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2025

Journal Article

Cre-Lox miRNA-delivery technology optimized for inducible microRNA and gene-silencing studies in zebrafish

Guo, Fangfei, Tromp, Alisha, Wang, Haitao, Hall, Thomas E and Giacomotto, Jean (2025). Cre-Lox miRNA-delivery technology optimized for inducible microRNA and gene-silencing studies in zebrafish. Nucleic Acids Research, 53 (2) gkaf004, 2. doi: 10.1093/nar/gkaf004

Cre-Lox miRNA-delivery technology optimized for inducible microRNA and gene-silencing studies in zebrafish

2024

Journal Article

Clinical and molecular spectrum of autosomal recessive CA8‐related cerebellar ataxia

Kaiyrzhanov, Rauan, Ortigoza‐Escobar, Juan Darío, Stringer, Brett W., Ganieva, Manizha, Gowda, Vykuntaraju K., Srinivasan, Varunvenkat M., Macaya, Alfons, Laner, Andreas, Onbool, Enas, Al‐Shammari, Randa, Al‐Owain, Mohammed, Deconinck, Nicolas, Vilain, Catheline, Dontaine, Pauline, Self, Eleanor, Akram, Rabia, Hussain, Ghulam, Baig, Shahid Mahmood, Iqbal, Javed, Salpietro, Vincenzo, Neshatdoust, Maedeh, Kasiri, Mahboubeh, Yesil, Gozde, Uygur, Turkan, Pysden, Karen, Berry, Ian R., Alves, Cesar Augusto, Giacomotto, Jean, Houlden, Henry and Maroofian, Reza (2024). Clinical and molecular spectrum of autosomal recessive CA8‐related cerebellar ataxia. Movement Disorders, 39 (6), 983-995. doi: 10.1002/mds.29754

Clinical and molecular spectrum of autosomal recessive CA8‐related cerebellar ataxia

2023

Journal Article

Cell surface plasticity in response to shape change in the whole organism

Hall, Thomas E., Ariotti, Nicholas, Lo, Harriet P., Rae, James, Ferguson, Charles, Martel, Nick, Lim, Ye-Wheen, Giacomotto, Jean and Parton, Robert G. (2023). Cell surface plasticity in response to shape change in the whole organism. Current Biology, 33 (19), 4276-4284. doi: 10.1016/j.cub.2023.08.068

Cell surface plasticity in response to shape change in the whole organism

2023

Journal Article

Optimising the zebrafish Cre/Lox toolbox. Codon improved iCre, new gateway tools, Cre protein and guidelines

Tromp, Alisha, Wang, Haitao, Hall, Thomas E., Mowry, Bryan and Giacomotto, Jean (2023). Optimising the zebrafish Cre/Lox toolbox. Codon improved iCre, new gateway tools, Cre protein and guidelines. Frontiers in Physiology, 14 1221310, 1-10. doi: 10.3389/fphys.2023.1221310

Optimising the zebrafish Cre/Lox toolbox. Codon improved iCre, new gateway tools, Cre protein and guidelines

2022

Journal Article

Brain-wide visual habituation networks in wild type and fmr1 zebrafish

Marquez-Legorreta, Emmanuel, Constantin, Lena, Piber, Marielle, Favre-Bulle, Itia A., Taylor, Michael A., Blevins, Ann S., Giacomotto, Jean, Bassett, Dani S., Vanwalleghem, Gilles C. and Scott, Ethan K. (2022). Brain-wide visual habituation networks in wild type and fmr1 zebrafish. Nature Communications, 13 (1) 895. doi: 10.1038/s41467-022-28299-4

Brain-wide visual habituation networks in wild type and fmr1 zebrafish

2022

Journal Article

Functional characterisation of the amyotrophic lateral sclerosis risk locus GPX3/TNIP1

Restuadi, Restuadi, Steyn, Frederik J., Kabashi, Edor, Ngo, Shyuan T., Cheng, Fei-Fei, Nabais, Marta F., Thompson, Mike J., Qi, Ting, Wu, Yang, Henders, Anjali K., Wallace, Leanne, Bye, Chris R., Turner, Bradley J., Ziser, Laura, Mathers, Susan, McCombe, Pamela A., Needham, Merrilee, Schultz, David, Kiernan, Matthew C., van Rheenen, Wouter, van den Berg, Leonard H., Veldink, Jan H., Ophoff, Roel, Gusev, Alexander, Zaitlen, Noah, McRae, Allan F., Henderson, Robert D., Wray, Naomi R., Giacomotto, Jean and Garton, Fleur C. (2022). Functional characterisation of the amyotrophic lateral sclerosis risk locus GPX3/TNIP1. Genome Medicine, 14 (1) 7, 7. doi: 10.1186/s13073-021-01006-6

Functional characterisation of the amyotrophic lateral sclerosis risk locus GPX3/TNIP1

2021

Journal Article

Cavin4 interacts with Bin1 to promote T-tubule formation and stability in developing skeletal muscle

Lo, Harriet P., Lim, Ye-Wheen, Xiong, Zherui, Martel, Nick, Ferguson, Charles, Ariotti, Nicholas, Giacomotto, Jean, Rae, James, Floetenmeyer, Matthias, Moradi, Shayli Varasteh, Gao, Ya, Tillu, Vikas A., Xia, Di, Wang, Huang, Rahnama, Samira, Nixon, Susan J., Bastiani, Michele, Day, Ryan D., Smith, Kelly A., Palpant, Nathan J., Johnston, Wayne A., Alexandrov, Kirill, Collins, Brett M., Hall, Thomas E. and Parton, Robert G. (2021). Cavin4 interacts with Bin1 to promote T-tubule formation and stability in developing skeletal muscle. Journal of Cell Biology, 220 (12) e201905065. doi: 10.1083/jcb.201905065

Cavin4 interacts with Bin1 to promote T-tubule formation and stability in developing skeletal muscle

2021

Journal Article

Pipeline for generating stable large genomic deletions in zebrafish, from small domains to whole gene excisions

Tromp, Alisha, Robinson, Kate, Hall, Thomas E., Mowry, Bryan and Giacomotto, Jean (2021). Pipeline for generating stable large genomic deletions in zebrafish, from small domains to whole gene excisions. G3 Genes|Genomes|Genetics, 11 (12) jkab321. doi: 10.1093/g3journal/jkab321

Pipeline for generating stable large genomic deletions in zebrafish, from small domains to whole gene excisions

2021

Journal Article

Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish

Butti, Zoé, Pan, Yingzhou Edward, Giacomotto, Jean and Patten, Shunmoogum A. (2021). Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish. Communications Biology, 4 (1) 792, 1-16. doi: 10.1038/s42003-021-02302-y

Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish

2020

Journal Article

Modular transient nanoclustering of activated β2-adrenergic receptors revealed by single-molecule tracking of conformation-specific nanobodies

Gormal, Rachel S., Padmanabhan, Pranesh, Kasula, Ravikiran, Bademosi, Adekunle T., Coakley, Sean, Giacomotto, Jean, Blum, Ailisa, Joensuu, Merja, Wallis, Tristan P., Lo, Harriet P., Budnar, Srikanth, Rae, James, Ferguson, Charles, Bastiani, Michele, Thomas, Walter G., Pardon, Els, Steyaert, Jan, Yap, Alpha S., Goodhill, Geoffrey J., Hilliard, Massimo A., Parton, Robert G. and Meunier, Frédéric A. (2020). Modular transient nanoclustering of activated β2-adrenergic receptors revealed by single-molecule tracking of conformation-specific nanobodies. Proceedings of the National Academy of Sciences of the United States of America, 117 (48), 30476-30487. doi: 10.1073/pnas.2007443117

Modular transient nanoclustering of activated β2-adrenergic receptors revealed by single-molecule tracking of conformation-specific nanobodies

2020

Journal Article

Neurexins in autism and schizophrenia-a review of patient mutations, mouse models and potential future directions

Tromp, Alisha, Mowry, Bryan and Giacomotto, Jean (2020). Neurexins in autism and schizophrenia-a review of patient mutations, mouse models and potential future directions. Molecular Psychiatry, 26 (3), 747-760. doi: 10.1038/s41380-020-00944-8

Neurexins in autism and schizophrenia-a review of patient mutations, mouse models and potential future directions

2020

Journal Article

Characterisation of δ-Conotoxin TxVIA as a mammalian T-type calcium channel modulator

Wang, Dan, Himaya, S.W.A., Giacomotto, Jean, Hasan, Md. Mahadhi, Cardoso, Fernanda C., Ragnarsson, Lotten and Lewis, Richard J. (2020). Characterisation of δ-Conotoxin TxVIA as a mammalian T-type calcium channel modulator. Marine Drugs, 18 (7) 343, 1-13. doi: 10.3390/md18070343

Characterisation of δ-Conotoxin TxVIA as a mammalian T-type calcium channel modulator

2019

Journal Article

The α1-adrenoceptor inhibitor ρ-TIA facilitates net hunting in piscivorous Conus tulipa

Dutt, Mriga, Giacomotto, Jean, Ragnarsson, Lotten, Andersson, Åsa, Brust, Andreas, Dekan, Zoltan, Alewood, Paul F. and Lewis, Richard J. (2019). The α1-adrenoceptor inhibitor ρ-TIA facilitates net hunting in piscivorous Conus tulipa. Scientific Reports, 9 (1) 17841, 17841. doi: 10.1038/s41598-019-54186-y

The α1-adrenoceptor inhibitor ρ-TIA facilitates net hunting in piscivorous Conus tulipa

2019

Journal Article

Association of schizophrenia risk with disordered niacin metabolism in an Indian genome-wide association study

Periyasamy, Sathish, John, Sujit, Padmavati, Raman, Rajendren, Preeti, Thirunavukkarasu, Priyadarshini, Gratten, Jacob, Vinkhuyzen, Anna, McRae, Allan, Holliday, Elizabeth G., Nyholt, Dale R., Nancarrow, Derek, Bakshi, Andrew, Hemani, Gibran, Nertney, Deborah, Smith, Heather, Filippich, Cheryl, Patel, Kalpana, Fowdar, Javed, McLean, Duncan, Tirupati, Srinivasan, Nagasundaram, Arunkumar, Gundugurti, Prasad Rao, Selvaraj, Krishnamurthy, Jegadeesan, Jayaprakash, Jorde, Lynn B., Wray, Naomi R., Brown, Matthew A., Suetani, Rachel, Giacomotto, Jean ... Mowry, Bryan J. (2019). Association of schizophrenia risk with disordered niacin metabolism in an Indian genome-wide association study. JAMA Psychiatry, 76 (10), 1026-1034. doi: 10.1001/jamapsychiatry.2019.1335

Association of schizophrenia risk with disordered niacin metabolism in an Indian genome-wide association study

2017

Journal Article

Caveolae protect notochord cells against catastrophic mechanical failure during development

Lim, Ye-Wheen, Lo, Harriet P., Ferguson, Charles, Martel, Nick, Giacomotto, Jean, Gomez, Guillermo A., Yap, Alpha S., Hall, Thomas E. and Parton, Robert G. (2017). Caveolae protect notochord cells against catastrophic mechanical failure during development. Current Biology, 27 (13), 1968-1981.e7. doi: 10.1016/j.cub.2017.05.067

Caveolae protect notochord cells against catastrophic mechanical failure during development

2016

Journal Article

Developmental suppression of schizophrenia-associated miR-137 alters sensorimotor function in zebrafish

Giacomotto, J., Carroll, A. P., Rinkwitz, S., Mowry, B., Cairns, M. J. and Becker, T. S. (2016). Developmental suppression of schizophrenia-associated miR-137 alters sensorimotor function in zebrafish. Translational Psychiatry, 6 (5) e818, 1-10. doi: 10.1038/tp.2016.88

Developmental suppression of schizophrenia-associated miR-137 alters sensorimotor function in zebrafish

2016

Journal Article

Tissue-specific models of spinal muscular atrophy confirm a critical role of SMN in motor neurons from embryonic to adult stages

Laird, Angela S., Mackovski, Nikolce, Rinkwitz, Silke, Becker, Thomas S. and Giacomotto, Jean (2016). Tissue-specific models of spinal muscular atrophy confirm a critical role of SMN in motor neurons from embryonic to adult stages. Human Molecular Genetics, 25 (9) ddw044, 1728-1738. doi: 10.1093/hmg/ddw044

Tissue-specific models of spinal muscular atrophy confirm a critical role of SMN in motor neurons from embryonic to adult stages

2015

Journal Article

Copy number variants in patients with intellectual disability affect the regulation of ARX transcription factor gene

Ishibashi, Minaka, Manning, Elizabeth, Shoubridge, Cheryl, Krecsmarik, Monika, Hawkins, Thomas A, Giacomotto, Jean, Zhao, Ting, Mueller, Thomas, Bader, Patricia I., Cheung, Sau W., Stankiewicz, Pawel, Bain, Nicole L., Hackett, Anna, Reddy, Chilamakuri C. S., Mechaly, Alejandro S., Peers, Bernard, Wilson, Stephen W., Lenhard, Boris, Bally-Cuif, Laure, Gecz, Jozef, Becker, Thomas S. and Rinkwitz, Silke (2015). Copy number variants in patients with intellectual disability affect the regulation of ARX transcription factor gene. Human Genetics, 134 (11-12), 1163-1182. doi: 10.1007/s00439-015-1594-x

Copy number variants in patients with intellectual disability affect the regulation of ARX transcription factor gene

2015

Journal Article

Comparative Venomics Reveals the Complex Prey Capture Strategy of the Piscivorous Cone Snail Conus catus

Himaya S.W.A., Jin, Ai-Hua, Dutertre, Sebastien, Giacomotto, Jean, Mohialdeen, Hoshyar, Vetter, Irina, Alewood, Paul F. and Lewis, Richard J. (2015). Comparative Venomics Reveals the Complex Prey Capture Strategy of the Piscivorous Cone Snail Conus catus. Journal of Proteome Research, 14 (10), 4372-4381. doi: 10.1021/acs.jproteome.5b00630

Comparative Venomics Reveals the Complex Prey Capture Strategy of the Piscivorous Cone Snail Conus catus

2015

Journal Article

miR-124 Contributes to the functional maturity of microglia

Svahn, Adam J., Giacomotto, Jean, Graeber, Manuel B., Rinkwitz, Silke and Becker, Thomas S. (2015). miR-124 Contributes to the functional maturity of microglia. Developmental Neurobiology, 76 (5), 1-12. doi: 10.1002/dneu.22328

miR-124 Contributes to the functional maturity of microglia