndrome.
Van der Aa, N., & Kooy, R. F. (2020) · European Journal of Paediatric Neurology · doi:10.1016/j.ejpn.2019.12.022 · Cited by 26 · see citing works
Emerging pharmacological therapies in fragile X syndrome and autism
Yamasue, H., Aran, A. A., & Berry‐Kravis, E. (2019) · Current Opinion in Neurology · doi:10.1097/wco.0000000000000703 · Cited by 27 · see citing works
Self-citationDhaliwal, N. K., Weng, O. Y., Dong, X., Bhattacharya, A., Ahmed, M. E., Nishimura, H., Choi, W. W. Y., Aggarwal, A., Luikart, B. W., Shu, Q., et al. (2024) · Cell Reports · doi:10.1016/j.celrep.2024.114173 · Cited by 27 · see citing works
Hessl, D. R., Harvey, D. J., Sansone, S. M., Crestodina, C., Chin, J. A., Joshi, R., Hagerman, R. J., & Berry‐Kravis, E. (2019) · PLoS ONE · doi:10.1371/journal.pone.0209984 · Cited by 26 · see citing works
Self-citationPhotocontrol of Endogenous Glycine Receptors In Vivo
Gomila, A. M. J., Rustler, K., Maleeva, G., Nin‐Hill, A., Wutz, D., Bautista-Barrufet, A., Rovira, X., Bosch, M., Mukhametova, E., Petukhova, E. O., et al. (2020) · Cell chemical biology · doi:10.1016/j.chembiol.2020.08.005 · Cited by 23 · see citing works
Jiang, S., Xiao, L., Sun, Y., He, M., Gao, C., Zhu, C., Chang, H., Ding, J., Li, W., Wang, Y., et al. (2022) · Molecular Medicine Reports · doi:10.3892/mmr.2022.12670 · Cited by 23 · see citing works
Dysregulated translational control in brain disorders: from genes to behavior
Wiebe, S. A., Nagpal, A., & Sonenberg, N. (2020) · Current Opinion in Genetics & Development · doi:10.1016/j.gde.2020.05.005 · Cited by 24 · see citing works
Molosh, A. I., & Shekhar, A. (2018) · Progress in brain research · doi:10.1016/bs.pbr.2018.09.014 · Cited by 22 · see citing works
An “Omic” Overview of Fragile X Syndrome
Dionne, O., & Corbin, F. (2021) · Biology · doi:10.3390/biology10050433 · Cited by 22 · see citing works
Fragile X syndrome clinical trials: exploring parental decision‐making
D'Amanda, C. S., Peay, H. L., Wheeler, A., Turbitt, E., & Biesecker, B. B. (2019) · Journal of Intellectual Disability Research · doi:10.1111/jir.12605 · Cited by 21 · see citing works
Sato, A., & Ikeda, K. (2021) · Biological Psychiatry Global Open Science · doi:10.1016/j.bpsgos.2021.08.005 · Cited by 22 · see citing works
Gaboxadol in Fragile X Syndrome: A 12-Week Randomized, Double-Blind, Parallel-Group, Phase 2a Study
Budimirovic, D. B., Dominick, K. C., Gabis, L. V., Adams, M., Adera, M., Huang, L., Ventola, P. E., Tartaglia, N. R., & Berry‐Kravis, E. (2021) · Frontiers in Pharmacology · doi:10.3389/fphar.2021.757825 · Cited by 21 · see citing works
Self-citationZeidler, S., de Boer, H., Hukema, R. K., & Willemsen, R. (2017) · Frontiers in Molecular Neuroscience · doi:10.3389/fnmol.2017.00368 · Cited by 21 · see citing works
Sudhahar, S., Özer, B., Chang, J., Chadwick, W., O’Donovan, D., Campbell, A., Tulip, E., Thompson, N. T., & Roberts, I. (2024) · Nature Communications · doi:10.1038/s41467-024-50024-6 · Cited by 20 · see citing works
Deciphering the physiopathology of neurodevelopmental disorders using brain organoids
Dionne, O., Sabatié, S., & Laurent, B. (2024) · Brain · doi:10.1093/brain/awae281 · Cited by 21 · see citing works
mTOR-Dependent Spine Dynamics in Autism
Chaudry, S., & Vasudevan, N. (2022) · Frontiers in Molecular Neuroscience · doi:10.3389/fnmol.2022.877609 · Cited by 22 · see citing works
Heterogeneity in Fragile X Syndrome Highlights the Need for Precision Medicine-Based Treatments
Verdura, E., Pérez‐Cano, L., Sabido-Vera, R., Güney, E., Hyvelin, J.-M., Durham, L. A., & Gomez‐Mancilla, B. (2021) · Frontiers in Psychiatry · doi:10.3389/fpsyt.2021.722378 · Cited by 19 · see citing works
Leone, R., Zuglian, C., Brambilla, R., & Morella, I. (2024) · Frontiers in Pharmacology · doi:10.3389/fphar.2024.1407865 · Cited by 19 · see citing works
Pandina, G., Ring, R. H., Bangerter, A., & Ness, S. L. (2020) · Psychiatric Clinics of North America · doi:10.1016/j.psc.2020.08.003 · Cited by 18 · see citing works
Berry‐Kravis, E. (2022) · Journal of Child Neurology · doi:10.1177/08830738221089740 · Cited by 18 · see citing works
Self-citationBrašić, J. R., Nandi, A., Russell, D., Jennings, D. L., Barret, O., Mathur, A., Slifer, K. J., Sedlak, T. W., Martin, S., Brinson, Z. S., et al. (2020) · Brain Sciences · doi:10.3390/brainsci10120899 · Cited by 18 · see citing works
Self-citationDerieux, C., Léauté, A., Brugoux, A., Jaccaz, D., Terrier, C., Pin, J., Kniazeff, J., Merrer, J. L., & Becker, J. A. J. (2022) · Neuropsychopharmacology · doi:10.1038/s41386-022-01317-1 · Cited by 16 · see citing works
Agmatine relieves behavioral impairments in Fragile X mice model
Jeon, S. J., Kwon, H., Bae, H. J., Gonzales, E. L., Kim, J.-H., Chung, H. J., Kim, D. H., Ryu, J. H., & Shin, C. Y. (2022) · Neuropharmacology · doi:10.1016/j.neuropharm.2022.109234 · Cited by 17 · see citing works
Paradoxical effect of baclofen on social behavior in the fragile X syndrome mouse model
Zeidler, S., Pop, A. S., Jaafar, I. A., de Boer, H., Buijsen, R. A. M., de Esch, C. E. F., Nieuwenhuizen‐Bakker, I., Hukema, R. K., & Willemsen, R. (2018) · Brain and Behavior · doi:10.1002/brb3.991 · Cited by 16 · see citing works
Bruno, J. L., Hosseini, S. M. H., Lightbody, A. A., Manchanda, M. K., & Reiss, A. L. (2019) · Journal of Psychopharmacology · doi:10.1177/0269881119858304 · Cited by 17 · see citing works
Jiang, S., He, M., Xiao, L., Sun, Y., Ding, J., Li, W., Guo, B., Wang, L., Wang, Y., Gao, C., et al. (2022) · Frontiers in Psychiatry · doi:10.3389/fpsyt.2022.835993 · Cited by 17 · see citing works
Neurodevelopment and early pharmacological interventions in Fragile X Syndrome
Milla, L. A., López‐Corral, L., Rivera, J., Zúñiga, N., Pino, G., Nunez-Parra, A., & Rio, C. A. C.-D. (2023) · Frontiers in Neuroscience · doi:10.3389/fnins.2023.1213410 · Cited by 15 · see citing works
New Therapeutic Options for Fragile X Syndrome
Jalnapurkar, I. R., Cochran, D., & Frazier, J. A. (2019) · Current Treatment Options in Neurology · doi:10.1007/s11940-019-0551-8 · Cited by 15 · see citing works
Pandina, G., Ring, R. H., Bangerter, A., & Ness, S. L. (2020) · Child and Adolescent Psychiatric Clinics of North America · doi:10.1016/j.chc.2019.12.004 · Cited by 16 · see citing works
Bassetti, D., Lombardi, A., Kirischuk, S., & Luhmann, H. J. (2020) · Cerebral Cortex · doi:10.1093/cercor/bhaa187 · Cited by 15 · see citing works
Reines, V., Charen, K. H., Rosser, T. C., Eisen, A., Sherman, S. L., & Visootsak, J. (2017) · Journal of Genetic Counseling · doi:10.1007/s10897-017-0111-x · Cited by 15 · see citing works
Self-citationMüller, A. R., van Silfhout, N. Y., den Hollander, B., Kampman, D. H. C., Bakkum, L., Brands, M. M. M. G., Haverman, L., Terwee, C. B., Schuengel, C., Daams, J. G., et al. (2024) · Therapeutic Advances in Rare Disease · doi:10.1177/26330040241245721 · Cited by 13 · see citing works
A novel eye‐tracking paradigm for indexing social avoidance‐related behavior in fragile X syndrome
Klusek, J., Moser, C., Schmidt, J., Abbeduto, L., & Roberts, J. E. (2019) · American Journal of Medical Genetics Part B Neuropsychiatric Genetics · doi:10.1002/ajmg.b.32757 · Cited by 13 · see citing works
Zeid, A. M., Nasr, J. J., Belal, F. F., Walash, M. I., Kaji, N., & Baba, Y. (2020) · Microchemical Journal · doi:10.1016/j.microc.2020.105770 · Cited by 13 · see citing works
An Update on Psychopharmacological Treatment of Autism Spectrum Disorder
Aishworiya, R., Valica, T., Hagerman, R. J., & Restrepo, B. (2024) · FOCUS The Journal of Lifelong Learning in Psychiatry · doi:10.1176/appi.focus.24022006 · Cited by 15 · see citing works
Self-citationCordeiro, L., Braden, M. L., Coan, E., Welnick, N., Tanda, T., & Tartaglia, N. R. (2020) · Brain Sciences · doi:10.3390/brainsci10040248 · Cited by 12 · see citing works
Cordeiro, L., Villagomez, A. N., Swain, D. M., Deklotz, S., & Tartaglia, N. R. (2020) · Brain Sciences · doi:10.3390/brainsci10060351 · Cited by 12 · see citing works
Saraf, T. S., McGlynn, R. P., Bhatavdekar, O., Booth, R. G., & Canal, C. E. (2022) · ACS Chemical Neuroscience · doi:10.1021/acschemneuro.2c00574 · Cited by 12 · see citing works
- Authors
- Berry-Kravis, E., Hagerman, R., Visootsak, J., Budimirovic, D., Kaufmann, W. E., Cherubini, M., Zarevics, P., Walton-Bowen, K., Wang, P., Bear, M. F., & Carpenter, R. L.
- Affiliations
- University of California Davis Medical Center (US)
- Emory University (US)
- Kennedy Krieger Institute (US)
- Boston Children's Hospital (US)
- Seaside Therapeutics (United States) (US)
- Simons Foundation (US)
- Autism Speaks (US)
- Massachusetts Institute of Technology (US)
- Rett Syndrome Research Trust (US)
- Venue
- Journal of Neurodevelopmental Disorders
- Published
- 2017-06-12
- Volume
- 9
- Issue
- 1
- Pages
- 3-3
- ISSN
- 1866-1947 (ISSN-L)
- 1866-1955
- PMC
- PMC5467054
- OpenAlex
- W2576165191
- Cited by
- 183
- References
- 39
- Citations by year
- 2026: 11 · 2025: 16 · 2024: 16 · 2023: 13 · 2022: 22 · 2021: 21 · 2020: 24 · 2019: 23 · 2018: 17 · 2017: 11 · 2016: 6 · 2015: 3
- FWCI
- 13.61
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Licence
- cc-by
- Locations
- Journal of Neurodevelopmental Disorders (publishedVersion, cc-by)
- Europe PMC (PubMed Central) (publishedVersion, other-oa)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion)
- DSpace@MIT (Massachusetts Institute of Technology) (submittedVersion, cc-by-nc)
- eScholarship (California Digital Library) (submittedVersion)
- eScholarship (California Digital Library) (submittedVersion, cc-by)
- Funders
- Seaside Therapeutics, Inc.
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Cognitive Neuroscience › Autism Spectrum Disorder Research
- Life Sciences › Neuroscience › Developmental Neuroscience › Williams Syndrome Research
- Keywords
- fragile X syndrome, social avoidance, irritability, hyperactivity, parental stress, adaptive behavior, adverse events, dose-response relationship, children and adolescents, serotonin reuptake inhibitors, NMDA receptor antagonists, behavioral phenotypes, drug safety and efficacy
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