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REVIEW
Lok Ming Tam, Matthew D Rand
A number of environmental toxicants are noted for their activity that leads to declined motor function. However, the role of muscle as a proximal toxicity target organ for environmental agents has received considerably less attention than the toxicity targets in the nervous system. Nonetheless, the effects of conventional neurotoxicants on processes of myogenesis and muscle maintenance are beginning to resolve a concerted role of muscle as a susceptible toxicity target. A large body of evidence from epidemiological, animal, and in vitro studies has established that methylmercury (MeHg) is a potent developmental toxicant, with the nervous system being a preferred target...
March 28, 2024: Archives of Toxicology
#2
JOURNAL ARTICLE
Zulvikar Syambani Ulhaq, William Ka Fai Tse
Nager syndrome (NS) is a rare acrofacial dysostosis caused by heterozygous loss-of-function variants in the splicing factor 3B subunit 4 (SF3B4). The main clinical features of patients with NS are characterized by facial-mandibular and preaxial limb malformations. The migration and specification of neural crest cells are crucial for craniofacial development, and mitochondrial fitness appears to play a role in such processes. Here, by analyzing our previously published transcriptome dataset, we aim to investigate the potential involvement of mitochondrial components in the pathogenesis of craniofacial malformations, especially in sf3b4 mutant zebrafish...
March 18, 2024: Biochimica et Biophysica Acta. Molecular Basis of Disease
#3
JOURNAL ARTICLE
Ana Töpf, Dan Cox, Irina T Zaharieva, Valeria Di Leo, Jaakko Sarparanta, Per Harald Jonson, Ian M Sealy, Andrei Smolnikov, Richard J White, Anna Vihola, Marco Savarese, Munise Merteroglu, Neha Wali, Kristen M Laricchia, Cristina Venturini, Bas Vroling, Sarah L Stenton, Beryl B Cummings, Elizabeth Harris, Chiara Marini-Bettolo, Jordi Diaz-Manera, Matt Henderson, Rita Barresi, Jennifer Duff, Eleina M England, Jane Patrick, Sundos Al-Husayni, Valerie Biancalana, Alan H Beggs, Istvan Bodi, Shobhana Bommireddipalli, Carsten G Bönnemann, Anita Cairns, Mei-Ting Chiew, Kristl G Claeys, Sandra T Cooper, Mark R Davis, Sandra Donkervoort, Corrie E Erasmus, Mahmoud R Fassad, Casie A Genetti, Carla Grosmann, Heinz Jungbluth, Erik-Jan Kamsteeg, Xavière Lornage, Wolfgang N Löscher, Edoardo Malfatti, Adnan Manzur, Pilar Martí, Tiziana E Mongini, Nuria Muelas, Atsuko Nishikawa, Anne O'Donnell-Luria, Narumi Ogonuki, Gina L O'Grady, Emily O'Heir, Stéphanie Paquay, Rahul Phadke, Beth A Pletcher, Norma B Romero, Meyke Schouten, Snehal Shah, Izelle Smuts, Yves Sznajer, Giorgio Tasca, Robert W Taylor, Allysa Tuite, Peter Van den Bergh, Grace VanNoy, Nicol C Voermans, Julia V Wanschitz, Elizabeth Wraige, Kimihiko Yoshimura, Emily C Oates, Osamu Nakagawa, Ichizo Nishino, Jocelyn Laporte, Juan J Vilchez, Daniel G MacArthur, Anna Sarkozy, Heather J Cordell, Bjarne Udd, Elisabeth M Busch-Nentwich, Francesco Muntoni, Volker Straub
In digenic inheritance, pathogenic variants in two genes must be inherited together to cause disease. Only very few examples of digenic inheritance have been described in the neuromuscular disease field. Here we show that predicted deleterious variants in SRPK3, encoding the X-linked serine/argenine protein kinase 3, lead to a progressive early onset skeletal muscle myopathy only when in combination with heterozygous variants in the TTN gene. The co-occurrence of predicted deleterious SRPK3/TTN variants was not seen among 76,702 healthy male individuals, and statistical modeling strongly supported digenic inheritance as the best-fitting model...
March 1, 2024: Nature Genetics
#4
REVIEW
Sabrina C Fox, Andrew J Waskiewicz
Humans and other jawed vertebrates rely heavily on their craniofacial skeleton for eating, breathing, and communicating. As such, it is vital that the elements of the craniofacial skeleton develop properly during embryogenesis to ensure a high quality of life and evolutionary fitness. Indeed, craniofacial abnormalities, including cleft palate and craniosynostosis, represent some of the most common congenital abnormalities in newborns. Like many other organ systems, the development of the craniofacial skeleton is complex, relying on specification and migration of the neural crest, patterning of the pharyngeal arches, and morphogenesis of each skeletal element into its final form...
2024: Frontiers in Cell and Developmental Biology
#5
JOURNAL ARTICLE
Ada Jimenez-Gonzalez, Federico Ansaloni, Constance Nebendahl, Ghazal Alavioon, David Murray, Weronika Robak, Remo Sanges, Ferenc Müller, Simone Immler
Dietary restriction in the form of fasting is a putative key to a healthier and longer life, but these benefits may come at a trade-off with reproductive fitness and may affect the following generation(s). The potential inter- and transgenerational effects of long-term fasting and starvation are particularly poorly understood in vertebrates when they originate from the paternal line. We utilised the externally fertilising zebrafish amenable to a split-egg clutch design to explore the male-specific effects of fasting/starvation on fertility and fitness of offspring independently of maternal contribution...
February 15, 2024: Molecular Ecology
#6
Julia Peloggia, Mark E Lush, Ya-Yin Tsai, Christopher Wood, Tatjana Piotrowski
Organisms adjust their physiology to cope with environmental fluctuations and maintain fitness. These adaptations occur via genetic changes over multiple generations or through acclimation, a set of reversible phenotypic changes that confer resilience to the individual. Aquatic organisms are subject to dramatic seasonal fluctuations in water salinity, which can affect the function of lateral line mechanosensory hair cells. To maintain hair cell function when salinity decreases, ion-regulating cells, Neuromast-associated ionocytes (Nm ionocytes), increase in number and invade lateral line neuromasts...
January 13, 2024: bioRxiv
#7
Jagmeet S Kanwal, Bhavjeet Sanghera, Riya Dabbi, Eric Glasgow
Movement requires maneuvers that generate thrust to either make turns or move the body forward in physical space. The computational space for perpetually controlling the relative position of every point on the body surface can be vast. We hypothesize the evolution of efficient design for movement that minimizes active (neural) control by leveraging the passive (reactive) forces between the body and the surrounding medium at play. To test our hypothesis, we investigate the presence of stereotypical postures during free-swimming in adult zebrafish, Danio rerio ...
January 1, 2024: bioRxiv
#8
JOURNAL ARTICLE
Alejandra Godino-Gimeno, Esther Leal, Mauro Chivite, Elisabeth Tormos, Josep Rotllant, Daniela Vallone, Nicholas S Foulkes, Jesús M Míguez, Jose Miguel Cerdá-Reverter
Temporal signals such as light and temperature cycles profoundly modulate animal physiology and behaviour. Via endogenous timing mechanisms which are regulated by these signals, organisms can anticipate cyclic environmental changes and thereby enhance their fitness. The pineal gland in fish, through the secretion of melatonin, appears to play a critical role in the circadian system, most likely acting as an element of the circadian clock system. An important output of this circadian clock is the locomotor activity circadian rhythm which is adapted to the photoperiod and thus determines whether animals are diurnal or nocturnal...
January 2024: Journal of Pineal Research
#9
JOURNAL ARTICLE
Alexandra Y Kreins, Emma Roux, Juanita Pang, Iek Cheng, Oscar Charles, Sunando Roy, Reem Mohammed, Stephen Owens, David M Lowe, Rossa Brugha, Rachel Williams, Evey Howley, Timothy Best, E Graham Davies, Austen Worth, Caroline Solas, Joseph F Standing, Richard A Goldstein, Joana Rocha-Pereira, Judith Breuer
Chronic human norovirus (HuNoV) infections in immunocompromised patients result in severe disease, yet approved antivirals are lacking. RNA-dependent RNA polymerase (RdRp) inhibitors inducing viral mutagenesis display broad-spectrum in vitro antiviral activity, but clinical efficacy in HuNoV infections is anecdotal and the potential emergence of drug-resistant variants is concerning. Upon favipiravir (and nitazoxanide) treatment of four immunocompromised patients with life-threatening HuNoV infections, viral whole-genome sequencing showed accumulation of favipiravir-induced mutations which coincided with clinical improvement although treatment failed to clear HuNoV...
January 11, 2024: Clinical Immunology: the Official Journal of the Clinical Immunology Society
#10
JOURNAL ARTICLE
Amanda K Pettersen, Neil B Metcalfe, Frank Seebacher
Metabolic rates are linked to key life-history traits that are thought to set the pace of life and affect fitness, yet the role that parents may have in shaping the metabolism of their offspring to enhance survival remains unclear. Here, we investigated the effect of temperature (24°C or 30°C) and feeding frequency experienced by parent zebrafish ( Danio rerio ) on offspring phenotypes and early survival at different developmental temperatures (24°C or 30°C). We found that embryo size was larger, but survival lower, in offspring from the parental low food treatment...
February 26, 2024: Philosophical Transactions of the Royal Society of London. Series B, Biological Sciences
#11
JOURNAL ARTICLE
Laura González-Llera, Daniel Sobrido-Cameán, Ana Quelle-Regaldie, Laura Sánchez, Antón Barreiro-Iglesias
The study of neurogenesis is essential to understanding fundamental developmental processes and for the development of cell replacement therapies for central nervous system disorders. Here, we designed an in vivo drug screening protocol in developing zebrafish to find new molecules and signalling pathways regulating neurogenesis in the ventral spinal cord. This unbiased drug screen revealed that 4 cyclooxygenase (COX) inhibitors reduced the generation of serotonergic interneurons in the developing spinal cord...
December 28, 2023: Cell Proliferation
#12
JOURNAL ARTICLE
Ismahene Ben Khemis, Fatma Aouaini, Lamies Bukhari, Amani Alruwaili, Salah Knani, Abdelmottaleb Ben Lamine
In this work, an adsorption phenomenon putatively involved in the olfactory sense of phenylacetic acid, 4-chlorophenylacetic acid, and 4-methoxyphenylacetic acid pheromones in the Zebrafish olfactory receptor ORA1 was a helpful mechanism in interpreting and characterizing the olfaction process at a molecular level. Hence, the experimental dose-olfactory response curves were fitted by applying the one-layer adsorption model with a single energy (1LM1E). On one hand, the different parameters introduced in the selected model were used to microscopically study the three olfactory systems...
December 11, 2023: International Journal of Biological Macromolecules
#13
JOURNAL ARTICLE
Nityashree K L, Manoj K Pandey, Mohammed F Albeshr, Abdulwahed Fahad Alrefaei, Devaraj Bharathi, Jintae Lee, Vinay B Raghavendra
Phenanthrene is a persistent organic pollutant released by numerous industries. The purpose of the study is to construct a batch reactor for phenanthrene degradation using a bimetallic ZnS-SnS nanoparticle as a photocatalyst. ZnS-SnS BM NPs were used as a photocatalyst, employed from precursors Zinc acetate dihydrate and tin (II) chloride dihydrate, with crystalline cubic-shaped particle sizes. ZnS-SnS BM NPs were utilized in batch adsorption assays to assess the impact of phenanthrene degradation parameters on various PAHs (Polycyclic aromatic hydrocarbons) concentrations, pH levels, and irradiation sources...
December 3, 2023: Chemosphere
#14
JOURNAL ARTICLE
Frank Seebacher, Stephanie M Bamford
Energy transfer efficiency across different trophic levels, from food to new biomass, can determine population dynamics and food-web function. Here we show that the energy needed to produce a unit of new biomass increases with warming and exposure to bisphenol A (BPA), an endocrine disrupting compound. These environmental effects are at least partially transmitted across generations via DNA methylation. We raised parental (F0) and their offspring (F1) zebrafish (Danio rerio) of two genotypes (DNA methyltransferase 3a knock-out [DNMT3a-/- ] and wild type [DNMT3a+/+ ]) at different temperatures (24 and 30 °C), with and without BPA (0 and 10 μg l-1 ) to test whether the effects of BPA are i) temperature specific, ii) mediated by DNA methylation, and iii) transmitted across generations even if offspring are not exposed...
December 1, 2023: Science of the Total Environment
#15
JOURNAL ARTICLE
Itishree Jogamaya Das, Trishna Bal
The process of wound healing involves complex interplay of systems biology, dependent on coordination of various cell types, both intra and extracellular mechanisms, proteins, and signaling pathways. To enhance these interactions, drugs must be administered precisely and continuously, effectively regulating the intricate mechanisms involved in the body's response to injury. Controlled drug delivery systems (DDS) play a pivotal role in achieving this objective. A proficient DDS shields the wound from mechanical, oxidative, and enzymatic stress, against bacterial contamination ensuring an adequate oxygen supply while optimizing the localized and sustained delivery of drugs to target tissue...
November 29, 2023: International Journal of Biological Macromolecules
#16
JOURNAL ARTICLE
Katsiaryna Tarbashevich, Laura Ermlich, Julian Wegner, Jana Pfeiffer, Erez Raz
To maintain a range of cellular functions and to ensure cell survival, cells must control their levels of reactive oxygen species (ROS). The main source of these molecules is the mitochondrial respiration machinery, and the first line of defense against these toxic substances is the mitochondrial enzyme superoxide dismutase 2 (Sod2). Thus, investigating early expression patterns and functions of this protein is critical for understanding how an organism develops ways to protect itself against ROS and enhance tissue fitness...
2023: Frontiers in Cell and Developmental Biology
#17
JOURNAL ARTICLE
Alexandra Kroll, Peter C von der Ohe, Heinz-R Köhler, Odile Sellier, Marion Junghans
Diclofenac, a widely used nonsteroidal anti-inflammatory drug (NSAID), enters the aquatic environment worldwide. The effect values available for the derivation of an environmental quality standard (EQS) are markedly heterogeneous, even within the same species. This heterogeneity could partially be attributed to inter-laboratory variation, but is also observed in repeated tests within the same facility. Diclofenac is ionisable; its speciation and potential for uptake and thus toxicity is influenced by pH. A high correlation has previously been observed between effects in zebrafish embryos and the pH-specific partitioning coefficient logD for diclofenac...
November 10, 2023: Science of the Total Environment
#18
JOURNAL ARTICLE
Sian-Tai Liu, Jiun-Lin Horng, Li-Yih Lin, Ming-Yi Chou
Fenpropathrin is one of the widely used pyrethroid pesticides in agriculture and is frequently detected in the environment, groundwater, and food. While fenpropathrin was found to have neurotoxic effects in mammals, it remains unclear whether it has similar effects on fish. Here, we used adult zebrafish to investigate the impacts of fenpropathrin on fish social behaviors and neural activity. Exposure of adult zebrafish to 500 ppb of fenpropathrin for 72 h increased anxiety levels but decreased physical fitness, as measured by a novel tank diving test and swimming tunnel test...
November 7, 2023: Aquatic Toxicology
#19
JOURNAL ARTICLE
Aya Takesono, Sylvia Dimitriadou, Nathaniel J Clark, Richard D Handy, Sulayman Mourabit, Matthew J Winter, Tetsuhiro Kudoh, Charles R Tyler
Zinc (Zn) is an essential metal present in numerous enzymes throughout the body, playing a vital role in animal and human health. However, the increasing use of zinc oxide nanomaterials (ZnONPs) in a diverse range of products has raised concerns regarding their potential impacts on health and the environment. Despite these concerns, the toxicity of ZnONP exposure on animal health remain poorly understood. To help address this knowledge gap, we have developed a highly sensitive oxidative stress (OS) biosensor zebrafish capable of detecting cell/tissue-specific OS responses to low doses of various oxidative stressors, including Zn, in a live fish embryo...
September 21, 2023: Environment International
#20
JOURNAL ARTICLE
Christina L Miller, Derek Sun, Lauren H Thornton, Katrina McGuigan
AbstractThe contribution of new mutations to phenotypic variation and the consequences of this variation for individual fitness are fundamental concepts for understanding genetic variation and adaptation. Here, we investigated how mutation influenced variation in a complex trait in zebrafish, Danio rerio . Typical of many ecologically relevant traits in ectotherms, swimming speed in fish is temperature dependent, with evidence of adaptive evolution of thermal performance. We chemically induced novel germline point mutations in males and measured sprint speed in their sons at six temperatures (between 16°C and 34°C)...
October 2023: American Naturalist
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