[{"month":"02","year":"2022","doi":"10.21203/rs.3.rs-1316167/v1","date_published":"2022-02-16T00:00:00Z","title":"WDFY3 cell autonomously controls neuronal migration","external_id":{"pmid":["PPR454733"]},"publisher":"Research Square","language":[{"iso":"eng"}],"tmp":{"image":"/images/cc_by.png","legal_code_url":"https://creativecommons.org/licenses/by/4.0/legalcode","name":"Creative Commons Attribution 4.0 International Public License (CC-BY 4.0)","short":"CC BY (4.0)"},"main_file_link":[{"url":"https://doi.org/10.21203/rs.3.rs-1316167/v1","open_access":"1"}],"department":[{"_id":"SiHi"}],"date_created":"2022-02-25T07:53:26Z","day":"16","citation":{"ista":"Schaaf Z, Tat L, Cannizzaro N, Green R, Rülicke T, Hippenmeyer S, Zarbalis K. WDFY3 cell autonomously controls neuronal migration. <a href=\"https://doi.org/10.21203/rs.3.rs-1316167/v1\">10.21203/rs.3.rs-1316167/v1</a>.","short":"Z. Schaaf, L. Tat, N. Cannizzaro, R. Green, T. Rülicke, S. Hippenmeyer, K. Zarbalis, (n.d.).","ama":"Schaaf Z, Tat L, Cannizzaro N, et al. WDFY3 cell autonomously controls neuronal migration. doi:<a href=\"https://doi.org/10.21203/rs.3.rs-1316167/v1\">10.21203/rs.3.rs-1316167/v1</a>","mla":"Schaaf, Zachary, et al. <i>WDFY3 Cell Autonomously Controls Neuronal Migration</i>. Research Square, doi:<a href=\"https://doi.org/10.21203/rs.3.rs-1316167/v1\">10.21203/rs.3.rs-1316167/v1</a>.","chicago":"Schaaf, Zachary, Lyvin Tat, Noemi Cannizzaro, Ralph Green, Thomas Rülicke, Simon Hippenmeyer, and K Zarbalis. “WDFY3 Cell Autonomously Controls Neuronal Migration.” Research Square, n.d. <a href=\"https://doi.org/10.21203/rs.3.rs-1316167/v1\">https://doi.org/10.21203/rs.3.rs-1316167/v1</a>.","apa":"Schaaf, Z., Tat, L., Cannizzaro, N., Green, R., Rülicke, T., Hippenmeyer, S., &#38; Zarbalis, K. (n.d.). WDFY3 cell autonomously controls neuronal migration. Research Square. <a href=\"https://doi.org/10.21203/rs.3.rs-1316167/v1\">https://doi.org/10.21203/rs.3.rs-1316167/v1</a>","ieee":"Z. Schaaf <i>et al.</i>, “WDFY3 cell autonomously controls neuronal migration.” Research Square."},"publication_status":"submitted","type":"preprint","abstract":[{"lang":"eng","text":"Background\r\nProper cerebral cortical development depends on the tightly orchestrated migration of newly born neurons from the inner ventricular and subventricular zones to the outer cortical plate. Any disturbance in this process during prenatal stages may lead to neuronal migration disorders (NMDs), which can vary in extent from focal to global. Furthermore, NMDs show a substantial comorbidity with other neurodevelopmental disorders, notably autism spectrum disorders (ASDs). Our previous work demonstrated focal neuronal migration defects in mice carrying loss-of-function alleles of the recognized autism risk gene WDFY3. However, the cellular origins of these defects in Wdfy3 mutant mice remain elusive and uncovering it will provide critical insight into WDFY3-dependent disease pathology .\r\nMethods\r\nHere, in an effort to untangle the origins of NMDs in Wdfy3lacZ mice, we employed mosaic analysis with double markers (MADM). MADM technology enabled us to genetically distinctly track and phenotypically analyze mutant and wild type cells concomitantly in vivo using immunofluorescent techniques.\r\nResults\r\nWe revealed a cell autonomous requirement of WDFY3 for accurate laminar positioning of cortical projection neurons and elimination of mispositioned cells during early postnatal life. In addition, we identified significant deviations in dendritic arborization, as well as synaptic density and morphology between wild type, heterozygous, and homozygous Wdfy3 mutant neurons in Wdfy3-MADM reporter mice at postnatal stages. Limitations While Wdfy3 mutant mice have provided valuable insight into prenatal aspects of ASD pathology that remain inaccessible to investigation in humans, like most animal models, they do not a perfectly replicate all aspects of human ASD biology. The lack of human data makes it indeterminate whether morphological deviations described here apply to ASD patients.\r\nConclusions\r\nOur genetic approach revealed several cell autonomous requirements of Wdfy3 in neuronal development that could underly the pathogenic mechanisms of WDFY3-related ASD conditions. The results are also consistent with findings in other ASD animal models and patients and suggest an important role for Wdfy3 in regulating neuronal function and interconnectivity in postnatal life."}],"author":[{"last_name":"Schaaf","full_name":"Schaaf, Zachary","first_name":"Zachary"},{"last_name":"Tat","full_name":"Tat, Lyvin","first_name":"Lyvin"},{"first_name":"Noemi","full_name":"Cannizzaro, Noemi","last_name":"Cannizzaro"},{"first_name":"Ralph","last_name":"Green","full_name":"Green, Ralph"},{"first_name":"Thomas","last_name":"Rülicke","full_name":"Rülicke, Thomas"},{"orcid":"0000-0003-2279-1061","last_name":"Hippenmeyer","full_name":"Hippenmeyer, Simon","first_name":"Simon","id":"37B36620-F248-11E8-B48F-1D18A9856A87"},{"first_name":"K","last_name":"Zarbalis","full_name":"Zarbalis, K"}],"status":"public","article_processing_charge":"No","page":"30","oa":1,"date_updated":"2023-10-17T13:06:52Z","publication_identifier":{"eissn":["2693-5015"]},"_id":"10792","pmid":1,"oa_version":"Preprint","user_id":"2DF688A6-F248-11E8-B48F-1D18A9856A87"},{"publication":"Research Square","page":"21","file_date_updated":"2021-08-31T14:02:19Z","day":"18","type":"preprint","status":"public","has_accepted_license":"1","department":[{"_id":"StFr"}],"file":[{"content_type":"application/pdf","relation":"main_file","file_id":"9979","creator":"cchlebak","success":1,"date_updated":"2021-08-31T14:02:19Z","access_level":"open_access","file_name":"2021_ResearchSquare_Cao.pdf","file_size":1019662,"date_created":"2021-08-31T14:02:19Z","checksum":"1878e91c29d5769ed5a827b0b7addf00"}],"date_created":"2021-08-31T12:54:16Z","month":"08","date_published":"2021-08-18T00:00:00Z","publisher":"Research Square","language":[{"iso":"eng"}],"article_processing_charge":"No","oa":1,"date_updated":"2023-10-17T13:06:29Z","publication_identifier":{"eissn":["2693-5015"]},"_id":"9978","oa_version":"Preprint","acknowledgement":"This work was financially supported by the National Natural Science Foundation of China (51773092, 21975124, 11874254, 51802187, U2030206). S.A.F. is indebted to IST Austria for support. ","user_id":"2DF688A6-F248-11E8-B48F-1D18A9856A87","citation":{"short":"D. Cao, X. Shen, A. Wang, F. Yu, Y. Wu, S. Shi, S.A. Freunberger, Y. Chen, Research Square (n.d.).","ista":"Cao D, Shen X, Wang A, Yu F, Wu Y, Shi S, Freunberger SA, Chen Y. Sharp kinetic acceleration potentials during mediated redox catalysis of insulators. Research Square, <a href=\"https://doi.org/10.21203/rs.3.rs-750965/v1\">10.21203/rs.3.rs-750965/v1</a>.","ama":"Cao D, Shen X, Wang A, et al. Sharp kinetic acceleration potentials during mediated redox catalysis of insulators. <i>Research Square</i>. doi:<a href=\"https://doi.org/10.21203/rs.3.rs-750965/v1\">10.21203/rs.3.rs-750965/v1</a>","mla":"Cao, Deqing, et al. “Sharp Kinetic Acceleration Potentials during Mediated Redox Catalysis of Insulators.” <i>Research Square</i>, Research Square, doi:<a href=\"https://doi.org/10.21203/rs.3.rs-750965/v1\">10.21203/rs.3.rs-750965/v1</a>.","chicago":"Cao, Deqing, Xiaoxiao Shen, Aiping Wang, Fengjiao Yu, Yuping Wu, Siqi Shi, Stefan Alexander Freunberger, and Yuhui Chen. “Sharp Kinetic Acceleration Potentials during Mediated Redox Catalysis of Insulators.” <i>Research Square</i>. Research Square, n.d. <a href=\"https://doi.org/10.21203/rs.3.rs-750965/v1\">https://doi.org/10.21203/rs.3.rs-750965/v1</a>.","apa":"Cao, D., Shen, X., Wang, A., Yu, F., Wu, Y., Shi, S., … Chen, Y. (n.d.). Sharp kinetic acceleration potentials during mediated redox catalysis of insulators. <i>Research Square</i>. Research Square. <a href=\"https://doi.org/10.21203/rs.3.rs-750965/v1\">https://doi.org/10.21203/rs.3.rs-750965/v1</a>","ieee":"D. Cao <i>et al.</i>, “Sharp kinetic acceleration potentials during mediated redox catalysis of insulators,” <i>Research Square</i>. Research Square."},"publication_status":"submitted","abstract":[{"text":"Redox mediators could catalyse otherwise slow and energy-inefficient cycling of Li-S and Li-O 2 batteries by shuttling electrons/holes between the electrode and the solid insulating storage materials. For mediators to work efficiently they need to oxidize the solid with fast kinetics yet the lowest possible overpotential. Here, we found that when the redox potentials of mediators are tuned via, e.g., Li + concentration in the electrolyte, they exhibit distinct threshold potentials, where the kinetics accelerate several-fold within a range as small as 10 mV. This phenomenon is independent of types of mediators and electrolyte. The acceleration originates from the overpotentials required to activate fast Li + /e – extraction and the following chemical step at specific abundant surface facets. Efficient redox catalysis at insulating solids requires therefore carefully considering the surface conditions of the storage materials and electrolyte-dependent redox potentials, which may be tuned by salt concentrations or solvents.","lang":"eng"}],"author":[{"last_name":"Cao","full_name":"Cao, Deqing","first_name":"Deqing"},{"full_name":"Shen, Xiaoxiao","last_name":"Shen","first_name":"Xiaoxiao"},{"first_name":"Aiping","full_name":"Wang, Aiping","last_name":"Wang"},{"first_name":"Fengjiao","last_name":"Yu","full_name":"Yu, Fengjiao"},{"first_name":"Yuping","last_name":"Wu","full_name":"Wu, Yuping"},{"full_name":"Shi, Siqi","last_name":"Shi","first_name":"Siqi"},{"id":"A8CA28E6-CE23-11E9-AD2D-EC27E6697425","orcid":"0000-0003-2902-5319","last_name":"Freunberger","full_name":"Freunberger, Stefan Alexander","first_name":"Stefan Alexander"},{"last_name":"Chen","full_name":"Chen, Yuhui","first_name":"Yuhui"}],"keyword":["Catalysis","Energy engineering","Materials theory and modeling"],"tmp":{"image":"/images/cc_by.png","legal_code_url":"https://creativecommons.org/licenses/by/4.0/legalcode","name":"Creative Commons Attribution 4.0 International Public License (CC-BY 4.0)","short":"CC BY (4.0)"},"related_material":{"record":[{"relation":"later_version","id":"10813","status":"public"}]},"ddc":["541"],"year":"2021","doi":"10.21203/rs.3.rs-750965/v1","title":"Sharp kinetic acceleration potentials during mediated redox catalysis of insulators"}]