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
Multidisciplinary Sciences
William J. Spencer, Nicholas F. Schneider, Tylor R. Lewis, Carson M. Castillo, Nikolai P. Skiba, Vadim Y. Arshavsky
Summary: The photoreceptor outer segment is a cilium filled with flat-tened disc membranes for light capture. The addition of new discs at the base of outer segments is driven by Arp2/3-mediated actin polymerization initiated by the pentameric WAVE complex. Knockout of WASF3, a subunit of the WAVE complex, disrupts actin polymerization and leads to disorganized membrane lamellae instead of a proper outer segment.
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA
(2023)
Article
Biochemistry & Molecular Biology
Nemanja Milicevic, Ouafa Ait-Hmyed Hakkari, Udita Bagchi, Cristina Sandu, Aldo Jongejan, Perry D. Moerland, Jacoline B. ten Brink, David Hicks, Arthur A. Bergen, Marie-Paule Felder-Schmittbuhl
Summary: The study showed that Per1 and Per2 are essential clock components for driving photoreceptor outer segment phagocytosis, and this process may be transcriptionally driven by the retinal pigment epithelium (RPE). No differentially expressed genes were found in photoreceptors, while several genes were differentially expressed in the RPE, both in wild type and Per1, Per2 double mutant mice. STRING analysis identified a group of interacting genes potentially involved in driving photoreceptor outer segment phagocytosis in the RPE.
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
Biology
Matthias Poege, Julia Mahamid, Sanae S. Imanishi, Juergen M. Plitzko, Krzysztof Palczewski, Wolfgang Baumeister
Summary: Studies have identified two molecular connectors/spacers in the membrane structure of rod outer segment (ROS) that likely contribute to the precise stacking of ROS disks. Additionally, a continuous supramolecular assembly composed of peripherin-2 (PRPH2) and rod outer segment membrane protein 1 (ROM1) oligomers enforces the extreme radius of curvature at the disk rims. Together, these molecular assemblies constitute the structural basis of the highly specialized ROS functional architecture.
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)
Review
Biochemistry & Molecular Biology
Ailis L. Moran, John D. Fehilly, Daniel Floss Jones, Ross Collery, Breandan N. Kennedy
Summary: Outer segment phagocytosis (OSP) is a regulated process in which photoreceptor outer segment tips are consumed by adjacent retinal pigment epithelium (RPE) cells. However, there is still limited knowledge about the key regulators of OSP in vivo. Understanding the regulation of OSP is crucial for the treatment of ocular diseases related to OSP.
Article
Cell Biology
Amanda Miles, Clarke Blair, Andrew Emili, Vincent Tropepe
Summary: This study discovered the association between the USH1 gene pcdh15b and retinopathy in zebrafish model, revealing that defects in rod and cone photoreceptors will lead to vision impairment.
DISEASE MODELS & MECHANISMS
(2021)
Article
Biochemistry & Molecular Biology
Matias Guerrero, Carlos Ruiz, Andres Romero, Luka Robeson, Diego Ruiz, Francisco Salinas
Summary: BcWCL1 and WC-1 proteins have similar domains, BcWCL1 can self-dimerize and activate transcription in yeast upon blue-light stimulation.
INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES
(2023)
Article
Biochemistry & Molecular Biology
Karolina Rassek, Katarzyna Izykowska, Magdalena Zurawek, Monika Pieniawska, Karina Nowicka, Xing Zhao, Grzegorz K. Przybylski
Summary: TMEM244 is a transmembrane protein that belongs to the TMEM family and is involved in various cellular processes. It has been identified as a diagnostic marker for Sezary syndrome. In this study, it was found that TMEM244 is a necessary long non-coding RNA for the growth of CTCL cells.
INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES
(2023)
Article
Cell Biology
Brittany J. Carr, Paloma Stanar, Orson L. Moritz
Summary: Mutations in prom1 and cdhr1 genes are associated with inherited retinal degenerative disorders. Prom1-null mutations result in severely dysmorphic photoreceptors while cdhr1-null photoreceptors do not appear grossly abnormal. Double-null mutants do not differ significantly from prom1-null mutants.
JOURNAL OF CELL SCIENCE
(2021)
Article
Multidisciplinary Sciences
Iva Filova, Kateryna Pysanenko, Mitra Tavakoli, Simona Vochyanova, Martina Dvorakova, Romana Bohuslavova, Ondrej Smolik, Valeria Fabriciova, Petra Hrabalova, Sarka Benesova, Lukas Valihrach, Jiri Cerny, Ebenezer N. Yamoah, Josef Syka, Bernd Fritzsch, Gabriela Pavlinkova
Summary: ISL1 is found to regulate the molecular and cellular features of auditory neurons, including the formation of the auditory map and spiral ganglion development. Loss of Isl1 leads to abnormal auditory pathway, but auditory processing features are preserved, revealing the plasticity of the central auditory pathway.
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA
(2022)
Article
Developmental Biology
Sumitha Prameela Bharathan, Angela Ferrario, Kayla Stepanian, G. Esteban Fernandez, Mark W. Reid, Justin S. Kim, Chloe Hutchens, Narine Harutyunyan, Carolyn Marks, Matthew E. Thornton, Brendan H. Grubbs, David Cobrinik, Jennifer G. Aparicio, Aaron Nagiel
Summary: This study establishes a pseudo-temporal staging system for the development of the first synapse in the visual system in the human retina, termed OPL-Stages 0 to 4. By studying the maturation and organization of photoreceptors, depolarizing bipolar cells, and horizontal cells, the study validates this staging system through precise subcellular localization analysis of bassoon. Additionally, the study demonstrates the utility of human retinal organoids (HROs) derived from human embryonic and induced pluripotent stem cells in replicating key aspects of human photoreceptor-bipolar cell synaptogenesis in vitro.
Article
Neurosciences
Veronica T. Cheli, Diara A. Santiago Gonzalez, Rensheng Wan, Shaina L. Rosenblum, Giancarlo E. Denaroso, Christina G. Angeliu, Zachary Smith, Congying Wang, Pablo M. Paez
Summary: By disrupting the expression of the Tf receptor gene in oligodendrocyte progenitor cells, researchers found that the Tf cycle is uniquely important for the development and function of oligodendrocytes. Deleting the Tfr gene resulted in impaired iron absorption and hypomyelination in mice. The Tf cycle plays a crucial role in iron homeostasis and energy production in oligodendrocyte progenitor cells.
JOURNAL OF NEUROSCIENCE
(2023)
Article
Multidisciplinary Sciences
Rebecca L. Belmonte, Isabella L. Engbretson, Jung-Hyun Kim, Illiana Cajias, Eun-Young Erin Ahn, David L. Stachura
Summary: The gene SON located on human chromosome 21 is associated with hematopoietic disorders in Down syndrome. Reduction of SON expression in zebrafish resulted in abnormal development of blood cells, indicating the essential role of SON in hematopoiesis. These findings suggest that SON influences differentiation of the immune system and further studies on molecular pathways can provide insights into vertebrate HSPC generation.
Article
Developmental Biology
Sarika Tiwari, Subramanian Dharmarajan, Mahesh Shivanna, Deborah C. Otteson, Teri L. Belecky-Adams
BMC DEVELOPMENTAL BIOLOGY
(2014)
Article
Developmental Biology
Nandini Rao, Fengyu Song, Deepali Jhamb, Mu Wang, Derek J. Milner, Nathaniel M. Price, Teri L. Belecky-Adams, Mathew J. Palakal, Jo Ann Cameron, Bingbing Li, Xiaoping Chen, David L. Stocum
BMC DEVELOPMENTAL BIOLOGY
(2014)
Article
Developmental Biology
Rachna Sehgal, Rachel Karcavich, Scott Carlson, Teri L. Belecky-Adams
DEVELOPMENTAL BIOLOGY
(2008)
Article
Developmental Biology
Rachna Sehgal, Nader Sheibani, Simon J. Rhodes, Teri L. Belecky Adams
DEVELOPMENTAL BIOLOGY
(2009)
Article
Developmental Biology
Jonathan M. Wilson, Reyna I. Martinez-De Luna, Heithem M. El Hodiri, Rosamund Smith, Michael W. King, Anthony L. Mescher, Anton W. Neff, Teri L. Belecky-Adams
GENE EXPRESSION PATTERNS
(2010)
Article
Endocrinology & Metabolism
Subramanian Dharmarajan, Casandra Carrillo, Zhonghua Qi, Jonathan M. Wilson, Anthony J. Baucum II, Christine M. Sorenson, Nader Sheibani, Teri L. Belecky-Adams
Summary: This study aimed to investigate the impact of chronic inflammation on the loss of pericytes in early diabetic retinopathy. The results showed that both type 1 and type 2 diabetes led to similar progression of retinal pericyte loss and gliosis. However, the expression of inflammatory factors differed between the two diabetes models, with peak expression occurring at different ages. Additionally, retinal vascular changes were more severe in the type 2 diabetes model. Chronic exposure of retinal pericytes to IFN gamma resulted in decreased PDGFR beta signaling and increased levels of active PKC delta and CC3, indicating increased pericyte apoptosis.
Article
Biochemistry & Molecular Biology
Subramanian Dharmarajan, Zafer Gurel, Shoujian Wang, Christine M. Sorenson, Nader Sheibani, Teri L. Belecky-Adams
Article
Developmental Biology
Jonathan M. Wilson, Kazuna Sato, Ellen A. G. Chernoff, Teri L. Belecky-Adams
GENE EXPRESSION PATTERNS
(2007)
Article
Ophthalmology
Rachna Sehgal, Dirk J. Andres, Ruben Adler, Teri L. Belecky-Adams
INVESTIGATIVE OPHTHALMOLOGY & VISUAL SCIENCE
(2006)
Article
Ophthalmology
SL Fong, MH Criswell, T Belecky-Adams, WB Fong, JN McClintick, WW Kao, HJ Edenberg
EXPERIMENTAL EYE RESEARCH
(2005)
Article
Neurosciences
T Belecky-Adams, M Holmes, YQ Shan, CS Tedesco, C Mascari, A Kaul, DC Wight, RE Morris, M Sussman, J Diamond, LM Parysek
JOURNAL OF NEUROSCIENCE
(2003)
Article
Developmental Biology
TL Belecky-Adams, R Adler, DC Beebe
Article
Developmental Biology
R Adler, TL Belecky-Adams
Article
Developmental Biology
R Adler, A Tamres, RL Bradford, TL Belecky-Adams
DEVELOPMENTAL BIOLOGY
(2001)