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October 2017

Mutations in the gene OCRL are known to cause Lowe Syndrome, though it has remained unclear how loss of OCRL causes the diverse eye, kidney, and neurological symptoms suffered by patients. Here, Del Signore et al. show that in Drosophila, loss of OCRL causes activation of macrophage-like immune cells by disrupting late and recycling endosomal traffic, suggesting that neuroinflammation may be a cause of seizures in Lowe Syndrome patients. The image shows a color-coded time series projection of a four-minute timelapse movie of GFP-Rab5 in primary Drosophila immune cells. Multicolor tracks demonstrate motility of individual Rab5-positive endosomes. See Del Signore et al.

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Image Credit: Steven Del Signore, Brandeis University

Viewpoints

Guidelines for morpholino use in zebrafish

Didier Y. R. Stainier, Erez Raz, Nathan D. Lawson, Stephen C. Ekker, Rebecca D. Burdine, Judith S. Eisen, Philip W. Ingham, Stefan Schulte-Merker, Deborah Yelon, Brant M. Weinstein, Mary C. Mullins, Stephen W. Wilson, Lalita Ramakrishnan, Sharon L. Amacher, Stephan C. F. Neuhauss, Anming Meng, Naoki Mochizuki, Pertti Panula, Cecilia B. Moens

Research Articles

Genetic anticipation in Swedish Lynch syndrome families

Jenny von Salomé, Philip S. Boonstra, Masoud Karimi, Gustav Silander, Marie Stenmark-Askmalm, Samuel Gebre-Medhin, Christos Aravidis, Mef Nilbert, Annika Lindblom, Kristina Lagerstedt-Robinson

AtHKT1 drives adaptation of Arabidopsis thaliana to salinity by reducing floral sodium content

Dong An, Jiu-Geng Chen, Yi-Qun Gao, Xiang Li, Zhen-Fei Chao, Zi-Ru Chen, Qian-Qian Li, Mei-Ling Han, Ya-Ling Wang, Yong-Fei Wang, Dai-Yin Chao

A dominant-negative mutant inhibits multiple prion variants through a common mechanism

Fen Pei, Susanne DiSalvo, Suzanne S. Sindi, Tricia R. Serio

Broadening the functionality of a J-protein/Hsp70 molecular chaperone system

Brenda A. Schilke, Szymon J. Ciesielski, Thomas Ziegelhoffer, Erina Kamiya, Marco Tonelli, Woonghee Lee, Gabriel Cornilescu, Justin K. Hines, John L. Markley, Elizabeth A. Craig

Common, low-frequency, and rare genetic variants associated with lipoprotein subclasses and triglyceride measures in Finnish men from the METSIM study

James P. Davis, Jeroen R. Huyghe, Adam E. Locke, Anne U. Jackson, Xueling Sim, Heather M. Stringham, Tanya M. Teslovich, Ryan P. Welch, Christian Fuchsberger, Narisu Narisu, Peter S. Chines, Antti J. Kangas, Pasi Soininen, Mika Ala-Korpela, Johanna Kuusisto, Francis S. Collins, Markku Laakso, Michael Boehnke, Karen L. Mohlke

Non-muscle myosin IIB (Myh10) is required for epicardial function and coronary vessel formation during mammalian development

Liam A. Ridge, Karen Mitchell, Ali Al-Anbaki, Wasay Mohiuddin Shaikh Qureshi, Louise A. Stephen, Gennadiy Tenin, Yinhui Lu, Irina-Elena Lupu, Christopher Clowes, Abigail Robertson, Emma Barnes, Jayne A. Wright, Bernard Keavney, Elisabeth Ehler, Simon C. Lovell, Karl E. Kadler, Kathryn E. Hentges

A trehalose biosynthetic enzyme doubles as an osmotic stress sensor to regulate bacterial morphogenesis

Ximing Chen, Lizhe An, Xiaochuan Fan, Furong Ju, Binglin Zhang, Haili Sun, Jianxi Xiao, Wei Hu, Tao Qu, Liping Guan, Shukun Tang, Tuo Chen, Guangxiu Liu, Paul Dyson

A systems level approach to temporal expression dynamics in Drosophila reveals clusters of long term memory genes

Julianna Bozler, Balint Z. Kacsoh, Hao Chen, William E. Theurkauf, Zhiping Weng, Giovanni Bosco

Quantifying connectivity between local Plasmodium falciparum malaria parasite populations using identity by descent

Aimee R. Taylor, Stephen F. Schaffner, Gustavo C. Cerqueira, Standwell C. Nkhoma, Timothy J. C. Anderson, Kanlaya Sriprawat, Aung Pyae Phyo, François Nosten, Daniel E. Neafsey, Caroline O. Buckee

A bacterial toxin-antitoxin module is the origin of inter-bacterial and inter-kingdom effectors of Bartonella

Alexander Harms, Marius Liesch, Jonas Körner, Maxime Québatte, Philipp Engel, Christoph Dehio

The GYF domain protein PSIG1 dampens the induction of cell death during plant-pathogen interactions

Hidenori Matsui, Yuko Nomura, Mayumi Egusa, Takahiro Hamada, Gang-Su Hyon, Hironori Kaminaka, Yuichiro Watanabe, Takashi Ueda, Marco Trujillo, Ken Shirasu, Hirofumi Nakagami

Rnr1, but not Rnr3, facilitates the sustained telomerase-dependent elongation of telomeres

André Maicher, Inbal Gazy, Sushma Sharma, Lisette Marjavaara, Gilad Grinberg, Keren Shemesh, Andrei Chabes, Martin Kupiec

Involvement of DNA mismatch repair in the maintenance of heterochromatic DNA stability in Saccharomyces cerevisiae

Basanta K. Dahal, Lyudmila Y. Kadyrova, Kristin R. Delfino, Igor B. Rogozin, Vaibhavi Gujar, Kirill S. Lobachev, Farid A. Kadyrov

RhoGEF9 splice isoforms influence neuronal maturation and synapse formation downstream of α2 GABAA receptors

Claire de Groot, Amalia Floriou-Servou, Yuan-Chen Tsai, Simon Früh, Manuela Kohler, Georgia Parkin, Cornelia Schwerdel, Giovanna Bosshard, Kai Kaila, Jean-Marc Fritschy, Shiva K. Tyagarajan

Functional convergence of histone methyltransferases EHMT1 and KMT2C involved in intellectual disability and autism spectrum disorder

Tom S. Koemans, Tjitske Kleefstra, Melissa C. Chubak, Max H. Stone, Margot R. F. Reijnders, Sonja de Munnik, Marjolein H. Willemsen, Michaela Fenckova, Connie T. R. M. Stumpel, Levinus A. Bok, Margarita Sifuentes Saenz, Kyna A. Byerly, Linda B. Baughn, Alexander P. A. Stegmann, Rolph Pfundt, Huiqing Zhou, Hans van Bokhoven, Annette Schenck, Jamie M. Kramer

Genetic modifiers of muscular dystrophy act on sarcolemmal resealing and recovery from injury

Mattia Quattrocelli, Joanna Capote, Joyce C. Ohiri, James L. Warner, Andy H. Vo, Judy U. Earley, Michele Hadhazy, Alexis R. Demonbreun, Melissa J. Spencer, Elizabeth M. McNally

G protein-coupled estrogen receptor regulates embryonic heart rate in zebrafish

Shannon N. Romano, Hailey E. Edwards, Jaclyn Paige Souder, Kevin J. Ryan, Xiangqin Cui, Daniel A. Gorelick

Profiling RNA-Seq at multiple resolutions markedly increases the number of causal eQTLs in autoimmune disease

Christopher A. Odhams, Deborah S. Cunninghame Graham, Timothy J. Vyse

Role of Neuropilin-1/Semaphorin-3A signaling in the functional and morphological integrity of the cochlea

Pezhman Salehi, Marshall X. Ge, Usha Gundimeda, Leah Michelle Baum, Homero Lael Cantu, Joel Lavinsky, Litao Tao, Anthony Myint, Charlene Cruz, Juemei Wang, Angeliki Maria Nikolakopoulou, Carolina Abdala, Matthew William Kelley, Takahiro Ohyama, Thomas Matthew Coate, Rick A. Friedman

BBSome function is required for both the morphogenesis and maintenance of the photoreceptor outer segment

Ying Hsu, Janelle E. Garrison, Gunhee Kim, Addison R. Schmitz, Charles C. Searby, Qihong Zhang, Poppy Datta, Darryl Y. Nishimura, Seongjin Seo, Val C. Sheffield

Defective replication initiation results in locus specific chromosome breakage and a ribosomal RNA deficiency in yeast

Joseph C. Sanchez, Elizabeth X. Kwan, Thomas J. Pohl, Haley M. Amemiya, M. K. Raghuraman, Bonita J. Brewer

The homeodomain-interacting protein kinase HPK-1 preserves protein homeostasis and longevity through master regulatory control of the HSF-1 chaperone network and TORC1-restricted autophagy in Caenorhabditis elegans

Ritika Das, Justine A. Melo, Manjunatha Thondamal, Elizabeth A. Morton, Adam B. Cornwell, Beresford Crick, Joung Heon Kim, Elliot W. Swartz, Todd Lamitina, Peter M. Douglas, Andrew V. Samuelson

BAK1 is involved in AtRALF1-induced inhibition of root cell expansion

Keini Dressano, Paulo H. O. Ceciliato, Aparecida L. Silva, Juan Carlos Guerrero-Abad, Tábata Bergonci, Fausto Andrés Ortiz-Morea, Marco Bürger, Marcio C. Silva-Filho, Daniel S. Moura

dOCRL maintains immune cell quiescence by regulating endosomal traffic

Steven J. Del Signore, Sarah A. Biber, Katherine S. Lehmann, Stephanie R. Heimler, Benjamin H. Rosenfeld, Tania L. Eskin, Sean T. Sweeney, Avital A. Rodal

Condensin II and GAIT complexes cooperate to restrict LINE-1 retrotransposition in epithelial cells

Jacqueline R. Ward, Kommireddy Vasu, Emily Deutschman, Dalia Halawani, Peter A. Larson, Dongmei Zhang, Belinda Willard, Paul L. Fox, John V. Moran, Michelle S. Longworth

Alx4 relays sequential FGF signaling to induce lacrimal gland morphogenesis

Ankur Garg, Mukesh Bansal, Noriko Gotoh, Gen-Sheng Feng, Jian Zhong, Fen Wang, Ariana Kariminejad, Steven Brooks, Xin Zhang

Environmental perturbations lead to extensive directional shifts in RNA processing

Allison L. Richards, Donovan Watza, Anthony Findley, Adnan Alazizi, Xiaoquan Wen, Athma A. Pai, Roger Pique-Regi, Francesca Luca

Combinatorial action of Grainyhead, Extradenticle and Notch in regulating Hox mediated apoptosis in Drosophila larval CNS

Risha Khandelwal, Rashmi Sipani, Sriivatsan Govinda Rajan, Raviranjan Kumar, Rohit Joshi

Loss of Xist RNA from the inactive X during B cell development is restored in a dynamic YY1-dependent two-step process in activated B cells

Camille M. Syrett, Vishal Sindhava, Suchita Hodawadekar, Arpita Myles, Guanxiang Liang, Yue Zhang, Satabdi Nandi, Michael Cancro, Michael Atchison, Montserrat C. Anguera

Role of UHRF1 in de novo DNA methylation in oocytes and maintenance methylation in preimplantation embryos

Shoji Maenohara, Motoko Unoki, Hidehiro Toh, Hiroaki Ohishi, Jafar Sharif, Haruhiko Koseki, Hiroyuki Sasaki

The creation and selection of mutations resistant to a gene drive over multiple generations in the malaria mosquito

Andrew M. Hammond, Kyros Kyrou, Marco Bruttini, Ace North, Roberto Galizi, Xenia Karlsson, Nace Kranjc, Francesco M. Carpi, Romina D’Aurizio, Andrea Crisanti, Tony Nolan

A C-terminally truncated form of β-catenin acts as a novel regulator of Wnt/β-catenin signaling in planarians

Hanxia Su, Miquel Sureda-Gomez, Neus Rabaneda-Lombarte, Maria Gelabert, Jianlei Xie, Wei Wu, Teresa Adell

MiR-125a Is a critical modulator for neutrophil development

Yuting Qin, Lingling Wu, Ye Ouyang, Ping Zhou, Haibo Zhou, Yan Wang, Jianyang Ma, Jinsong Zhang, Yanan Chen, Jie Qian, Yuanjia Tang, Nan Shen

Dopamine negatively modulates the NCA ion channels in C. elegans

Irini Topalidou, Kirsten Cooper, Laura Pereira, Michael Ailion

SNPs near the cysteine proteinase cathepsin O gene (CTSO) determine tamoxifen sensitivity in ERα-positive breast cancer through regulation of BRCA1

Junmei Cairns, James N. Ingle, Lawrence D. Wickerham, Richard Weinshilboum, Mohan Liu, Liewei Wang

Division-induced DNA double strand breaks in the chromosome terminus region of Escherichia coli lacking RecBCD DNA repair enzyme

Anurag Kumar Sinha, Adeline Durand, Jean-Michel Desfontaines, Ielyzaveta Iurchenko, Hélène Auger, David R. F. Leach, François-Xavier Barre, Bénédicte Michel