Редактирование: FIG4 (раздел)

==Publications==

{{medline-entry
|title=Knockdown of the Drosophila [[FIG4]] induces deficient locomotive behavior, shortening of motor neuron, axonal targeting aberration, reduction of life span and defects in eye development.
|pubmed-url=https://pubmed.ncbi.nlm.nih.gov/26708557
|abstract=Mutations in Factor-Induced-Gene 4 ([[FIG4]]) gene have been identified in Charcot-Marie-Tooth disease type 4J (CMT4J), Yunis-Varon syndrome and epilepsy with polymicrogyria. [[FIG4]] protein regulates a cellular abundance of phosphatidylinositol 3,5-bisphosphate (PI(3,5)P2), a signaling lipid on the cytosolic surface of membranes of the late endosomal compartment. PI(3,5)P2 is required for retrograde membrane trafficking from lysosomal and late endosomal compartments to the Golgi. However, it is still unknown how the neurodegeneration that occurs in these diseases is related to the loss of [[FIG4]] function. Drosophila has CG17840 (d[[FIG4]]) as a human [[FIG4]] homolog. Here we specifically knocked down d[[FIG4]] in various tissues, and investigated their phenotypes. Neuron-specific knockdown of d[[FIG4]] resulted in axonal targeting aberrations of photoreceptor neurons, shortened presynaptic terminals of motor neurons in 3rd instar larvae and reduced climbing ability in adulthood and life span. Fat body-specific knockdown of d[[FIG4]] resulted in enlarged lysosomes in cells that were detected by staining with LysoTracker. In addition, eye imaginal disk-specific knockdown of d[[FIG4]] disrupted differentiation of pupal ommatidial cell types, such as cone cells and pigment cells, suggesting an additional role of d[[FIG4]] during eye development.
|mesh-terms=* Animals
* Animals, Genetically Modified
* Axons
* Central Nervous System
* Disease Models, Animal
* Drosophila
* Drosophila Proteins
* Eye Abnormalities
* Flavoproteins
* Gait Disorders, Neurologic
* Green Fluorescent Proteins
* Humans
* Imaginal Discs
* Longevity
* Lysosomes
* Motor Neurons
* Neuromuscular Junction
* Phosphoric Monoester Hydrolases
* Photoreceptor Cells, Invertebrate
* Psychomotor Disorders
* Sequence Alignment
|keywords=* CMT4J
* Drosophila
* Eye
* FIG4
* Neuron
|full-text-url=https://sci-hub.do/10.1016/j.expneurol.2015.12.011
}}