Publications by Year












Krench, M. & LittletonJ.T. (2016) Neurotoxicity pathways in Drosophila models of the polyglutamine disorders. Current Topics in Developmental Biology, In Press.

Weiss, K.R. & LittletonJ.T. (2016) Fast axonal transport defects in Drosophila Huntingtin mutants. J. Neurogenetics 16:1-30.

Krench, M., Cho, R. & LittletonJ.T. (2016) A Drosophila model of Huntington disease-like 2 exhibits nuclear toxicity and distinct pathogenic mechanisms from Huntington disease. Human Molecular Genetics, ddw166.

Harris, K.P., Cho, R.W., Akbergenova, Y., Baas-Thomas, M. & Littleton, J.T. (2016) Shank modulates postsynaptic Wnt signaling to regulate synaptic development. J. Neuroscience 36: 5820-5832.

Harris, K.P., Piccioli, Z.D., Perrimon, N. & Littleton, J.T. (2016) The postsynaptic t-SNARE Syntaxin 4 controls traffic of Neuroligin 1 and Synaptotagmin 4 to regulate retrograde signaling. Elife: e13881.

Park, S.M., Littleton, J.T., Park, H.R. & Lee, J. (2016) Drosophila homolog of human KIF22 at the autism-linked 16p11.2 loci influences synaptic connectivity at larval neuromuscular junctions. Exp. Neurobiol. 25: 33-39.



Cho, R.W., Buhl L.K., Volfson, D., Tran, A., Feng, L., Akbergenova, Y. & Littleton, J.T. (2015) Phosphorylation of Complexin by PKA regulates activity-dependent spontaneous neurotransmitter release and structural synaptic plasticity. Neuron 88: 749-761.

Whittaker, R.G., Herrmann, D.N., Bansagi, B., Hasan B.A.S., Lofra, R.M., Logigian, E.L., Sowden, J.E., Almodovar, J.L., Littleton, J.T., Zuchner, S., Horvath, R. & Lochmuller, H. (2015) Electrophysiological features of SYT2 mutations causing a treatable neuromuscular syndrome. Neurology 85: 1964-1971.

Harris, K.P. & Littleton, J.T. (2015) Transmission, Development and Plasticity of Synapses. Genetics 201: 345-375.

Lee, J. & Littleton, J.T. (2015) Transmembrane tethering of Synaptotagmin to synaptic vesicles controls multiple modes of neurotransmitter release. PNAS 112: 3793-3798.


Herrmann D.N., Horvath R., Sowden J.E., Gonzalez M., Sanchez-Mejias A., Guan Z., Whittaker R.G., Almodovar J.L., Lane M., Bansagi B., Pyle A., Boczonadi V., Lochmüller H., Griffin H., Chinnery P.F., Lloyd T.E., Littleton J.T., Zuchner S. Synaptotagmin 2 mutations cause an autosomal-dominant form of lambert-eaton myasthenic syndrome and nonprogressive motor neuropathy. Am J Hum Genet 95, 332-339.

Cho R.W., Kümmel D., Li F., Baguley S.W., Coleman J., Rothman J.E.,Littleton J.T.Genetic analysis of the Complexin trans-clamping model for cross-linking SNARE complexes in vivo. PNAS 111, 10317-10322.

Blunk A.D., Akbergenova Y., Cho R.W., Lee J., Walldorf U., Xu K., Zhong G., ZHuang X., Littleton J.T. Postsynaptic actin regulates active zone spacing and glutamate receptor apposition at the Drosophila neuromuscular junction.Mol Cell Neuroscience 61, 241-254. 


Piccioli Z.D., Littleton J.T. Retrograde BMP signaling modulates rapid activity-dependent synaptic growth via presynaptic LIM kinase regulation of cofilin. J. Neuroscience 34, 4371-4381. 


Vasin, A., Zueva, L., Torrez, C., Volfson, D., Littleton, J.T. & Bykhovskaia, M. (2014) Synapsin regulates activity-dependent outgrowth of synaptic boutons at the Drosophila neuromuscular junction. J. Neuroscience 34: 10554-10563. 



Melom, J.E. & Littleton, J.T. (2013) Mutation of a NCKX eliminates glial microdomain calcium oscillations and enhances seizure susceptibility. J. Neuroscience 33: 1169-1178.

Lee, J., Guan, Z., Akbergenova, Y. & Littleton, J.T. (2013) Genetic analysis of Synaptotagmin C2 domain specificity in regulating spontaneous and evoked neurotransmitter release. J. Neuroscience 33: 187-200.

Melom, J.E., Akbergenova, Y., Gavornik, J.P. & Littleton, J.T. (2013) Spontaneous and evoked release are independently regulated at individual active zones.  J. Neuroscience 33: 17253-17263.

Bykhovskaia, M., Jagota, A., Gonzalez, A., Vasin, A. & Littleton, J.T. (2013) Interaction of the Complexin accessory helix with the C-terminus of the SNARE complex: Molecular dynamics model of the fusion clamp. Biophysical Journal 105: 679-690.

Buhl, L.K., Jorquera, R.A., Akbergenova, Y., Huntwork-Rodriguez, S., Volfson, D. & Littleton, J.T. (2013) Differential regulation of evoked and spontaneous neurotransmitter release by C-terminal modifications of Complexin. Mol. Cell. Neuroscience 52: 161-172.

Krench, M. & Littleton, J.T. (2013) Modeling Huntington’s Disease in Drosophila: Insights into axonal transport defects and modifiers of toxicity. Fly 7: 229-236.



Weiss, K.R., Kimura, Y., Lee, W.M. & Littleton, J.T.  (2012) Huntingtin aggregation kinetics and their pathological role in a Drosophila Huntington’s Disease Model. Genetics 190, 581-600.

Jorquera, R.A., Huntwork-Rodriguez, S., Akbergenova, Y., Cho, R.W. & Littleton, J.T. (2012) Complexin controls spontaneous and evoked neurotransmitter release by regulating the timing and properties of Synaptotagmin activity. J. Neuroscience 32, 18234-18245.

Stevens, R., Akbergenova, Y., Jorquera, R.A. & Littleton, J.T. (2012) Abnormal synaptic vesicle biogenesis in Drosophila synaptogyrin mutants. J. Neuroscience 32, 18054-18067.



Guan, Z., Barr, L.K., Quinn, W. & Littleton, J.T. (2011) Altered gene regulation and synaptic morphology in Drosophila learning and memory mutants. Learning & Memory 18, 191-206.

Rodal, AA, Blunk, A.D., Akbergenova, Y., Jorquera, R.A., Buhl, L.K. & Littleton, J.T. (2011) A presynaptic endosomal trafficking pathway controls synaptic growth signaling. J. Cell Biology 193, 201-217.

Stevens, R.J. & Littleton, J.T. (2011) Synaptic growth: dancing with adducin. Current Biology 21, R402-405.

Melom, J.E. & Littleton, J. T. (2011) Synapse development in health and disease. Current Opinions in Genetics & Development 21, 256-261.

Schulte, J., Sepp, K., Wu, C., Hong, P. & Littleton, J.T. (2011) High-content chemical and RNAi screens for suppressors of neurotoxicity in a Huntington’s Disease model. PLoS ONE 6, e23841, 1-13.

Harris, K.P. & Littleton, J.T. (2011) Neuronal vesicle trafficking: a Rab family profile. Current Biology 21, R841-843.

Schulte, J. & Littleton, J.T. (2011) The biological function of the Huntington protein and its relevance to Huntington’s Disease pathology. Current Trends in Neurology 5, 65-78.



Schulte, J., Sepp, K., Jorquera, R.A., Wu, C., Song, Y., Hong, P. & Littleton, J.T. (2010) DMob4/Phocein regulates synapse formation, axonal transport, and microtubule organization. J. Neuroscience30, 5189-5203.

Wu, C., Schulte, J., Sepp, K., Littleton, J.T. & Hong, P. (2010) Automatic robust neurite detection and morphological analysis of neuronal cell cultures in high-content screening. Neuroinformatics 8, 83-100.

Yoshihara, M., Guan, Z. & Littleton J.T. (2010) Differential regulation of synchronous versus asynchronous neurotransmitter release by the C2 domains of Synaptotagmin 1. PNAS 107, 14869-14874.

Cho, R., Song, Y. & Littleton, J.T. (2010) Comparative analysis of Drosophila and mammalian complexins as fusion clamps and facilitators of neurotransmitter release. Mol. Cell. Neuroscience 45, 389-397.



Zhang, S., Feany, M.B., Saraswati, S., Littleton, J.T. & Perrimon, N. (2009) Inactivation of Drosophila Huntingtin affects long-term adult functioning and the pathogenesis of a Huntington’s disease model. Disease Models & Mechanisms 2, 247-266.

Barber, C., Jorquera, R.A., Melom, J.E. & Littleton, J.T. (2009) Postsynaptic regulation of synaptic plasticity by Synaptotagmin 4 requires both C2 domains. J. Cell Biology 187, 295-310.

Rodal, A.A. & Littleton, J.T. (2008) Synaptic endocytosis: Illuminating the role of clathrin assembly. Current Biology 18, 259-261.

Rodal, A.A., Motola-Barnes, R.N. & Littleton, J.T. (2008) Nervous Wreck and Cdc42 cooperate to regulate endocytic actin assembly during synaptic growth.  J. Neuroscience 28, 8316-8325.

Kimura, Y., Lee, W.C. & Littleton, J.T. (2007) Therapeutic prospects for the prevention of neurodegeneration in Huntington’s Disease and the polyglutamine repeat disorders. Mini Rev. Med. Chem. 7, 99-106.

Barber, C. & Littleton, J. T. (2007) Synaptic Growth and Transcriptional Regulation in Drosophila, 253-275.  In: Regulation of Transcription by Neuronal Activity, Ed. S. Dudek, Springer Science Publishing @2007.

Saraswati, S., Adolfsen, B. & Littleton, J.T.  (2007) Characterization of the role of the Synaptotagmin family as calcium sensors in facilitation and asynchronous neurotransmitter release.  PNAS 104, 14122-14127.

Mattaliano, M.D., Montana, E.S., Parisky, K., Littleton, J.T., Griffeth, L.C. (2007) The Drosophila ARC homolog regulates behavioral responses to starvation.  Molecular and Cellular Neuroscience 36, 211-221.

Huntwork, S. & Littleton, J.T. (2007) A complexin fusion clamp regulates spontaneous neurotransmitter release and synaptic growth.  Nature Neuroscience 10, 1235-1237.

 The Honey Bee Genome Sequencing Consortium (2006) Insights into social insects from the genome of the honeybee Apis mellifera.  Nature 443, 931-949.

Stilwell, G., Saraswati, S., Littleton, J.T. & Chouinard S.W.  (2006) Development of a Drosophila seizure model for in vivo high-throughput drug screening. Eur. J. Neuroscience 24, 2211-2222.

Montana, E.S. & Littleton, J.T. (2006) Expression profiling of a hypercontraction-induced myopathy in Drosophila suggests a compensatory cytoskeletal remodeling response. J. Biol. Chem. 281, 8100-8109.

 Littleton, J.T. (2006) Mixing and matching during synaptic vesicle endocytosis.  Neuron 51, 149-151.

Yoshihara, M., Adolfsen, B., Galle, K & Littleton J.T. (2005) Retrograde signaling by Syt 4 induces presynaptic release and synapse-specific growth.  Science 310, 858-863.

Guan, Z., Saraswati, S., Adolfsen, B. & Littleton, J.T. (2005) Genome-wide transcriptional changes associated with enhanced activity in the Drosophila nervous system.  Neuron 48, 91-107.

Montana, E.S. & Littleton, J.T. (2004) Characterization of a hypercontraction induced myopathy in Drosophila caused by mutations in Myosin heavy chain. J. Cell Biology 164, 1045-1054.

Wang, P., Saraswati, S., Guan, Z., Watkins, C., Wurtman, R.J. & Littleton, J.T. (2004) A Drosophila temperature-sensitive seizure mutant in phosphoglycerate kinase disrupts ATP generation and alters synaptic function.  J. Neuroscience 24, 4518-4529. 

Coyle, I., Koh, Y-H., Lee, W.C.M, Slind, J., Fergestad, T., Littleton, J.T. & Ganetzky, B. (2004)  Nervous Wreck, an SH3 adapter protein that interacts with Wsp, regulates synaptic growth inDrosophila.  Neuron 41, 521-534.

Lee, W.C., Yoshihara, M. & Littleton, J.T. (2004) Cytoplasmic aggregates trap polyglutamine-containing proteins and block axonal transport in a Drosophila model of Huntington’s Disease.  PNAS 101, 3224-3229.

Adolfsen, B., Saraswati, S., Yoshihara, M. & Littleton, J.T (2004) Synaptotagmins are trafficked to distinct subcellular domains including the postsynaptic compartment.  J. Cell Biology 166, 249-260.

Rieckhof, G.E., Yoshihara, M., Guan, Z. & Littleton, J.T. (2003) Presynaptic N-type calcium channels regulate synaptic growth.  J. Biol. Chem. 278, 41099-41108.

Littleton, J.T. & Sheng, M. (2003) Synapses unplugged.  Nature 423, 931-932.

Yoshihara, M., Adolfsen, B. & Littleton, J.T. (2003) Is synaptotagmin the calcium sensor?  Current Opinions in Neurobiology 13, 315-323.

Yoshihara, M. & Littleton, J.T. (2002) Synaptotagmin I functions as a calcium sensor to synchronize neurotransmitter release.  Neuron 36, 897-908.

Littleton, J.T., Vyas, B., Bai, J., Desai, R.C., Garment, M.B., Carlson, S.D., Ganetzky, B. & Chapman, E.R.  (2001) synaptotagmin mutants reveal essential functions for the C2B-domain in Ca2+-triggered fusion and recycling of synaptic vesicles.  J. Neuroscience 21, 1421-1433.

Littleton, J.T., Barnard, R.J., Titus, S., Slind, J., Chapman E.R. & Ganetzky, B.  (2001) SNARE complex disassembly by NSF follows synaptic vesicle fusion.  PNAS 98, 12233-12238.

Adolfsen, B. & Littleton, J.T.  (2001) Genetic and molecular analysis of the synaptotagmin family.  Cell. Mol. Life Sci. 58, 393-402.

Yoshihara, M., Ensminger, A. & Littleton, J.T. (2001) Neurobiology and the Drosophila genome.   Functional & Integrative Genomics 1, 235-240. 

Desai, R.C., Vyas, B., Earles, C., Littleton, J.T., Kowalchyck, J.A., Martin, T.F.J. & Chapman, E.R.  (2000) The C2B domain of synaptotagmin is a Ca2+-sensing module essential for exocytosis. J. Cell Biology 150, 1125-1135.

Rubin, G.M., et al. (2000) Comparative genomics of the eukaryotes.  Science 287, 2204-2215.

Littleton, J.T. & Ganetzky, B. (2000) Ion Channels and Synaptic Organization: Analysis of the Drosophila Genome.  Neuron 26, 35-43.

Littleton, J.T. (2000) A genomics perspective on membrane trafficking and neurotransmitter release in Drosophila.  J. Cell Biology 150, F77-F81.



Littleton, J.T., Pallanck, L. & Ganetzky, B.  (1999)  Mechanisms of neurotransmitter release. Int. Rev. Neurobiol. 43, 139-61; in Neuromuscular Junctions in Drosophila, Eds. Vivian Budnik and L. Sian Gramates, Academic Press @1999.

Littleton, J.T., Sereno T.L., Rubin, G.M., Ganetzky, B. & Chapman E.R. (1999) Synaptic function modulated by changes in the ratio of synaptotagmin I and IV.  Nature  400, 757-600.

Wu, M.N., Littleton, J.T., Bhat, M.A., Prokop, A. & Bellen, H.J. (1998) ROP, the Drosophila Sec1 homolog, interacts with syntaxin and regulates neurotransmitter release in a dosage dependent manner. EMBO J. 17, 127-139.

Littleton, J.T., Chapman, E.R., Kreber, R., Garment, M.B., Carlson, S.D. & Ganetzky, B. (1998) Temperature-sensitive paralytic mutations demonstrate that synaptic exocytosis requires SNARE complex assembly and disassembly.  Neuron  21, 401-413.

Littleton, J.T., Bhat, M.A. & Bellen, H.J. (1997) Deciphering the role of neurexins at cellular junctions.  J. Cell Biology 137, 793-796.

Baumgartner S.*, Littleton, J.T.*, Broadie, K., Bhat, M., Harbecke, R., Lengyel, J., Chiquet-Ehrismann, R., Prokop, A. & Bellen, H.J. (1996)  A Drosophila neurexin is required for septate junction and blood-nerve barrier formation and function.  Cell 87, 1059-1068.  *Equal Contribution. 

Littleton, J.T., Upton, L. & Kania, A. (1995) Immunocytochemical analysis of axonal outgrowth in synaptotagmin mutations.  J. Neurochemistry  65, 32-40.

Littleton, J.T. & Bellen, H.J. (1995) Presynaptic proteins involved in exocytosis in Drosophila melanogaster:  a genetic analysis.  Invertebrate Neuroscience  1, 3-13.

Littleton, J.T. & Bellen, H.J. (1995) Synaptotagmin controls and modulates synaptic vesicle fusion in a calcium dependent manner.  Trends in Neurosciences  18, 177-183.

Littleton, J.T. & Bellen, H.J.  (1994) Genetic and phenotypic analysis of 13 essential genes in cytological interval 22F1-2; 23B1-2 reveals novel genes required for neuronal development in DrosophilaGenetics 138, 111-123.

Littleton, J.T., Stern, M., Perin, M. & Bellen, H.J. (1994) Calcium dependence of neurotransmitter release and rate of spontaneous vesicle fusions are altered in Drosophila synaptotagmin mutants.  PNAS 91, 10888-10892.

Broadie, K., Bellen, H.J., DiAntonio, A., Littleton, J.T. & Schwarz, T.L. (1994) The absence of synaptotagmin disrupts excitation-secretion coupling during synaptic transmission.  PNAS  91, 10727-10731.

Schulze, K.*, Littleton, J.T.*, Salzberg, A., Cohen, N., Stern, M., Lev, Z. & Bellen, H.J.  (1994) ROP, a Drosophila homolog of yeast Sec1 and vertebrate n-Sec1/Munc-18 proteins, is a negative regulator of neurotransmitter release in vivo.  Neuron 13, 1099-1108.  *Equal Contribution.

Littleton, J.T., Bellen, H.J. & Perin, M. (1993) Expression of synaptotagmin in Drosophila reveals transport and localization of synaptic vesicles to the synapse.  Development 118, 1077-1088.

Littleton, J.T., Stern, M., Schulze, K., Perin, M. & Bellen, H.J. (1993) Mutational analysis of Drosophila synaptotagmin demonstrates its essential role in calcium-activated neurotransmitter release.  Cell  74, 1125-1134.



Gleeson, R.A., Trapido-Rosenthal, H.G., Littleton, J.T. & Carr, W.E.S. (1989) Purinergic receptors and dephosphorylating enzymes occur in both the gustatory and olfactory systems of the Spiny Lobster.  Comp. Biochem. Physiol. 92C, 413-417.