Identification of CR43467 encoding a long non-coding RNA as a novel genetic interactant with dFIG4, a CMT-causing gene

Shimada, Saki; Muraoka, Yuuka; Ibaraki, Kimihide; Takano-Shimizu-Kouno, Toshiyuki; Yoshida, Hideki; Yamaguchi, Masamitsu

doi:10.1016/j.yexcr.2019.111711

Cited by 7 publications

(11 citation statements)

References 38 publications

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“…The CR18854 gene also shows a genetic interaction with Cabeza (dFUS), a Drosophila homolog of human FUS and one of the causing genes of amyotrophic lateral sclerosis (ALS) [ 18 ]. Another lncRNA, CR43467, was also identified as a suppressor of the rough-eye phenotype of dFIG4 and as a rescuer of the enlarged lysosome phenotype induced by fat body-specific knockdown of dFIG4 [ 19 ]. The lncRNAs of the hsrω gene also interact with the dFIG4 in neurons, which suggests that CR18854 lncRNAs and hsrω lncRNAs may interact with common factors such as Cabeza in CMT and ALS pathogenesis [ 16 , 17 ].…”

Section: Lncrnas Associated With Neuronal Functions and Diseases In ...mentioning

confidence: 99%

Hsrω and Other lncRNAs in Neuronal Functions and Disorders in Drosophila

Singh

2022

Life

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Long noncoding RNAs (lncRNAs) have a crucial role in epigenetic, transcriptional and posttranscriptional regulation of gene expression. Many of these regulatory lncRNAs, such as MALAT1, NEAT1, HOTAIR, etc., are associated with different neurodegenerative diseases in humans. The lncRNAs produced by the hsrω gene are known to modulate neurotoxicity in polyQ and amyotrophic lateral sclerosis disease models of Drosophila. Elevated expression of hsrω lncRNAs exaggerates, while their genetic depletion through hsrω-RNAi or in an hsrω-null mutant background suppresses, the disease pathogenicity. This review discusses the possible mechanistic details and implications of the functions of hsrω lncRNAs in the modulation of neurodegenerative diseases.

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Section: Lncrnas Associated With Neuronal Functions and Diseases In ...mentioning

confidence: 99%

Hsrω and Other lncRNAs in Neuronal Functions and Disorders in Drosophila

Singh

2022

Life

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“…Vacuoles were visualized as described previously [11,12]. In brief, the fat body dissected from third instar larva was stained with 100 nM LysoTracker Blue DND22 (Invitrogen, California, USA) for 1 min.…”

Section: Visualization Of Vacuolesmentioning

confidence: 99%

“…In brief, the fat body dissected from third instar larva was stained with 100 nM LysoTracker Blue DND22 (Invitrogen, California, USA) for 1 min. The stained samples were inspected under a confocal laser scanning microscope [11,12]. The diameters of vacuoles were measured using MetaMorph Imaging System 7.7 [11,12].…”

Section: Visualization Of Vacuolesmentioning

confidence: 99%

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Identification of Rpd3 as a novel epigenetic regulator of Drosophila FIG 4, a Charcot-Marie-Tooth disease-causing gene

et al. 2021

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Mutations in the factor-induced-gene 4 (FIG 4) gene are associated with multiple disorders, including Charcot-Marie-Tooth disease (CMT), epilepsy with polymicrogyria, Yunis-Varón syndrome and amyotrophic lateral sclerosis. The wide spectrum of disorders associated with FIG 4 may be related to the dysregulated epigenetics. Using Gene Expression Omnibus, we found that HDAC1 binds to the FIG 4 gene locus in the genome of human CD4 + T cells. Rpd3 is a well-known Drosophila homolog of human HDAC1. We previously established Drosophila models targeting Drosophila FIG 4 (dFIG 4) that exhibited defective locomotive ability, abnormal synapse morphology at neuromuscular junctions, enlarged vacuoles in the fat body and aberrant compound eye morphology. Genetic crossing experiments followed by physiological and immunocytochemical analyses revealed that Rpd3 mutations suppressed these defects induced by dFIG 4 knockdown. This demonstrated Rpd3 to be an important epigenetic regulator of dFIG 4, suggesting that the inhibition of HDAC1 represses the pathogenesis of FIG 4-associated disorders, including CMT. Defects in epigenetic regulators, such as HDAC1, may also explain the diverse symptoms of FIG 4-associated disorders.

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“…Furthermore, Muraoka et al and Shimada et al, using genetic screening, showed that Drosophila long non-coding RNA (lncRNA) interacted genetically with dFIG4 and that knockdown of this lncRNA improved phenotypes including motor deficits such as reduced climbing ability and enlarged lysosomes caused by dFIG4 knockdown [ 86 , 87 ]. Up to now, exome analyses are mainly carried out for the genetic diagnosis of CMT.…”

Section: Drosophila Cmt Models For Investigatinmentioning

confidence: 99%

Recent Advances in Drosophila Models of Charcot-Marie-Tooth Disease

Kitani-Morii

Noto

2020

IJMS

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Charcot-Marie-Tooth disease (CMT) is one of the most common inherited peripheral neuropathies. CMT patients typically show slowly progressive muscle weakness and sensory loss in a distal dominant pattern in childhood. The diagnosis of CMT is based on clinical symptoms, electrophysiological examinations, and genetic testing. Advances in genetic testing technology have revealed the genetic heterogeneity of CMT; more than 100 genes containing the disease causative mutations have been identified. Because a single genetic alteration in CMT leads to progressive neurodegeneration, studies of CMT patients and their respective models revealed the genotype-phenotype relationships of targeted genes. Conventionally, rodents and cell lines have often been used to study the pathogenesis of CMT. Recently, Drosophila has also attracted attention as a CMT model. In this review, we outline the clinical characteristics of CMT, describe the advantages and disadvantages of using Drosophila in CMT studies, and introduce recent advances in CMT research that successfully applied the use of Drosophila, in areas such as molecules associated with mitochondria, endosomes/lysosomes, transfer RNA, axonal transport, and glucose metabolism.

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Identification of CR43467 encoding a long non-coding RNA as a novel genetic interactant with dFIG4, a CMT-causing gene

Cited by 7 publications

References 38 publications

Hsrω and Other lncRNAs in Neuronal Functions and Disorders in Drosophila

Hsrω and Other lncRNAs in Neuronal Functions and Disorders in Drosophila

Identification of Rpd3 as a novel epigenetic regulator of Drosophila FIG 4, a Charcot-Marie-Tooth disease-causing gene

Recent Advances in Drosophila Models of Charcot-Marie-Tooth Disease

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