Publications

2022 Loganathan S, Wilson BA, Carey SB, Manzo E, Joardar A, Ugur B, Zarnescu DC. TDP-43 Proteinopathy Causes Broad Metabolic Alterations including TCA Cycle Intermediates and Dopamine Levels in Drosophila Models of ALS. Metabolites. 2022 Jan 21;12(2):101. DOI: 10.3390/metabo12020101. PMID: 35208176; PMCID: PMC8876928.

2021

Loganathan, S., Ball, H. E., Manzo, E., Zarnescu, D. C. Measuring Glucose Uptake in Drosophila Models of TDP-43 Proteinopathy. J. Vis. Exp. (174), e62936, DOI:10.3791/62936

2021

Lehmkuhl, E. M., Loganathan, S., Alsop, E., Blythe, A. D., Kovalik, T., Mortimore, N. P., Barrameda, D., Kueth, C., Eck, R. J., Siddegowda, B. B., Joardar, A., Ball, H., Macias, M. E., Bowser, R., Van Keuren-Jensen, K., & Zarnescu, D. C. (2021). TDP-43 proteinopathy alters the ribosome association of multiple mRNAs including the glypican Dally-like protein (Dlp)/GPC6. Acta neuropathologica communications9(1), 52. DOI:10.1186/s40478-021-01148-z

2020

Daniela C. Zarnescu; Think globally, act locally: Centrosome-localized mRNAs ensure mitotic fidelity. J Cell Biol 7 December 2020; 219 (12): e202010172. DOI: 10.1083/jcb.202010172

2020

Nadine Bakkar, Alexander Starr, Benjamin E. Rabichow, Ileana Lorenzini, Zachary T. McEachin, Robert Kraft, Matthew Chaung, Sam Macklin-Isquierdo, Taylor Wingfield, Briggs Carhart, Nathan Zahler, Wen-Hsuan Chang, Gary J. Bassell, Alexandre Betourne, Nicholas Boulis, Samuel V. Alworth, Justin K. Ichida, Paul R. August, Daniela C. Zarnescu, Rita Sattler, Robert Bowser. The M1311V variant of ATP7A is associated with impaired trafficking and copper homeostasis in models of motor neuron disease,
Neurobiology of Disease 2021;149. DOI: 10.1016/j.nbd.2020.105228

2020

Loganathan S, Lehmkuhl E, Eck R, Zarnescu D. To Be or Not To Be…Toxic—Is RNA Association With TDP-43 Complexes Deleterious or Protective in Neurodegeneration? Frontiers in Molecular Biosciences 2020;6:154. DOI: 10.3389/fmolb.2019.00154

2019

Manzo E, Lorenzini I, Barrameda D, Conner A,  Barrows J, Starr A, Kovalik T, Shreiner D,
Joardar A, Lievens JC, Bowse R, Sattler R, Zarnescu D. Glycolysis upregulation is neuroprotective as a compensatory mechanism in ALS. eLife 2019;8:e45114. DOI: 10.7554/eLife.45114

2018

Ferro D, Yao S, Zarnescu D. Dynamic duo – FMRP and TDP-43: Regulating common targets, causing different diseases. Brain Research. 2018;1693(A):37-42. ISSN 0006-8993 DOI: 10.1016/j.brainres.2018.04.034

2018

Chu M, Novak SM, Cover C, Wang AA, Chinyere IR, Juneman EB, Zarnescu DC, Wong PK, Gregorio CC. Increased Cardiac Arrhythmogenesis Associated with Gap Junction Remodeling with Upregulation of RNA-Binding Protein FXR1. Circulation. 137:605-618. PMID:29101288 DOI: 10.1161/CIRCULATIONAHA.117.028976 

2018

Lehmkuhl E.M., Zarnescu D.C. Lost in Translation: Evidence for Protein Synthesis Deficits in ALS/FTD and Related Neurodegenerative Diseases. Advances in Neurobiology. 20:283-301.DOI: 10.1007/978-3-319-89689-2_11

2018

Manzo E, O’Conner AG, Barrows JM, Shreiner DD, Birchak GJ,  Zarnescu DC. Medium-chain fatty acids, beta-hydroxybutyric acid and genetic modulation of the carnitine shuttle are protective in a drosophila model of ALS based on TDP-43. Frontiers in Molecular Neuroscience. 11, 182. PMID: 29904341 DOI: 10.3389/fnmol.2018.00182

2018

Daniel SG, Russ AD, Guthridge KM, Raina AI, Estes PS, Parsons LM, Richardson HE, Schroeder JA, Zarnescu DC. miR-9a mediates the role of Lethal giant larvae as an epithelial growth inhibitor in Drosophila. Biology Open. PMID 29361610 DOI: 10.1242/bio.027391 

2018

Chou C-C, Zhang Y, Umoh ME, Vaughan SW, Lorenzini I, Liu F, Sayegh M, Donlin-Asp PG, Chen YH, Duong DM, Seyfried NT, Powers MA, Kukarm T, Hales CM, Gearing M, Cairns NJ,  Boylan KB, Dickson DW, Rademakers R, Zhang YJ, Petrucelli L, Sattler R, Zarnescu DC,  Glass JD, Rossoll W. TDP-43 pathology disrupts nuclear pore complexes and nucleocytoplasmic transport in ALS/FTD. Nat Neurosci. 2018 Feb; 21(2): 228–239. PMCID:PMC5800968 DOI: 10.1038/s41593-017-0047-3

2017

Daniel SG, Russ AD, Guthridge KM, Raina AI, Estes PS, Parsons LM, Richardson HE, Schroeder JA, Zarnescu DC. miR-9a mediates the role of Lethal giant larvae as an epithelial growth inhibitor in Drosophila. Biology Open. PMID 29233983 DOI: 10.1242/bio.027391

2017

Liu G, Coyne AN, Pei F, Vaughan S, Chaung M, Zarnescu DC, Buchan JR. Endocytosis regulates TDP-43 toxicity and turnover. Nature Communications. 8: 2092. PMID 29233983 DOI: 10.1038/s41467-017-02017-x 

2017

Coyne AN, Lorenzini I, Chou C-C, Torvund M, Rogers RS, Starr A, Zaepfel BL, Levy J, Johannesmeyer J, Schwartz J,  Nishimune H, Zinsmaier K, Rossoll W, Sattler R, Zarnescu DC. Post-transcriptional inhibition of Hsc70-4/HSPA8 expression leads to synaptic vesicle cycling defects in multiple models of ALS. Cell reports. 2017;21(1):110-125. DOI:10.1016/j.celrep.2017.09.028.

2017

Coyne AN, Zaepfel BL, Zarnescu DC. Failure to Deliver and Translate-New Insights into RNA Dysregulation in ALS. Frontiers in Cellular Neuroscience. 11: 243. PMID 28860970 DOI: 10.3389/fncel.2017.00243

2016

Coyne AN, Zarnescu DC. A Helping Hand: RNA-Binding Proteins Guide Gene-Binding Choices by Cohesin Complexes. Plos Genetics. 12: e1006419. PMID 27855157 DOI: 10.1371/journal.pgen.1006419

2015

Novak SM, Joardar A, Gregorio CC, Zarnescu DC. Regulation of Heart Rate in Drosophila via Fragile X Mental Retardation Protein. Plos One. 10: e0142836. PMID 26571124 DOI: 10.1371/journal.pone.0142836

2015

Coyne AN, Yamada SB, Siddegowda BB, Estes PS, Zaepfel BL, Johannesmeyer JS, Lockwood DB, Pham LT, Hart MP, Cassel JA, Freibaum B, Boehringer AV, Taylor JP, Reitz AB, Gitler AD, … Zarnescu DC, et al. Fragile X protein mitigates TDP-43 toxicity by remodeling RNA granules and restoring translation. Human Molecular Genetics. PMID 26385636 DOI: 10.1093/hmg/ddv389

2015

Chou CC, Alexeeva OM, Yamada S, Pribadi A, Zhang Y, Mo B, Williams KR, Zarnescu DC, Rossoll W. PABPN1 suppresses TDP-43 toxicity in ALS disease models. Human Molecular Genetics. PMID 26130692 DOI: 10.1093/hmg/ddv238

2015

Joardar A, Menzl J, Podolsky TC, Manzo E, Estes PS, Ashford S, Zarnescu DC. PPAR gamma activation is neuroprotective in a Drosophila model of ALS based on TDP-43. Human Molecular Genetics. 24: 1741-54. PMID 25432537 DOI: 10.1093/hmg/ddu587

2014

Coyne AN, Siddegowda BB, Estes PS, Johannesmeyer J, Kovalik T, Daniel SG, Pearson A, Bowser R, Zarnescu DC. Futsch/MAP1B mRNA is a translational target of TDP-43 and is neuroprotective in a Drosophila model of amyotrophic lateral sclerosis. The Journal of Neuroscience : the Official Journal of the Society For Neuroscience. 34: 15962-74. PMID 25429138 DOI: 10.1523/JNEUROSCI.2526-14.2014

2013

Zarnescu DC, Gregorio CC. Fragile hearts: new insights into translational control in cardiac muscle. Trends in Cardiovascular Medicine. 23: 275-81. PMID 23582851 DOI: 10.1016/j.tcm.2013.03.003

2013

Estes PS, Daniel SG, McCallum AP, Boehringer AV, Sukhina AS, Zwick RA, Zarnescu DC. Motor neurons and glia exhibit specific individualized responses to TDP-43 expression in a Drosophila model of amyotrophic lateral sclerosis. Disease Models & Mechanisms. 6: 721-33. PMID 23471911 DOI: 10.1242/dmm.010710

2013

Morera AA, Coyne A, Zarnescu DC. Flies lies in motion: What drosophila can tell us about amyotrophic lateral sclerosis Drosophila Melanogaster Models of Motor Neuron Disease. 57-83. (Abstract)

2012

Russ A, Louderbough JM, Zarnescu D, Schroeder JA. Hugl1 and Hugl2 in mammary epithelial cells: polarity, proliferation, and differentiation. Plos One. 7: e47734. PMID 23110097 DOI: 10.1371/journal.pone.0047734

2012

Callan MA, Clements N, Ahrendt N, Zarnescu DC. Fragile X Protein is required for inhibition of insulin signaling and regulates glial-dependent neuroblast reactivation in the developing brain. Brain Research. 1462: 151-61. PMID 22513101 DOI: 10.1016/j.brainres.2012.03.042

2011

Whitman SA, Cover C, Yu L, Nelson DL, Zarnescu DC, Gregorio CC. Desmoplakin and talin2 are novel mRNA targets of fragile X-related protein-1 in cardiac muscle. Circulation Research. 109: 262-71. PMID 21659647 DOI: 10.1161/CIRCRESAHA.111.244244

2011

Estes PS, Boehringer A, Zwick R, Tang JE, Grigsby B, Zarnescu DC. Wild-type and A315T mutant TDP-43 exert differential neurotoxicity in a Drosophila model of ALS. Human Molecular Genetics. 20: 2308-21. PMID 21441568 DOI: 10.1093/hmg/ddr124

2011

Callan MA, Zarnescu DC. Heads-up: new roles for the fragile X mental retardation protein in neural stem and progenitor cells. Genesis (New York, N.Y. : 2000). 49: 424-40. PMID 21404421 DOI: 10.1002/dvg.20745

2010

Callan MA, Cabernard C, Heck J, Luois S, Doe CQ, Zarnescu DC. Fragile X protein controls neural stem cell proliferation in the Drosophila brain. Human Molecular Genetics. 19: 3068-79. PMID 20504994 DOI: 10.1093/hmg/ddq213

2010

Lee HG, Zarnescu DC, MacIver B, Thomas GH. The cell adhesion molecule Roughest depends on beta(Heavy)-spectrin during eye morphogenesis in Drosophila. Journal of Cell Science. 123: 277-85. PMID 20048344 DOI: 10.1242/jcs.056853

2010

Mounsef J, Karam L, Estes P, Zarnescu D. Shape analysis and classification of lgl-type and wild-type neurons 2010 Acm International Conference On Bioinformatics and Computational Biology, Acm-Bcb 2010. 362-365. DOI: 10.1145/1854776.1854830

2009

Zarnescu DC, Zinsmaier KE. Ferrying wingless across the synaptic cleft. Cell. 139: 229-31. PMID 19837027 DOI: 10.1016/j.cell.2009.09.032

2009

Cziko AM, McCann CT, Howlett IC, Barbee SA, Duncan RP, Luedemann R, Zarnescu D, Zinsmaier KE, Parker RR, Ramaswami M. Genetic modifiers of dFMR1 encode RNA granule components in Drosophila. Genetics. 182: 1051-60. PMID 19487564 DOI: 10.1534/genetics.109.103234

2009

Epstein AM, Bauer CR, Ho A, Bosco G, Zarnescu DC. Drosophila Fragile X protein controls cellular proliferation by regulating cbl levels in the ovary. Developmental Biology. 330: 83-92. PMID 19306863 DOI: 10.1016/j.ydbio.2009.03.011

2008

Bauer CR, Epstein AM, Sweeney SJ, Zarnescu DC, Bosco G. Genetic and systems level analysis of Drosophila sticky/citron kinase and dFmr1 mutants reveals common regulation of genetic networks. Bmc Systems Biology. 2: 101. PMID 19032789 DOI: 10.1186/1752-0509-2-101

2008

Estes PS, O’Shea M, Clasen S, Zarnescu DC. Fragile X protein controls the efficacy of mRNA transport in Drosophila neurons. Molecular and Cellular Neurosciences. 39: 170-9. PMID 18655836 DOI: 10.1016/j.mcn.2008.06.012

2008

Chang S, Bray SM, Li Z, Zarnescu DC, He C, Jin P, Warren ST. Identification of small molecules rescuing fragile X syndrome phenotypes in Drosophila. Nature Chemical Biology. 4: 256-63. PMID 18327252 DOI: 10.1038/nchembio.78

2005

Zarnescu DC, Shan G, Warren ST, Jin P. Come FLY with us: toward understanding fragile X syndrome. Genes, Brain, and Behavior. 4: 385-92. PMID 16098136 DOI: 10.1111/j.1601-183X.2005.00136.x

2005

Zarnescu DC, Jin P, Betschinger J, Nakamoto M, Wang Y, Dockendorff TC, Feng Y, Jongens TA, Sisson JC, Knoblich JA, Warren ST, Moses K. Fragile X protein functions with lgl and the par complex in flies and mice. Developmental Cell. 8: 43-52. PMID 15621528 DOI: 10.1016/j.devcel.2004.10.020

2004

Zarnescu DC, Moses K. Born again at the synapse: a new function for the anaphase promoting complex/cyclosome. Developmental Cell. 7: 777-8. PMID 15572119 DOI: 10.1016/j.devcel.2004.11.011

2004

Jin P, Zarnescu DC, Ceman S, Nakamoto M, Mowrey J, Jongens TA, Nelson DL, Moses K, Warren ST. Biochemical and genetic interaction between the fragile X mental retardation protein and the microRNA pathway. Nature Neuroscience. 7: 113-7. PMID 14703574 DOI: 10.1038/nn1174

2003

Jin P, Zarnescu DC, Zhang F, Pearson CE, Lucchesi JC, Moses K, Warren ST. RNA-mediated neurodegeneration caused by the fragile X premutation rCGG repeats in Drosophila. Neuron. 39: 739-47. PMID 12948442 DOI: 10.1016/S0896-6273(03)00533-6

2002

Médina E, Williams J, Klipfell E, Zarnescu D, Thomas G, Bivic AL. Crumbs interacts with moesin and βHeavy-spectrin in the apical membrane skeleton of Drosophila. Journal of Cell Biology. 158: 941-951. PMID 12213838 DOI: 10.1083/jcb.200203080

1999

Zarnescu DC, Thomas GH. Apical spectrin is essential for epithelial morphogenesis but not apicobasal polarity in Drosophila. The Journal of Cell Biology. 146: 1075-86. PMID 10477760 DOI: 10.1083/jcb.146.5.1075

1998

Thomas GH, Zarnescu DC, Juedes AE, Bales MA, Londergan A, Korte CC, Kiehart DP. Drosophila betaHeavy-spectrin is essential for development and contributes to specific cell fates in the eye. Development (Cambridge, England). 125: 2125-34. PMID 9570776