2024
Huie EZ, Yang X, Rioult-Pedotti MS, Naik M, Huang YA, Silverman JL, Marshall J. Peptidomimetic inhibitors targeting TrkB/PSD-95 signaling improves cognition and seizure outcomes in an Angelman Syndrome mouse model. bioRxiv [Preprint]. 2024 Jun 8:2024.06.07.597833. doi: 10.1101/2024.06.07.597833. PMID: 38895218; PMCID: PMC11185757.
Lin YY, Jbeily EH, Tjandra PM, Pride MC, Lopez-Torres M, Elmankabadi SB, Delman CM, Biris KK, Bang H, Silverman JL, Lee CA, Christiansen BA. Surgical restabilization reduces the progression of post-traumatic osteoarthritis initiated by ACL rupture in mice. Osteoarthritis Cartilage. 2024 Apr 30:S1063-4584(24)01170-1. doi: 10.1016/j.joca.2024.04.013. Epub ahead of print. PMID: 38697509.
Fenton TA, Haouchine OY, Hallam EL, Smith EM, Jackson KC, Rahbarian D, Canales C, Adhikari A, Nord AS, Ben-Shalom R, Silverman JL. Hyperexcitability and translational phenotypes in a preclinical mouse model of SYNGAP1-Related Intellectual Disability. Res Sq [Preprint]. 2024 Mar 19:rs.3.rs-4067746. doi: 10.21203/rs.3.rs-4067746/v1. PMID: 38562838; PMCID: PMC10984035.
2023
Silverman JL. Animal models for psychiatric research: Novel directions for behavioral neuroscience in translation. Neurosci Biobehav Rev. 2023 Sep;152:105309. doi: 10.1016/j.neubiorev.2023.105309. Epub 2023 Jul 7. PMID: 37423590.
Duis J, Skinner A, Carson R, Gouelle A, Annoussamy M, Silverman JL, Apkon S, Servais L, Carollo J. Quantitative measures of motor development in Angelman syndrome. Am J Med Genet A. 2023 Jul;191(7):1711-1721. doi: 10.1002/ajmg.a.63192. Epub 2023 Apr 5. PMID: 37019838; PMCID: PMC11068498.
Anderson LL, Everett-Morgan D, Petkova SP, Silverman JL, Arnold JC. Ictal vocalizations in the Scn1a+/- mouse model of Dravet syndrome. Epilepsia Open. 2023 Sep;8(3):776-784. doi: 10.1002/epi4.12715. Epub 2023 May 15. PMID: 36811143; PMCID: PMC10472354.
O'Geen H, Beitnere U, Garcia MS, Adhikari A, Cameron DL, Fenton TA, Copping NA, Deng P, Lock S, Halmai JANM, Villegas IJ, Liu J, Wang D, Fink KD, Silverman JL, Segal DJ. Transcriptional reprogramming restores UBE3A brain-wide and rescues behavioral phenotypes in an Angelman syndrome mouse model. Mol Ther. 2023 Apr 5;31(4):1088-1105. doi: 10.1016/j.ymthe.2023.01.013. Epub 2023 Jan 14. PMID: 36641623; PMCID: PMC10124086.
Beitnere U, Vilanova-Cuevas B, Christian SG, Taylor C, Berg EL, Copping NA, Dindot SV, Silverman JL, Gareau MG, Segal DJ. Unique Features of the Gut Microbiome Characterized in Animal Models of Angelman Syndrome. mSystems. 2023 Feb 23;8(1):e0060822. doi: 10.1128/msystems.00608-22. Epub 2023 Jan 4. PMID: 36598241; PMCID: PMC9948700.
2022
Adhikari A, Buchanan FKB, Fenton TA, Cameron DL, Halmai JANM, Copping NA, Fink KD, Silverman JL. Touchscreen cognitive deficits, hyperexcitability and hyperactivity in males and females using two models of Cdkl5 deficiency. Hum Mol Genet. 2022 Sep 10;31(18):3032-3050. doi: 10.1093/hmg/ddac091. PMID: 35445702; PMCID: PMC9476626.
Silverman JL, Thurm A, Ethridge S, Soller MM, Petkova SP, Abel T, Bauman MD, Brodkin ES, Harony-Nicolas H, Clayton AL, Wöhr M, Halladay AK (2022). Reconsidering Animal Models for Autism Spectrum Disorder: Current State and Optimizing Future. Genes Brain Behavior. Special Issue on Autism and Neurodevelopmental Disorders. Workshop on Animal Models Report.
Petkova SP, Adhikari A, Berg EL, Fenton TA, Duis J, Silverman JL. Gait as a quantitative translational outcome measure in Angelman syndrome. Autism Res. 2022 May;15(5):821-833. doi: 10.1002/aur.2697. Epub 2022 Mar 10. PMID: 35274462; PMCID: PMC9311146.
Bein KJ, Wallis CD, Silverman JL, Lein PJ, Wexler AS. Emulating Near-Roadway Exposure to Traffic-Related Air Pollution via Real-Time Emissions from a Major Freeway Tunnel System. Environ Sci Technol. 2022 Mar 2:10.1021/acs.est.1c07047. doi: 10.1021/acs.est.1c07047. Epub ahead of print. PMID: 35235290; PMCID: PMC9437142.
Deng P, Halmai JANM, Beitnere U, Cameron D, Martinez ML, Lee CC, Waldo JJ, Thongphanh K, Adhikari A, Copping NA, Petkova SP, Lee RD, Lock S, Palomares M, O’Geen H, Carter J, Gonzalez CE, Buchanan FKB, Anderson JD, Fierro FA, Nolta JA, Tarantal AF, Silverman JL, Segal DJ, Fink KD (2022) An In vivo Cell-Based Delivery Platform for Zinc Finger Artificial Transcription Factors in Preclinical Animal Models Front Molecular Neurosci DOI: 10.3389/fnmol.2021.789913.
2021
Sethi S, Keil Stietz KP, Valenzuela AE, Klocke CR, Silverman JL, Puschner B, Pessah IN, Lein PJ (2021) Developmental Exposure to a Human-Relevant Polychlorinated Biphenyl Mixture Causes Behavioral Phenotypes That Vary by Sex and Genotype in Juvenile Mice Expressing Human Mutations That Modulate Neuronal Calcium. Front Neurosci Dec 6;15:766826. doi: 10.3389/fnins.
Sethi S, Keil Stietz KP, Valenzuela AE, Klocke CR, Silverman JL, Puschner B, Pessah IN, Lein PJ. (2021) Developmental Exposure to a Human-Relevant Polychlorinated Biphenyl Mixture Causes Behavioral Phenotypes That Vary by Sex and Genotype in Juvenile Mice Expressing Human Mutations That Modulate Neuronal Calcium. Frontiers in Neuroscience. Dec 6;15:766826. doi: 10.3389/fnins.2021.766826. PMID: 34938155
YouJin Lee, Morgan R. Miller, Marty A. Fernandez, Elizabeth L. Berg, Adriana M. Prada, Qing Ouyang, Michael Schmidt, Jill L. Silverman, Tracy L. Young-Pearse, and Eric M. Morrow. Early lysosome defects precede neurodegeneration with Aβ and tau aggregation in NHE6-null rat brain. Brain (2021).
Nycole A. Copping, Stephanie M. McTighe, Kyle D. Fink, and Jill L. Silverman. Emerging Gene and Small Molecule Therapies for the Neurodevelopmental Disorder Angelman Syndrome. Neurotherapeutics (2021). https://doi.org/10.1007/s13311-021-01082-x.
Berg EL*, Petkova SP*, Born HA, Adhikari A, Anderson AE, and Silverman JL. Insulin-like growth factor-2 does not improve behavioral deficits in Ube3a deletion mouse and rat models of Angelman Syndrome. Molecular Autism 12, 59 (2021). https://doi.org/10.1186/s13229-021-00467-1.
Berg EL, Shekib AJ, Petkova SP, Ellegood J, Fenton TA, Segal DJ, Gray JA, Wöhr M, and Silverman JL (2021) Elevated 50-kHz affiliative vocalizations and long-term potentiation deficits in the Ube3a deletion rat model of Angelman Syndrome. J Neuroscience. Sep 2:JN-RM-0925-21. doi: 10.1523/JNEUROSCI.0925-21.2021. Online ahead of print.PMID: 34475199
Berg EL, Jami SA, Petkova SP, Berz A, Fenton TA, Lerch JP, Segal DJ, Gray JA, Ellegood J, Wöhr M, Silverman JL. Excessive laughter-like vocalizations, microcephaly, and translational outcomes in the Ube3a deletion rat model of Angelman Syndrome. J Neurosci. 2021 Sep 2:JN-RM-0925-21. doi: 10.1523/JNEUROSCI.0925-21.2021. Online ahead of print. PMID: 34475199.
Berg EL and Silverman JL. Measuring social communication in rodent models of neurodevelopmental disorders. Encyclopedia of Behavioural Neuroscience 2e, edited by Sergio Della Sala. https://www.elsevier.com/books/encyclopedia-of-behavioral-neuroscience-2nd-edition/9780128216361. https://doi.org/10.1016/B978-0-12-819641-0.00002-5.
Eduardo A. González, Jonas J. Calsbeek, Yi-Hua Tsai, Mei-Yun Tang, Peter Andrew, Joan Vu, Elizabeth L. Berg, Naomi H. Saito, Danielle J. Harvey, Suangsuda Supasai, Gene G. Gurkoff, Jill L. Silverman, and Pamela J. Lein. Sex-specific acute and chronic neurotoxicity of acute diisopropylfluorophosphate (DFP)-intoxication in juvenile Sprague-Dawley rats. Current Research in Toxicology, 2(2021), 341-356.
Jonas J. Calsbeek, Eduardo A. González, Donald A. Bruun, Michelle A. Guignet, Nycole Copping, Mallory E. Dawson, Alexandria J. Yu, Jeremy A. MacMahon, Naomi H. Saito, Danielle J. Harvey, Jill L. Silverman, Pamela J. Lein. Persistent neuropathology and behavioral deficits in a mouse model of status epilepticus induced by acute intoxication with diisopropylfluorophosphate. NeuroToxicology. Online ahead of print. https://doi.org/10.1016/j.neuro.2021.09.001.
Anna Adhikari, Nycole A. Copping, Julie Beegle, David L. Cameron, Peter Deng, Henriette O’Geen, David J. Segal, Kyle D. Fink, Jill L. Silverman*, Joseph S. Anderson *. Functional rescue in an Angelman syndrome model following treatment with lentivector transduced hematopoietic stem cells. Human Molecular Genetics 2021 Jun 9;30(12):1067-1083. doi: 10.1093/hmg/ddab104.
Jessica L. Haigh*, Anna Adhikari*, Nycole A. Copping, Tyler Stradleigh, A. Ayanna Wade, Rinaldo Catta-Preta, Linda Su-Feher, Iva Zdilar, Sarah Morse, Timothy A Fenton, Anh Nguyen, Diana Quintero, Samrawit Agezew, Michael Sramek, Ellie J Kreun, Jasmine Carter, Andrea Gompers, Jason Lambert, Cesar P. Canales, Len A. Pennacchio, Axel Visel, Diane E. Dickel, Jill L. Silverman#, Alex S. Nord#. Deletion of a non-canonical regulatory sequence causes loss of Scn1a expression and epileptic phenotypes in mice. Genome Medicine. 13, 69 (2021). https://doi.org/10.1186/s13073-021-00884-0.
Patten KT, Valenzuela AE, Wallis C, Berg EL, Silverman JL, Bein KJ, Wexler AS, Lein PJ. The effects of chronic exposure to ambient traffic-related air pollution on Alzheimer’s Disease phenotypes in wildtype and genetically predisposed male and female rats. Environmental Health Perspectives. 2021 May 10;129(5):057005.
Copping, N.A., Silverman, J.L. Abnormal electrophysiological phenotypes and sleep deficits in a mouse model of Angelman Syndrome. Molecular Autism 12, 9 (2021). https://doi.org/10.1186/s13229-021-00416-y.
Ellegood J, Petkova SP, Kinman A, Qiu LR, Adhikari A, Wade AA, Fernandes D, Lindenmaier Z, Creighton A, Nutter LMJ, Nord AS, Silverman JL, Lerch JP. Neuroanatomy and behavior in mice with a haploinsufficiency of AT-rich interactive domain 1B (ARID1B) throughout development. Mol Autism. 2021 Mar 23;12(1):25. doi: 10.1186/s13229-021-00432-y. PMID: 33757588; PMCID: PMC7986278.
Born HA, Martinez LA, Levine AT, Harris SE, Mehra S, Lee WL, Dindot SV, Nash KR, Silverman JL, Segal DJ, Weeber EJ. Early developmental EEG and seizure phenotypes in a full gene deletion of ubiquitin protein ligase E3A rat model of Angelman syndrome. eNeuro. 2021 Mar;8(2).
Accepted papers to be published online soon:
Petkova SP, Berg EL, Copping NA, and Silverman JL. Animal models. Textbook of Autism Spectrum Disorders, Second Edition, edited by Eric Hollander, Randi J. Hagerman, and Casara J. Ferretti, APA Publishing, in press.
2020
Kelly E, Meng F, Fujita H, Morgado F, Kazemi Y, Rice LC, Ren C, Escamilla CO, Gibson JM, Sajadi S, Pendry RJ. Regulation of autism-relevant behaviors by cerebellar–prefrontal cortical circuits. Nature Neuroscience. 2020 Sep;23(9):1102-10.
Silverman JL, Nithianantharajah J, Der-Avakian A, Young JW, Rizzo SS. Lost in translation: At the crossroads of face validity and translational utility of behavioral assays in animal models for the development of therapeutics. Neuroscience and Biobehavioral Reviews. 2020 Sep;116:452.
Dodge A, Peters MM, Greene HE, Dietrick C, Botelho R, Chung D, Willman J, Nenninger AW, Ciarlone S, Kamath SG, Houdek P. Generation of a novel rat model of Angelman Syndrome with a complete Ube3a gene deletion. Autism Research. 2020 Mar;13(3):397-409.
Berg EL, Pride MC, Petkova PS, Lee R, Copping, NA, Shen Y, Adhikari A, Fenton T, Pederson L, Noakes, LS, Nieman B, Lerch JP, Harris S, Born H, Peters M, Deng P, Cameron D, Fink KD, Beitnere U, O’ Geen H, Anderson AE, Dindot SV, Nash, K, Weeber E, Wohr M, Ellegood J, Segal DJ and Silverman JL., Translational outcomes in a full gene deletion of ubiquitin protein ligase E3A rat model of Angelman Syndrome. Translational Psychiatry. 2020 Jan 27;10(1):39. doi: 10.1038/s41398-020-0720-2.
Patten, K.T., González, E.A., Valenzuela, A. et al. Effects of early life exposure to traffic-related air pollution on brain development in juvenile Sprague-Dawley rats. Translational Psychiatry 10, 166 (2020). https://doi.org/10.1038/s41398-020-0845-3
Petkova SP, Pride M, Klocke C, Fenton TA, White J, Lein PJ, Ellegood J, Lerch JP, Silverman JL, Waldau B. Cyclin D2-knock-out mice with attenuated dentate gyrus neurogenesis have robust deficits in long-term memory formation. Sci Rep. 2020 May 18;10(1):8204. doi: 10.1038/s41598-020-65090-1. PMID: 32424171; PMCID: PMC7235216.
Berg EL, Pedersen LR, Pride MC, Petkova SP, Patten KT, Valenzuela AE, Wallis C, Bein KJ, Wexler A, Lein PJ, Silverman JL. Developmental exposure to near roadway pollution produces behavioral phenotypes relevant to neurodevelopmental disorders in juvenile rats. Translational Psychiatry. 2020 Aug 17;10(1):289. doi: 10.1038/s41398-020-00978-0. PMID: 32807767; PMCID: PMC7431542.
Berg, E.L., Ching, T.M., Bruun, D.A. et al. Translational outcomes relevant to neurodevelopmental disorders following early life exposure of rats to chlorpyrifos. J Neurodevelop Disord 12, 40 (2020). https://doi.org/10.1186/s11689-020-09342-1
2019
Copping NA, Adhikari A, Petkova SP, Silverman JL. Genetic backgrounds have unique seizure response profiles and behavioral outcomes following convulsant administration. Epilepsy Behav. 2019 Dec; 101(Pt A):106547. doi: 10.1016/j.yebeh.2019.106547. Epub 2019 Nov 4. PMID: 31698263
Lopez SJ, Laufer BI, Beitnere U, Berg EL, Silverman JL, O'Geen H, Segal DJ, LaSalle JM. Imprinting effects of UBE3A loss on synaptic gene networks and Wnt signaling pathways. Hum Mol Genet. 2019 Nov 15;28(22):3842-3852. doi: 10.1093/hmg/ddz221. PMID: 31625566
Keil KP, Sethi S, Wilson MD, Silverman JL, Pessah IN, Lein PJ. Genetic mutations in Ca2+ signaling alter dendrite morphology and social approach in juvenile mice. Genes Brain Behav. 2019 Jan; 18(1):e12526. doi: 10.1111/gbb.12526. Epub 2018 Nov 6. PMID: 30311737
Jessica L. Haigh, Anna Adhikari, Nycole A. Copping, Tyler Stradleigh, A. Ayanna Wade, Rinaldo Catta-Preta, Linda Su-Feher, Iva Zdilar, Sarah Morse, Timothy A Fenton, Anh Nguyen, Diana Quintero, Michael Sramek, Jasmine Carter, Andrea Gompers, Jason Lambert, Cesar P. Canales, Len A. Pennacchio, Axel Visel, Diane E. Dickel, Jill L. Silverman, Alex S. Nord. Deletion of a non-canonical promoter regulatory element causes loss of Scn1a expression and epileptic phenotypes in mice. bioRxiv 766634; doi: https://doi.org/10.1101/766634
2018
Adhikari, A., Copping N.A., Onaga B., Pride M.C., Coulson R.L., Yang M., Yasui D.H., LaSalle J.M., Silverman J.L., Cognitive deficits in the Snord116 deletion mouse model for Prader-Willi syndrome. Neurobiology of Learning and Memory. 2018 May 23. pii: S1074-7427(18)30119-9 2018. pii: S1074-7427(18)30119-9.
Silverman, J.L., Ellegood, J., Behavioral and neuroanatomical approaches in models of neurodevelopmental disorders: opportunities for translation. Current Opinions in Neurology. 2018 Apr;31(2):126-133.
Berg E.L., Copping N.A., Rivera J.K., Pride M.C., Careaga M., Bauman M.D., Berman R.F., Lein P.J., Harony-Nicolas H., Buxbaum J.D., Ellegood J., Lerch J.P., Wöhr M., Silverman J.L., Developmental social communication deficits in the Shank3 rat model of phelan-mcdermid syndrome and autism spectrum disorder. Autism Research. 2018 Apr;11(4):587-601.
Laman-Maharg A, Williams AV, Zufelt MD, Minie VA, Ramos-Maciel S, Hao R, Ordoñes Sanchez E, Copeland T, Silverman JL, Leigh A, Snyder R. Sex differences in the effects of a kappa opioid receptor antagonist in the forced swim test. Frontiers in pharmacology. 2018 Feb 14;9:93.
Matt L, Kirk LM, Chenaux G, Speca DJ, Puhger KR, Pride MC, Qneibi M, Haham T, Plambeck KE, Stern-Bach Y, Silverman JL. SynDIG4/Prrt1 is required for excitatory synapse development and plasticity underlying cognitive function. Cell reports. 2018 Feb 27;22(9):2246-53.
Kelly E, Schaeffer SM, Dhamne SC, Lipton JO, Lindemann L, Honer M, Jaeschke G, Super CE, Lammers SH, Modi ME, Silverman JL. mGluR5 modulation of behavioral and epileptic phenotypes in a mouse model of tuberous sclerosis complex. Neuropsychopharmacology. 2018 May;43(6):1457-65.
Velíšková, Jana, Jill L. Silverman, Melissa Benson, and Pierre-Pascal Lenck-Santini. "Autistic traits in epilepsy models: Why, when and how?." Epilepsy research 144 (2018): 62-70.
2017
Copping N.A., Christian S.G.B., Ritter D.J., Islam M.S., Buscher N., Zolkowska D., Pride M.C., Berg E.L., LaSalle J.M., Ellegood J., Lerch J.P., Reiter L.T., Silverman J.L., Dindot S.V., Neuronal overexpression of Ube3a isoform 2 causes behavioral impairments and neuroanatomical pathology relevant to 15q11.2-q13.3 duplication syndrome. Human Molecular Genetics. 2017 Oct 15;26(20):3995-4010.
Gompers A.L., Su-Feher L., Ellegood J., Copping N.A., Riyadh M.A., Stradleigh T.W., Pride M.C., Schaffler M.D., Wade A.A., Catta-Preta R., Zdilar I., Louis S., Kaushik G., Mannion B.J., Plajzer-Frick I., Afzal V., Visel A., Pennacchio L.A., Dickel D.E., Lerch J.P., Crawley J.N., Zarbalis K.S., Silverman J.L., Nord A.S., Germline Chd8 haploinsufficiency alters brain development in mouse. Nature Neuroscience. 2017 Aug;20(8):1062-1073.
Dhamne S.C., Silverman J.L., Super C.E., Lammers S.H.T., Hameed M.Q., Modi M.E., Copping N.A., Pride M.C., Smith D.G., Rotenberg A., Crawley J.N., Sahin M., Replicable in vivo physiological and behavioral phenotypes of the Shank3B null mutant mouse model of autism. Molecular Autism. 2017 Jun 15;8:26.
Copping, N.A., Berg, E.L., Foley, G.M., Schaffler, M.D., Onaga, B.L., Buscher, N., Silverman, J.L., Yang, M., Touchscreen learning deficits and normal social approach behavior in the Shank3B model of Phelan-McDermid Syndrome and autism. Neuroscience. 2017 Mar 14;345:155-165.
Earlier
Sukoff Rizzo S.J., Silverman J.L., Methodological Considerations for Optimizing and Validating Behavioral Assays. Current Protocols in Mouse Biology. 2016 Dec 1;6(4):364-379.
Silverman, J.L., Pride, M.C., Hayes, J.E., Puhger, K.R., Butler-Struben, H.M., Baker, S., Crawley, J.N., GABAB Receptor Agonist R-Baclofen Reverses Social Deficits and Reduces Repetitive Behavior in Two Mouse Models of Autism. Neuropsychopharmacology. 2015 Aug;40(9):2228-39.
Bales, K.L., Solomon, M., Jacob, S., Crawley, J.N., Silverman, J.L., Larke, R.H., Sahagun, E., Puhger, K.R., Pride, M.C., Mendoza, S.P., Long-Term Exposure to Intranasal Oxytocin in a mouse autism model. Translational Psychiatry. 2014.
Silverman, J. L., Smith, D. G., Rizzo, S. J. S., Karras, M. N., Turner, S. M., Tolu, S. S., ... & Crawley, J. N. (2012). Negative allosteric modulation of the mGluR5 receptor reduces repetitive behaviors and rescues social deficits in mouse models of autism. Science translational medicine, 4(131), 131ra51-131ra51.
Silverman, J. L., Yang, M., Lord, C., & Crawley, J. N. (2010). Behavioural phenotyping assays for mouse models of autism. Nature Reviews Neuroscience, 11(7), 490-502.