Genetic screening of the genes interacting with Drosophila FIG4 identified a novel link between CMT-causing gene and long noncoding RNAs

Experimental Neurology
Yuuka MuraokaMasamitsu Yamaguchi

Abstract

Neuron-specific knockdown of the dFIG4 gene, a Drosophila homologue of human FIG4 and one of the causative genes for Charcot-Marie-Tooth disease (CMT), reduces the locomotive abilities of adult flies, as well as causing defects at neuromuscular junctions, such as reduced synaptic branch length in presynaptic terminals of the motor neurons in third instar larvae. Eye imaginal disc-specific knockdown of dFIG4 induces abnormal morphology of the adult compound eye, the rough eye phenotype. In this study, we carried out modifier screening of the dFIG4 knockdown-induced rough eye phenotype using a set of chromosomal deficiency lines on the second chromosome. By genetic screening, we detected 9 and 15 chromosomal regions whose deletions either suppressed or enhanced the rough eye phenotype induced by the dFIG4 knockdown. By further genetic screening with mutants of individual genes in one of these chromosomal regions, we identified the gene CR18854 that suppressed the rough eye phenotype and the loss-of-cone cell phenotype. The CR18854 gene encodes a long non-coding RNA (lncRNA) consisting of 2566 bases. Mutation and knockdown of CR18854 patially suppressed the enlarged lysosome phenotype induced by Fat body-specific knockdown of dFIG...Continue Reading

Citations

Feb 26, 2020·Journal of Biomedical Science·Kuan-Wei Chen, Jun-An Chen
Sep 25, 2019·International Journal of Molecular Sciences·Keqin LiDeying Yang
Jan 10, 2021·International Journal of Molecular Sciences·Masamitsu YamaguchiHideki Yoshida
Oct 15, 2020·International Journal of Molecular Sciences·Fukiko Kitani-Morii, Yu-Ichi Noto
Apr 5, 2021·Experimental Cell Research·Masamitsu YamaguchiHideki Yoshida
May 1, 2021·Animals : an Open Access Journal From MDPI·Chhavi ChoudharyJayendra Nath Shukla
Jun 27, 2021·Developmental and Comparative Immunology·Hongjian ZhouShengjie Li

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