AXONAL-TRANSPORT DEFECTS;
HUMAN CHARGE SYNDROME;
IN-VIVO;
EPIGENETIC MECHANISMS;
BEHAVIORAL PHENOTYPES;
ALZHEIMERS-DISEASE;
GENE-EXPRESSION;
ACTIVE ZONES;
PROTEIN;
CHD7;
D O I:
10.1371/journal.pone.0113494
中图分类号:
O [数理科学和化学];
P [天文学、地球科学];
Q [生物科学];
N [自然科学总论];
学科分类号:
07 ;
0710 ;
09 ;
摘要:
The Drosophila neuromuscular junction (NMJ) is a glutamatergic synapse that is structurally and functionally similar to mammalian glutamatergic synapses. These synapses can, as a result of changes in activity, alter the strength of their connections via processes that require chromatin remodeling and changes in gene expression. The chromodomain helicase DNA binding (CHD) protein, Kismet (Kis), is expressed in both motor neuron nuclei and postsynaptic muscle nuclei of the Drosophila larvae. Here, we show that Kis is important for motor neuron synaptic morphology, the localization and clustering of postsynaptic glutamate receptors, larval motor behavior, and synaptic transmission. Our data suggest that Kis is part of the machinery that modulates the development and function of the NMJ. Kis is the homolog to human CHD7, which is mutated in CHARGE syndrome. Thus, our data suggest novel avenues of investigation for synaptic defects associated with CHARGE syndrome.
机构:
Baylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
Baylor Coll Med, Med Scientist Training Program, Houston, TX 77030 USABaylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
Bayat, Vafa
;
Jaiswal, Manish
论文数: 0引用数: 0
h-index: 0
机构:
Baylor Coll Med, Dept Mol & Human Genet, Houston, TX 77030 USABaylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
Jaiswal, Manish
;
Bellen, Hugo J.
论文数: 0引用数: 0
h-index: 0
机构:
Baylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
Baylor Coll Med, Dept Mol & Human Genet, Houston, TX 77030 USA
Baylor Coll Med, Howard Hughes Med Inst, Houston, TX 77030 USA
Baylor Coll Med, Dept Neurosci, Houston, TX 77030 USABaylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
机构:
Baylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
Baylor Coll Med, Med Scientist Training Program, Houston, TX 77030 USABaylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
Bayat, Vafa
;
Jaiswal, Manish
论文数: 0引用数: 0
h-index: 0
机构:
Baylor Coll Med, Dept Mol & Human Genet, Houston, TX 77030 USABaylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
Jaiswal, Manish
;
Bellen, Hugo J.
论文数: 0引用数: 0
h-index: 0
机构:
Baylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
Baylor Coll Med, Dept Mol & Human Genet, Houston, TX 77030 USA
Baylor Coll Med, Howard Hughes Med Inst, Houston, TX 77030 USA
Baylor Coll Med, Dept Neurosci, Houston, TX 77030 USABaylor Coll Med, Program Dev Biol, Houston, TX 77030 USA