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.
Neuron,
Journal Year:
2021,
Volume and Issue:
109(6), P. 918 - 937
Published: Feb. 6, 2021
The
superior
colliculus
is
a
conserved
sensorimotor
structure
that
integrates
visual
and
other
sensory
information
to
drive
reflexive
behaviors.
Although
the
evidence
for
this
strong
compelling,
number
of
experiments
reveal
role
in
behaviors
usually
associated
with
cerebral
cortex,
such
as
attention
decision-making.
Indeed,
addition
collicular
outputs
targeting
brainstem
regions
controlling
movements,
also
has
ascending
projections
linking
it
forebrain
structures
including
basal
ganglia
amygdala,
highlighting
fact
colliculus,
its
vast
inputs
outputs,
can
influence
processing
throughout
neuraxis.
Today,
modern
molecular
genetic
methods
combined
sophisticated
behavioral
assessments
have
potential
make
significant
breakthroughs
our
understanding
evolution
conservation
neuronal
cell
types
circuits
give
rise
simple
complex
Current Biology,
Journal Year:
2021,
Volume and Issue:
31(11), P. R741 - R762
Published: June 1, 2021
The
superior
colliculus,
or
tectum
in
the
case
of
non-mammalian
vertebrates,
is
a
part
brain
that
registers
events
surrounding
space,
often
through
vision
and
hearing,
but
also
electrosensation,
infrared
detection,
other
sensory
modalities
diverse
vertebrate
lineages.
This
information
used
to
form
maps
space
positions
different
salient
stimuli
relation
individual.
are
arranged
layers
with
visual
input
uppermost
layer,
senses
deeper
positions,
spatially
aligned
motor
map
deepest
layer.
Here,
we
will
review
organization
intrinsic
function
tectum/superior
colliculus
processed
within
tectal
circuits.
We
discuss
tectal/superior
outputs
conveyed
directly
downstream
circuits
via
thalamus
cortical
areas
control
various
aspects
behavior.
evolutionarily
conserved
among
all
tailored
specialties
each
lineage,
its
roles
have
shifted
emergence
cerebral
cortex
mammals.
illustrate
both
divergent
properties
evolution
by
comparing
processing
lampreys
belonging
oldest
group
extant
larval
zebrafish,
rodents,
vertebrates
including
primates.
Nature,
Journal Year:
2025,
Volume and Issue:
unknown
Published: Feb. 12, 2025
Abstract
Neuronal
phenotypic
traits
such
as
morphology,
connectivity
and
function
are
dictated,
to
a
large
extent,
by
specific
combination
of
differentially
expressed
genes.
Clusters
neurons
in
transcriptomic
space
correspond
distinct
cell
types
some
cases—for
example,
Caenorhabditis
elegans
1
retinal
ganglion
cells
2–4
—have
been
shown
share
morphology
function.
The
zebrafish
optic
tectum
is
composed
spatial
array
that
transforms
visual
inputs
into
motor
outputs.
Although
the
visuotopic
map
continuous,
subregions
functionally
specialized
5,6
.
Here,
uncover
cell-type
architecture
tectum,
we
transcriptionally
profiled
its
neurons,
revealing
more
than
60
organized
anatomical
layers.
We
measured
responses
thousands
tectal
two-photon
calcium
imaging
matched
them
with
their
transcriptional
profiles.
Furthermore,
characterized
morphologies
identified
using
transgenic
lines.
Notably,
found
similar
can
diverge
shape,
responses.
Incorporating
coordinates
within
volume
revealed
morphologically
defined
subclusters
individual
clusters.
Our
findings
demonstrate
extrinsic,
position-dependent
factors
expand
repertoire
genetically
neurons.
Neuroscience Bulletin,
Journal Year:
2022,
Volume and Issue:
38(12), P. 1519 - 1540
Published: April 28, 2022
Abstract
The
superior
colliculus
(SC),
one
of
the
most
well-characterized
midbrain
sensorimotor
structures
where
visual,
auditory,
and
somatosensory
information
are
integrated
to
initiate
motor
commands,
is
highly
conserved
across
vertebrate
evolution.
Moreover,
cell-type-specific
SC
neurons
integrate
afferent
signals
within
local
networks
generate
defined
output
related
innate
cognitive
behaviors.
This
review
focuses
on
recent
progress
in
understanding
phenotypic
diversity
amongst
their
intrinsic
circuits
long-projection
targets.
We
further
describe
relevant
neural
specific
cell
types
relation
behavioral
outputs
functions.
systematic
delineation
organization,
types,
connections
put
into
context
species
as
these
depend
upon
laminar
architecture.
we
focus
circuitry
involving
saccadic
eye
movement,
Overall,
provides
insight
functioning
represents
a
basis
for
pathology
associated
with
dysfunction.
Frontiers in Neuroscience,
Journal Year:
2023,
Volume and Issue:
17
Published: April 17, 2023
One
of
the
holy
grails
neuroscience
is
to
record
activity
every
neuron
in
brain
while
an
animal
moves
freely
and
performs
complex
behavioral
tasks.
While
important
steps
forward
have
been
taken
recently
large-scale
neural
recording
rodent
models,
single
resolution
across
entire
mammalian
remains
elusive.
In
contrast
larval
zebrafish
offers
great
promise
this
regard.
Zebrafish
are
a
vertebrate
model
with
substantial
homology
brain,
but
their
transparency
allows
whole-brain
recordings
genetically-encoded
fluorescent
indicators
at
single-neuron
using
optical
microscopy
techniques.
Furthermore
begin
show
repertoire
natural
behavior
from
early
age,
including
hunting
small,
fast-moving
prey
visual
cues.
Until
work
address
bases
these
behaviors
mostly
relied
on
assays
where
fish
was
immobilized
under
microscope
objective,
stimuli
such
as
were
presented
virtually.
However
significant
progress
has
made
developing
imaging
techniques
for
which
not
immobilized.
Here
we
discuss
recent
advances,
focusing
particularly
based
light-field
microscopy.
We
also
draw
attention
several
outstanding
issues
remain
be
addressed
increase
ecological
validity
results
obtained.
Nature Communications,
Journal Year:
2021,
Volume and Issue:
12(1)
Published: Nov. 12, 2021
Abstract
Complex
schooling
behaviors
result
from
local
interactions
among
individuals.
Yet,
how
sensory
signals
neighbors
are
analyzed
in
the
visuomotor
stream
of
animals
is
poorly
understood.
Here,
we
studied
aggregation
behavior
larval
zebrafish
and
found
that
over
development
larvae
transition
overdispersed
groups
to
tight
shoals.
Using
a
virtual
reality
assay,
characterized
algorithms
fish
use
transform
visual
inputs
into
movement
decisions.
We
young
turn
away
by
integrating
averaging
retina-wide
occupancy
within
each
eye,
using
winner-take-all
strategy
for
binocular
integration.
As
mature,
their
responses
expand
include
attraction
neighbors,
which
based
on
similar
model
simulations,
show
observed
accurately
predict
group
structure
development.
These
findings
allow
us
make
testable
predictions
regarding
neuronal
circuits
underlying
collective
zebrafish.
Frontiers in Neural Circuits,
Journal Year:
2023,
Volume and Issue:
17
Published: Feb. 1, 2023
A
key
challenge
for
neural
systems
is
to
extract
relevant
information
from
the
environment
and
make
appropriate
behavioral
responses.
The
larval
zebrafish
offers
an
exciting
opportunity
studying
these
sensing
processes
sensory-motor
transformations.
Prey
hunting
instinctual
behavior
of
that
requires
brain
combine
different
attributes
sensory
input
form
motor
outputs.
Due
its
small
size
transparency
allows
optical
recording
whole-brain
activity
reveal
mechanisms
involved
in
prey
capture.
In
this
review
we
discuss
how
visual
identify
locate
prey,
circuits
governing
generation
commands
response
can
be
modulated
by
internal
states
experience,
some
outstanding
questions
field.
Annual Review of Neuroscience,
Journal Year:
2024,
Volume and Issue:
47(1), P. 255 - 276
Published: April 25, 2024
The
zebrafish
visual
system
has
become
a
paradigmatic
preparation
for
behavioral
and
systems
neuroscience.
Around
40
types
of
retinal
ganglion
cells
(RGCs)
serve
as
matched
filters
stimulus
features,
including
light,
optic
flow,
prey,
objects
on
collision
course.
RGCs
distribute
their
signals
via
axon
collaterals
to
12
retinorecipient
areas
in
forebrain
midbrain.
major
visuomotor
hub,
the
tectum,
harbors
nine
RGC
input
layers
that
combine
information
multiple
features.
retinotopic
map
tectum
is
locally
adapted
scene
statistics
subfield–specific
demands.
Tectal
projections
premotor
centers
are
topographically
organized
according
commands.
known
connectivity
more
than
20
processing
streams
allows
us
dissect
cellular
basis
elementary
perceptual
cognitive
functions.
Visually
evoked
responses,
such
prey
capture
or
loom
avoidance,
controlled
by
dedicated
multistation
pathways
that—at
least
larva—resemble
labeled
lines.
This
architecture
serves
neuronal
code's
purpose
driving
adaptive
behavior.