Morphological, behavioral and cellular analyses revealed different phenotypes in Wolfram syndrome wfs1a and wfs1b zebrafish mutant lines DOI Creative Commons
Lucie Crouzier, Élodie M. Richard,

Camille Diez

et al.

Human Molecular Genetics, Journal Year: 2022, Volume and Issue: 31(16), P. 2711 - 2727

Published: March 17, 2022

Wolfram syndrome (WS) is a rare genetic disease characterized by diabetes, optic atrophy and deafness. Patients die at 35 years of age, mainly from respiratory failure or dysphagia. Unfortunately, there no treatment to block the progression symptoms an urgent need for adequate research models. Here, we report on phenotypical characterization two loss-of-function zebrafish mutant lines: wfs1aC825X wfs1bW493X. We observed that wfs1a deficiency altered size ear retina fish. also documented decrease in expression level unfolded protein response (UPR) genes basal condition stress condition, i.e. after tunicamycin treatment. Interestingly, both mutants lead their visual function measured behaviorally. These deficits were associated with UPR conditions. basal, ATP-linked maximal mitochondrial respirations transiently decreased wfs1b mutant. Taken together, these lines highlight critical role UPR, physiology. models will be useful tools better understand cellular Wfs1 develop novel therapeutic approaches WS.

Language: Английский

Quantity as a Fish Views It: Behavior and Neurobiology DOI Creative Commons
Andrea Messina, Davide Potrich,

Matilde Perrino

et al.

Frontiers in Neuroanatomy, Journal Year: 2022, Volume and Issue: 16

Published: July 14, 2022

An ability to estimate quantities, such as the number of conspecifics or size a predator, has been reported in vertebrates. Fish, particular zebrafish, may be instrumental advancing understanding magnitude cognition. We review here behavioral studies that have described ecological relevance quantity estimation fish and current status research aimed at investigating neurobiological bases these abilities. By combining methods with molecular genetics calcium imaging, involvement retina optic tectum documented for continuous quantities larval adult zebrafish brain, contributions thalamus dorsal-central pallium discrete brain. Evidence basic circuitry can now complemented extended make use transgenic lines deepen our cognition genetic levels.

Language: Английский

Citations

18
Blind cavefish retain functional connectivity in the tectum despite loss of retinal input DOI Creative Commons
Evan Lloyd,

Brittnee McDole,

Martin Privat

et al.

Current Biology, Journal Year: 2022, Volume and Issue: 32(17), P. 3720 - 3730.e3

Published: Aug. 4, 2022

Sensory systems display remarkable plasticity and are under strong evolutionary selection. The Mexican cavefish, Astyanax mexicanus, consists of eyed river-dwelling surface populations multiple independent cave that have converged on eye loss, providing the opportunity to examine evolution sensory circuits in response environmental perturbation. Functional analysis across transgenic expressing GCaMP6s showed functional connectivity optic tectum largely did not differ between populations, except for selective loss negatively correlated activity within cavefish tectum, suggesting positively neural is resistant an evolved input from retina. Furthermore, surface-cave hybrid fish reveals changes genetically distinct those encoding loss. Together, these findings uncover components visual system establish use imaging A. mexicanus study circuit evolution.

Language: Английский

Citations

17
Eat, flee, freeze: Division of labor in the larval zebrafish visuomotor system DOI Creative Commons
Qing Wang, Herwig Baier

Proceedings of the National Academy of Sciences, Journal Year: 2025, Volume and Issue: 122(19)

Published: May 5, 2025

Language: Английский

Citations

0
DSRP Theory: A Primer DOI Creative Commons
Derek Cabrera, Laura Cabrera

Systems, Journal Year: 2022, Volume and Issue: 10(2), P. 26 - 26

Published: March 2, 2022

DSRP Theory is now over 25 years old with more empirical evidence supporting it than any other systems thinking framework. Yet, often misunderstood and described in ways that are inaccurate. describes four patterns their underlying elements—identity (i) (o) for Distinctions (D), part (p) whole (w) Systems (S), action (a) reaction (r) Relationships (R), point (ρ) view (v) Perspectives (P)—that universal both cognitive complexity (mind) material (nature). provides a basis or as well (systems science). This paper, relatively short primer on the theory, clarity to those wanting understand its implications.

Language: Английский

Citations

15
Morphological, behavioral and cellular analyses revealed different phenotypes in Wolfram syndrome wfs1a and wfs1b zebrafish mutant lines DOI Creative Commons
Lucie Crouzier, Élodie M. Richard,

Camille Diez

et al.

Human Molecular Genetics, Journal Year: 2022, Volume and Issue: 31(16), P. 2711 - 2727

Published: March 17, 2022

Wolfram syndrome (WS) is a rare genetic disease characterized by diabetes, optic atrophy and deafness. Patients die at 35 years of age, mainly from respiratory failure or dysphagia. Unfortunately, there no treatment to block the progression symptoms an urgent need for adequate research models. Here, we report on phenotypical characterization two loss-of-function zebrafish mutant lines: wfs1aC825X wfs1bW493X. We observed that wfs1a deficiency altered size ear retina fish. also documented decrease in expression level unfolded protein response (UPR) genes basal condition stress condition, i.e. after tunicamycin treatment. Interestingly, both mutants lead their visual function measured behaviorally. These deficits were associated with UPR conditions. basal, ATP-linked maximal mitochondrial respirations transiently decreased wfs1b mutant. Taken together, these lines highlight critical role UPR, physiology. models will be useful tools better understand cellular Wfs1 develop novel therapeutic approaches WS.

Language: Английский

Citations

15