Article
Neurosciences
Tylor R. Lewis, Mustafa S. Makia, Carson M. Castillo, Muayyad R. Al-Ubaidi, Muna Naash, Vadim Y. Arshavsky
Summary: Mutations in the PRPH2 gene cause a broad range of retinal diseases by affecting the structure of outer segment of photoreceptor cells, resulting in irregular disc enclosure process. Abnormal PRPH2 oligomerization leads to defects in outer segment structure, particularly at the stage of disc enclosure.
JOURNAL OF NEUROSCIENCE
(2021)
Article
Multidisciplinary Sciences
Dianlei Guo, Jiali Ru, Lijing Xie, Mingjuan Wu, Yingchun Su, Shiyong Zhu, Shujuan Xu, Bin Zou, Yanhong Wei, Xialin Liu, Yizhi Liu, Chunqiao Liu
Summary: The study discovered that Tmem138 plays a crucial role in the functional organization of photoreceptor connecting cilium (CC), which is essential for rhodopsin localization and outer segment (OS) biogenesis. Inactivation of Tmem138 leads to abnormal development of OS and degeneration of photoreceptor cells.
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA
(2022)
Article
Neurosciences
Jillian N. Pearring, Jorge Martinez-Marquez, Jason R. Willer, Eric C. Lieu, Raquel Y. Salinas, Vadim Y. Arshavsky
Summary: The study found that the targeted delivery of the rod cyclic nucleotide-gated (CNG) channel to the outer segment uses the conventional secretory pathway and requires preassembly of its constituent alpha 1 and beta 1 subunits. The N-terminal glutamic acid-rich protein domain of CNG beta 1 encodes specific information targeting the channel to rod outer segments and connects the channel to photoreceptor disk rims through an interaction with peripherin-2. These findings reveal fine functional specializations within the CNG channel's structural domains and suggest that its sequestration to the outer segment plasma membrane requires an interaction with peripherin-2.
JOURNAL OF NEUROSCIENCE
(2021)
Article
Chemistry, Multidisciplinary
Jie Ran, Yao Zhang, Sai Zhang, Haixia Li, Liang Zhang, Qingchao Li, Juan Qin, Dengwen Li, Lei Sun, Songbo Xie, Xiaomin Zhang, Lin Liu, Min Liu, Jun Zhou
Summary: Retinopathy of prematurity (ROP) is a major cause of childhood visual impairment and blindness. Histone deacetylase 6 (HDAC6)-mediated disassembly of photoreceptor cilia has been identified as an early event in the pathogenesis of ROP. Enhancing the expression of HDAC6 or blocking HDAC6-mediated cilium disassembly can protect mice from ROP-associated retinal defects.
Article
Biochemistry & Molecular Biology
Yunlong Zhang, Ying Liu, Yu Dai, Yazhe Ren, Guangsen Bao, Bo Ai, Yu Jiang
Summary: Folliculin (FLCN) is a tumor suppressor protein that localizes to primary cilia in ciliated cells to control mTORC1 signaling, and this localization is crucial for its function in suppressing abnormal signaling activities and anchorage-independent growth in tumor cells.
Editorial Material
Biochemistry & Molecular Biology
David J. Simon, Joshua Levitz
Summary: This study reveals a new type of synapse that enables chemical transmission between serotonergic axons and the primary cilia of hippocampal neurons, providing insights into the role of the primary cilium in neuromodulatory signaling.
Review
Cell Biology
Laura Sanchez-Bellver, Vasileios Toulis, Gemma Marfany
Summary: Ciliopathies are a group of heterogeneous inherited disorders caused by dysfunction of the cilium, affecting cells in multiple organs. The unique feature of daily renewal in photoreceptor outer segments and the efficient transport of proteins in ciliary transport are crucial for photoreceptor survival.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2021)
Article
Cell Biology
Siebren Faber, Stef J. F. Letteboer, Katrin Junger, Rossano Butcher, Trinadh V. Satish Tammana, Sylvia E. C. van Beersum, Marius Ueffing, Rob W. J. Collin, Qin Liu, Karsten Boldt, Ronald Roepman
Summary: Mutations in PDE6D disrupt the function of its cognate protein and lead to Joubert Syndrome and retinal degeneration. Through affinity proteomics, we identified a set of interacting proteins involved in photoreceptor integrity, GTPase activity, nuclear import, and ubiquitination. We also discovered novel ciliary cargo proteins of PDE6D, including FAM219A, NIM1K, and UBL3.
Article
Cell Biology
Julio C. Corral-Serrano, Paul E. Sladen, Daniele Ottaviani, Olivia F. Rezek, Dimitra Athanasiou, Katarina Jovanovic, Jacqueline van der Spuy, Brian C. Mansfield, Michael E. Cheetham
Summary: This study demonstrates the potential therapeutic effect of eupatilin on CEP290-associated retinal diseases. Eupatilin improves cilia formation and length, reduces rhodopsin retention, and modulates gene expression in retinal organoids.
Review
Cell Biology
Bo Wang, Zheyong Liang, Peijun Liu
Summary: This article reviews the role of the primary cilium in cancer, including its architecture, signaling pathways, cilia assembly and disassembly regulators, summarizing new findings in tumor microenvironments and different cancers, highlighting novel possibilities for therapeutic targets in cancer.
JOURNAL OF CELLULAR PHYSIOLOGY
(2021)
Article
Cell Biology
Farah Haque, Christian Freniere, Qiong Ye, Nandini Mani, Elizabeth M. Wilson-Kubalek, Pei- Ku, Ronald A. Milligan, Radhika Subramanian
Summary: This article uncovers the mechanism of GLI tethering to microtubules through its interaction with ciliary kinesin KIF7. The coiled-coil dimerization domain of KIF7 forms a complex with the DNA-binding zinc fingers in GLI, increasing GLI's affinity for microtubules and modulating their localization to the cilium tip.
NATURE CELL BIOLOGY
(2022)
Article
Neurosciences
Kennedy Bolstad, Inigo Novales Flamarique
Summary: Fishes can quickly modulate their photoreceptor properties to optimize visual function in response to different light environments. In this study, Atlantic halibut larvae and juveniles exposed to blue light showed changes in cone opsin gene expression and photoreceptor structure, indicating rapid plasticity as a function of developmental stage. These adjustments help the fish perceive achromatic or chromatic contrasts in accordance with their ecological needs.
JOURNAL OF COMPARATIVE NEUROLOGY
(2023)
Article
Cell Biology
Lyudmila Dimitrova-Paternoga, Pravin Kumar Ankush Jagtap, Anna Cyrklaff, Vaishali, Karine Lapouge, Peter Sehr, Kathryn Perez, Simone Heber, Christian Low, Janosch Hennig, Anne Ephrussi
Summary: Kinesin-1 transports cargos within cells by forming a complex with atypical tropomyosin, aTm1, and a study on the Khch-aTm1 complex reveals the mechanism of this interaction. Additionally, aTm1 plays a stabilizing role in the interaction of Khc with RNA, distinguishing it from classical motor adaptors.
GENES & DEVELOPMENT
(2021)
Article
Multidisciplinary Sciences
Winna Siti, Hon Lin Too, Tommy Anderson, Xiao Rui Liu, Iong Ying Loh, Zhisong Wang
Summary: Researchers have developed an autonomous non-bridge-burning DNA motor and successfully integrated it with a triangle DNA origami, enabling high-speed and effective translocation on the origami, including sharp turns. The DNA motor-origami system has complex motion capabilities, offering potential for autonomous or scaled-up applications.
Article
Cell Biology
Jessica A. Cross, Derek. N. Woolfson, Mark P. Dodding
Summary: Researchers characterized the first structural interface between KHC and cargo adaptor, revealing a heterotrimeric coiled-coil complex that enhances direct binding between KHC and RNA. This finding demonstrates the structural plasticity of the KHC tail as a platform for protein-protein interactions and how a cargo adaptor protein can modify a motor-RNA interface to promote transport.
GENES & DEVELOPMENT
(2021)
Article
Genetics & Heredity
Niedharsan Pooranachandran, Jarema J. Malicki
Article
Biochemistry & Molecular Biology
Meriam Boubakri, Taro Chaya, Hiromi Hirata, Naoko Kajimura, Ryusuke Kuwahara, Akiko Ueno, Jarema Malicki, Takahisa Furukawa, Yoshihiro Omori
JOURNAL OF BIOLOGICAL CHEMISTRY
(2016)
Article
Genetics & Heredity
Niedharsan Pooranachandran, Jarema J. Malicki
Article
Genetics & Heredity
Khodor Hazime, Jarema J. Malicki
Review
Cell Biology
Jarema J. Malicki, Colin A. Johnson
TRENDS IN CELL BIOLOGY
(2017)
Article
Genetics & Heredity
Khodor Hazime, Jarema J. Malicki
Article
Biochemistry & Molecular Biology
Chengtian Zhao, Jarema Malicki
Article
Cell Biology
Daqing Jin, Terri T. Ni, Jianjian Sun, Haiyan Wan, Jeffrey D. Amack, Guangju Yu, Jonathan Fleming, Chin Chiang, Wenyan Li, Anna Papierniak, Satish Cheepala, Gwenaelle Conseil, Susan P. C. Cole, Bin Zhou, Iain A. Drummond, John D. Schuetz, Jarema Malicki, Tao P. Zhong
NATURE CELL BIOLOGY
(2014)
Article
Neurosciences
Seonhee Kim, Maria K. Lehtinen, Alessandro Sessa, Mauro W. Zappaterra, Seo-Hee Cho, Dilenny Gonzalez, Brigid Boggan, Christina A. Austin, Jan Wijnholds, Michael J. Gambello, Jarema Malicki, Anthony S. LaMantia, Vania Broccoli, Christopher A. Walsh
Article
Multidisciplinary Sciences
Chengtian Zhao, Yoshihiro Omori, Katarzyna Brodowska, Peter Kovach, Jarema Malicki
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA
(2012)
Review
Biochemistry & Molecular Biology
Jarema Malicki, Tomer Avidor-Reiss
Article
Genetics & Heredity
Andrei Avanesov, Shawn M. Honeyager, Jarema Malicki, Seth S. Blair
Article
Cell Biology
Xiaoming Fang, Andrew A. Peden, Fredericus J. M. van Eeden, Jarema J. Malicki
Summary: Opsin plays a crucial role in vertebrate photoreceptors by being transported to the outer segment for its function. Studies have shown that there are differences in the transport mechanisms of opsins in different species, which may be related to changes in amino acid sequences.
JOURNAL OF CELL SCIENCE
(2021)
Article
Ophthalmology
Jill Beyer, Xinping C. Zhao, Richard Yee, Shagufta Khaliq, Timothy T. McMahon, Hongyu Ying, Beatrice Y. J. T. Yue, Jarema J. Malicki
INVESTIGATIVE OPHTHALMOLOGY & VISUAL SCIENCE
(2010)
Article
Neurosciences
Ryan P. Silk, Hanagh R. Winter, Ouria Dkhissi-Benyahya, Carmella Evans -Molina, Alan W. Stitt, Vijay K. Tiwari, David A. Simpson, Eleni Beli
Summary: This study investigates whether diabetes affects the daily rhythm of gene expression in the retina. The results show that diabetic mice exhibited phase advancement in the expression of certain genes compared to non-diabetic mice. The study also identified oxygen-sensing mechanisms and HIF1alpha as potential upstream regulators. These findings provide important insights into the development of diabetic retinopathy.
Article
Neurosciences
Krishnamachari S. Prahalad, Daniel R. Coates
Summary: Visual stimuli presented around the time of a saccade can be perceived differently by the visual system, including a reduction in the harmful impact of flankers. This study investigated the effects of microsaccades on crowded stimuli placed 20 arc minutes from the center of gaze. The findings suggest two separate pre-saccadic benefits, one that regularizes the crowding zone and another that specifically benefits microsaccade targets surrounded by tangential flankers.
Article
Neurosciences
Chandrika Ravisankar, Christopher W. Tyler, Clifton M. Schor, Shrikant R. Bharadwaj
Summary: This study revealed that less than one-third of adults with normal binocular vision were able to successfully free-fuse random-dot image pairs and identify the embedded stereoscopic shapes. The successful participants showed a dissociation of vergence and accommodative responses, while the unsuccessful ones either exhibited strong vergence and accommodation or weak vergence and strong accommodation. Task performance of the unsuccessful cluster improved significantly with pharmacological paralysis of accommodation. A minority of participants also learned to dissociate one direction of their vergence and accommodation crosslinks with repeated free-fusion trials, optimizing their task performance.