Article
Biology
Tuo Shi, Marielle O. Beaulieu, Lauren M. Saunders, Peter Fabian, Cole Trapnell, Neil Segil, J. Gage Crump, David W. Raible
Summary: A single-cell RNA sequencing of zebrafish inner ear reveals the diversity of hair cells and non-sensory supporting cells. The study identifies a putative progenitor population for hair cells and supporting cells, as well as distinct hair and supporting cell types in different parts of the inner ear. The similarities between fish and mammals highlight the relevance of zebrafish as a model for studying inner ear-specific hair cell function and regeneration.
Article
Neurosciences
Melanie Holmgren, Lavinia Sheets
Summary: This study found that Mitochondrial Inner Membrane Protein 17 (Mpv17) plays an important role in maintaining hair-cell mitochondrial homeostasis. Zebrafish mutants with mpv17(a9/a9) displayed impaired mitochondrial function and were more vulnerable to morphological disruption.
FRONTIERS IN CELLULAR NEUROSCIENCE
(2021)
Article
Toxicology
Weihao Shao, Dan Zhong, Haowei Jiang, Yujie Han, Yu Yin, Ruining Li, Xiuping Qian, Daijie Chen, Lili Jing
Summary: The study compared the toxic effects of three aminoglycoside antibiotics on zebrafish embryos, revealing that etimicin and amikacin exhibit more developmental toxicities to young embryos but show reduced toxicities to kidney and neuromast hair cells at subtoxic doses. It was further demonstrated that etimicin is a new aminoglycoside with reduced risks of nephrotoxicity and ototoxicity compared to gentamicin.
JOURNAL OF APPLIED TOXICOLOGY
(2021)
Article
Chemistry, Multidisciplinary
Sunho Park, Yeon Ju Kim, Harshita Sharma, Dream Kim, Yonghyun Gwon, Woochan Kim, Sangbae Park, Cheol Woo Ha, Yun-Hoon Choung, Jangho Kim
Summary: In this study, the use of graphene oxide (GO) as a nanomaterial-based approach for enhancing inner ear organoids (IEOs) was proposed. GO was found to promote hair cell formation and show potential applications in drug testing. This nanomaterial-based approach may offer a reliable and effective method for building better IEOs in the future.
Article
Audiology & Speech-Language Pathology
Derek J. Sauer, Kara E. Yopak, Craig A. Radford
Summary: Studies on the auditory system of fishes provide important information about the early evolution of vertebrate hearing. This study used immunohistochemistry and fluorescence microscopy to quantify the sensory epithelia in the inner ear of elasmobranchs. The results showed that macular area and the number of hair cells increased significantly throughout ontogeny, while hair cell density displayed a concurrent ontogenetic decrease.
Article
Neurosciences
Yi Pan, Shuting Li, Shunji He, Guangqin Wang, Chao Li, Zhiyong Liu, Mingliang Xiang
Summary: The study generated a new knock-in strain Fgf8(GFP/+) that accurately reflects the expression of Fgf8 and can be used to obtain high-purity embryonic and neonatal inner hair cells. Furthermore, the study revealed that inner hair cells can also be derived from inner ear progenitors expressing Insm1, a marker previously regarded as specific to outer hair cells.
NEUROSCIENCE BULLETIN
(2023)
Review
Cell Biology
Mingxuan Wu, Mingyu Xia, Wenyan Li, Huawei Li
Summary: Single-cell sequencing technology has enabled researchers to analyze gene expression variations in different cell types within the inner ear, leading to the discovery of tissue heterogeneity, novel cell subtypes, unique cell markers, and dynamic signaling pathways during development. These advancements offer new possibilities for inner ear research, with the vast potential of the technology yet to be fully explored.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2021)
Article
Multidisciplinary Sciences
Lorenzo Rella, Euclides E. Fernandes Povoa, Jonas Mars, Annabel L. P. Ebbing, Luc Schoppink, Marco C. Betist, Hendrik C. Korswagen
Summary: Members of the Wnt family play a crucial role in regulating cell migration through distinct canonical and noncanonical signaling pathways. The switch from noncanonical to canonical Wnt signaling in Caenorhabditis elegans provides a model to study the mechanism of the functional interplay between these pathways. The study shows that antagonistic regulation of Rho GTPases is involved in the cross-talk between noncanonical and canonical Wnt signaling pathways.
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA
(2021)
Article
Neurosciences
Katherine Hardy, Ana E. Amariutei, Francesca De Faveri, Aenea Hendry, Walter Marcotti, Federico Ceriani
Summary: The study explores hair-cell regeneration in the zebrafish lateral line after exposure to copper, finding that regeneration is faster in early larval stages. Afferent input plays a key role in driving the regeneration process of neuromasts.
JOURNAL OF PHYSIOLOGY-LONDON
(2021)
Review
Biochemistry & Molecular Biology
Maurizio Cortada, Soledad Levano, Daniel Bodmer
Summary: Hearing loss affects many people worldwide, especially with the increasing elderly population, leading to a growing number of individuals suffering from hearing loss. Current treatment options are limited to hearing aids or cochlea implants, but the development of regenerative therapies or cell survival pathways may offer new directions for future treatments.
INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES
(2021)
Article
Multidisciplinary Sciences
Yuki Kobayashi, Kazuma Sugahara, Yosuke Takemoto, Junko Tsuda, Yoshinobu Hirose, Makoto Hashimoto, Hiroshi Yamashita
Summary: The study found that nanoemulsion of astaxanthin has a protective effect on mammalian inner ear hair cells against aminoglycoside-induced ototoxicity. It reduces hair cell loss by decreasing the production of hydroxy radicals and can penetrate the site of inner ear complications for effective treatment.
Review
Chemistry, Multidisciplinary
Sahar Sadat Mahshid, Aliaa Monir Higazi, Jacqueline Michelle Ogier, Alain Dabdoub
Summary: Rapid diagnostic testing is essential in patient care, but there is currently no rapid diagnostic test for hearing loss. Researchers are exploring potential biomarkers for inner ear-related disorders and advancements in biosensor technology to enable accurate diagnostics and personalized therapy in the future. These biomarkers could provide valuable information on the site of damage in the inner ear, aiding in treatment assessment.
Article
Neurosciences
Erin Jimenez, Claire C. Slevin, Luis Colon-Cruz, Shawn M. Burgess
Summary: Millions of Americans suffer from hearing or balance disorders due to the loss of hair cells in the inner ear. Research on transgenic zebrafish has shown that hair cells can regenerate in vivo after induced ablation, indicating potential for future treatments.
FRONTIERS IN CELLULAR NEUROSCIENCE
(2021)
Article
Genetics & Heredity
Le Sun, Lu Ping, Ruzhen Gao, Bo Zhang, Xiaowei Chen
Summary: This study aimed to investigate the role of LMO4 in zebrafish inner ear development. The results showed that lmo4a was constitutively expressed in the otic placode and otic vesicle during the early stages of zebrafish development, and knockdown and knockout of lmo4a resulted in smaller otocysts, fewer hair cells, immature statoacoustic ganglion, and malformed semicircular canals. Further analysis revealed that lmo4a had a regulatory effect on the Bmp pathway and was required for the normal development of zebrafish inner ear.
Article
Cell Biology
Maria Beatriz Duran Alonso, Victor Vendrell, Iris Lopez-Hernandez, Maria Teresa Alonso, Donna M. Martin, Fernando Giraldez, Laura Carramolino, Giovanna Giovinazzo, Enrique Vazquez, Miguel Torres, Thomas Schimmang
Summary: Meis2 gene plays a crucial role in controlling the development of the inner ear, particularly in the formation of the cochlea. The expression of Meis2 in tissues required for inner ear induction and the hindbrain is essential for otic vesicle formation. Inactivation of Meis2 in the inner ear results in an aberrant coiling of the cochlear duct, suggesting its importance in proper cochlear morphogenesis.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2021)
Article
Biology
Dylan J. M. Bergen, Qiao Tong, Ankit Shukla, Elis Newham, Jan Zethof, Mischa Lundberg, Rebecca Ryan, Scott E. Youlten, Monika Frysz, Peter I. Croucher, Gert Flik, Rebecca J. Richardson, John P. Kemp, Chrissy L. Hammond, Juriaan R. Metz
Summary: Dermal scales possess a strong osteogenic expression profile similar to bone, enriched in genes favoring collagen matrix growth. Despite differences in developmental processes between scale and endoskeleton, zebrafish scales express a subset of evolutionarily conserved genes relevant to human skeletal diseases.
Editorial Material
Hematology
Kathleen Freson
Article
Hematology
Fabienne Ver Donck, Kato Ramaekers, Chantal Thys, Christine Van Laer, Kathelijne Peerlinck, Chris Van Geet, Koji Eto, Veerle Labarque, Kathleen Freson
Summary: In this study, pathogenic variants in the SLFN14 gene were found to cause inherited thrombocytopenia. The function of SLFN14 in megakaryocyte and platelet biology was investigated, and it was discovered that SLFN14-defective platelets showed signs of rRNA degradation. Furthermore, dysregulation of the mTORC1 signaling pathway and ribosomal biogenesis were identified as the disease mechanism for SLFN14-related thrombocytopenia.
Article
Virology
Annabel Rector, Mandy Bloemen, Marijn Thijssen, Leen Delang, Joren Raymenants, Jonathan Thibaut, Bram Pussig, Lore Fondu, Bert Aertgeerts, Marc Van Ranst, Chris Van Geet, Jef Arnout, Elke Wollants
Summary: Wastewater surveillance is crucial in managing the global COVID-19 pandemic, serving as an early warning system for outbreaks and a tool to track the circulation of specific variants. This study used various wastewater collection points in Leuven to investigate the application of wastewater surveillance. Wastewater samples were collected and analyzed using RT-qPCR to quantify SARS-CoV-2 RNA, and specific mutations were detected to identify circulating variants. The study found that wastewater SARS-CoV-2 concentration preceded peaks in positive cases and detected the presence of different variants simultaneously during the transition period. Wastewater-based surveillance is a valuable tool for monitoring virus circulation and variants in larger populations.
JOURNAL OF MEDICAL VIROLOGY
(2023)
Article
Biochemistry & Molecular Biology
Daniel Greene, Daniela Pirri, Karen Frudd, Ege Sackey, Mohammed Al-Owain, Arnaud P. J. Giese, Khushnooda Ramzan, Sehar Riaz, Itaru Yamanaka, Nele Boeckx, Chantal Thys, Bruce D. Gelb, Paul Brennan, Verity Hartill, Julie Harvengt, Tomoki Kosho, Sahar Mansour, Mitsuo Masuno, Takako Ohata, Helen Stewart, Khalid Taibah, Claire L. S. Turner, Faiqa Imtiaz, Saima Riazuddin, Takayuki Morisaki, Pia Ostergaard, Bart L. Loeys, Hiroko Morisaki, Zubair M. Ahmed, Graeme M. Birdsey, Kathleen Freson, Andrew Mumford, Ernest Turro
Summary: A database containing rare variant genotypes and phenotypes of 77,539 participants sequenced by the 100,000 Genomes Project enables the identification of new disease-causing genes.
Article
Hematology
Asim Cengiz Akbulut, Ryanne A. Arisz, Constance C. F. M. J. Baaten, Gaukhar Baidildinova, Aarazo Barakzie, Rupert Bauersachs, Jur Ten Berg, Wout W. A. van den Broek, H. C. de Boer, Amandine Bonifay, Vanessa Broker, Richard J. Buka, Hugo ten Cate, Arina J. ten Cate-Hoek, S. Cointe, Ciro De Luca, Ilaria De Simone, Rocio Vacik Diaz, Francoise Dignat-George, Kathleen Freson, Giulia Gazzaniga, Eric C. M. van Gorp, Anxhela Habibi, Yvonne M. C. Henskens, Aaron F. J. Iding, Abdullah Khan, Gijsje H. Koenderink, Akhil Konkoth, Romaric Lacroix, Trisha Lahiri, Wilbur Lam, Rachel E. Lamerton, Roberto Lorusso, Qi Luo, Coen Maas, Owen J. T. McCarty, Paola E. J. van der Meijden, Joost C. M. Meijers, Adarsh K. Mohapatra, Neta Nevo, Alejandro Pallares Robles, Philippe Poncelet, Christoph Reinhardt, Wolfram Ruf, Ronald Saraswat, Claudia Schonichen, Roger Schutgens, Paolo Simioni, Stefano Spada, Henri M. H. Spronk, Karlygash Tazhibayeva, Jecko Thachil, Rocio Vacik Diaz, L. Vallier, Alicia Veninga, Peter Verhamme, Chantal Visser, Steve P. Watson, Philip Wenzel, Ruth A. L. Willems, Anne Willers, Pengyu Zhang, Konstantinos Zifkos, Anton Jan van Zonneveld
Summary: The Fourth Maastricht Consensus Conference on Thrombosis covered a wide range of topics, including the role of coagulation proteins in cardiovascular disease, novel mechanisms of thrombosis, strategies to limit bleeding risks, hemostasis in extracorporeal systems, and clinical dilemmas in thrombosis and antithrombotic management. The conference also revisited the coagulopathy associated with COVID-19.
THROMBOSIS AND HAEMOSTASIS
(2023)
Article
Cell Biology
Sophie T. Raterman, Johannes W. Von Den Hoff, Sietske Dijkstra, Cheyenne De Vriend, Tim Te Morsche, Sanne Broekman, Jan Zethof, Erik De Vrieze, Frank A. D. T. G. Wagener, Juriaan R. Metz
Summary: This study investigates the relationship between mutations in the FOXE1 gene and cleft palate and thyroid dysgenesis in zebrafish. A zebrafish mutant with a disruption in the nuclear localization signal of the foxe1 gene was generated to study skeletal development and thyroidogenesis. The mutant larvae showed abnormal skeletal phenotypes and reduced levels of Ca, Mg, and P, indicating the importance of foxe1 in early skeletal development. Additionally, differential expression of markers for bone and cartilage cells was observed in mutants.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2023)
Article
Biology
Marit de Beer, Deniz Daviran, Rona Roverts, Luco Rutten, Elena Macias-Sanchez, Juriaan R. Metz, Nico Sommerdijk, Anat Akiva
Summary: A cryoCLEM workflow that combines cryo confocal microscopy with 3D cryoFIB/SEM volume imaging allows targeted imaging of tissues in their near-native state, providing high resolution imaging in the unperturbed hydrated state. However, this current method cannot be accurately applied on bulky (3D) samples such as tissues and organoids. To address this, 3D cryo-correlative imaging of large volumes is needed to close the resolution gap between cryo-light microscopy and cryoET. Here, a cryoCLEM workflow for tissues is demonstrated, correlating cryo-Airyscan confocal microscopy with 3D cryoFIB/SEM volume imaging.
COMMUNICATIONS BIOLOGY
(2023)
Editorial Material
Hematology
Kathleen Freson
ARTERIOSCLEROSIS THROMBOSIS AND VASCULAR BIOLOGY
(2023)
Article
Hematology
Christine Van Laer, Marc Jacquemin, Sarissa Baert, Veerle Labarque, Chantal Thys, Thomas Vanassche, Chris Van Geet, Peter Verhamme, Karen Willekens, Anniek Corveleyn, Kathelijne Peerlinck, Kathleen Freson
Summary: This study focuses on the implementation of genetic testing for inherited bleeding, thrombotic, and platelet disorders in Belgian clinical practice. The results highlight the challenges in interpreting genetic results and suggest the need for continuous optimization of diagnostic outcomes. Variants were reclassified and submitted to the ISTH's GoldVariants database.
JOURNAL OF THROMBOSIS AND HAEMOSTASIS
(2023)
Meeting Abstract
Hematology
K. Van Der Zwet, M. de Kovel, J. Motwani, C. van Geet, B. Nolan, H. Glosli, C. Escuriola, C. Koenigs, G. Kenet, K. Fischer
Meeting Abstract
Hematology
C. Van Laer, M. Jacquemin, S. Baert, V. Labarque, C. Thys, T. Vanassche, C. Van Geet, P. Verhamme, K. Willekens, A. Corveleyn, K. Peerlinck, K. Freson
Article
Ophthalmology
Jessie M. Hendricks, Juriaan R. Metz, Hedwig M. Velde, Jack Weeda, Franca Hartgers, Suzanne Yzer, Carel B. Hoyng, Ronald J. E. Pennings, Rob W. J. Collin, Myrthe H. M. Boss, Erik de Vrieze, Erwin Van Wijk
Summary: This study aims to investigate the prevalence, level, and nature of sleep problems and fatigue in Usher syndrome type 2a (USH2a) patients. The results show that compared to healthy controls, USH2a patients experience poorer sleep quality, higher incidence of sleep disorders, and higher levels of fatigue and daytime sleepiness. Importantly, the sleep disturbances and fatigue are not related to the level of visual impairment. This study contributes to the understanding of Usher syndrome and emphasizes the importance of patient care.
OPHTHALMOLOGY SCIENCE
(2023)
Article
Hematology
Gloire Mbayabo, Mamy Ngole, Paul Kabuyi Lumbala, Aime Lumaka, Valerie Race, Gert Matthijs, Tite Minga Mikobi, Koenraad Devriendt, Chris Van Geet, Prosper Tshilobo Lukusa
Summary: This study evaluated the clinical and biological profile of sickle cell anemia children in rural Central Africa. The severity of the disease was found to depend on multiple factors, with fetal hemoglobin being the main modulator of disease severity. These findings can provide valuable information for treatment options.