Molecular Characterization of <i>teflon</i>, a Gene Required for Meiotic Autosome Segregation in Male <i>Drosophila melanogaster</i>

Arya, Gunjan H.; Lodico, Matthew J. P.; Ahmad, Omar; Amin, Rohul; Tomkiel, John E.

doi:10.1534/genetics.106.061556

Cited by 25 publications

(56 citation statements)

References 35 publications

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“…(A-G) Crystal phenotype in the indicated genotypes analyzed by epifluorescence microscope; antiSte labeling is in green, DAPI in blue. T76-Gal4 is considered as a germline driver (Hrdlicka et al, 2002;Arya et al, 2006). The images were taken with a 40× magnification objective.…”

Section: ▵113mentioning

confidence: 99%

“…(H) Western blot analysis with anti-dFmr1 antibody on protein extracts from of third-instar larvae of the indicated genotypes. The dFmr1 ▵50 mutation triggers the expression of a residual protein that is 17 amino acids shorter than the wt protein, whereas the dFmr1 ▵113 mutation results in the complete lack of dFmr1 protein (Reeve et al, 2005 Importantly, dFmr1 expression in testes driven by the T76 driver (Arya et al, 2006;Hrdlicka et al, 2002) rescues the 'crystal' phenotype of dFmr1 ▵113 heterozygous and homozygous flies as well as the lethality of the homozygous flies (Fig. 1F, T76-Gal4/+;UAS-dFmr1, dFmr1 ▵113 /+; and data not shown), confirming that these defects depend on the dFmr1 mutation.…”

Section: ▵113mentioning

confidence: 99%

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The Drosophila fragile X mental retardation protein participates in the piRNA pathway

Bozzetti

Specchia

Cattenoz

et al. 2015

Journal of Cell Science

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RNA metabolism controls multiple biological processes, and a specific class of small RNAs, called piRNAs, act as genome guardians by silencing the expression of transposons and repetitive sequences in the gonads. Defects in the piRNA pathway affect genome integrity and fertility. The possible implications in physiopathological mechanisms of human diseases have made the piRNA pathway the object of intense investigation, and recent work suggests that there is a role for this pathway in somatic processes including synaptic plasticity. The RNAbinding fragile X mental retardation protein (FMRP, also known as FMR1) controls translation and its loss triggers the most frequent syndromic form of mental retardation as well as gonadal defects in humans. Here, we demonstrate for the first time that germline, as well as somatic expression, of Drosophila Fmr1 (denoted dFmr1), the Drosophila ortholog of FMRP, are necessary in a pathway mediated by piRNAs. Moreover, dFmr1 interacts genetically and biochemically with Aubergine, an Argonaute protein and a key player in this pathway. Our data provide novel perspectives for understanding the phenotypes observed in Fragile X patients and support the view that piRNAs might be at work in the nervous system.

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Section: ▵113mentioning

confidence: 99%

Section: ▵113mentioning

confidence: 99%

The Drosophila fragile X mental retardation protein participates in the piRNA pathway

Bozzetti

Specchia

Cattenoz

et al. 2015

Journal of Cell Science

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“…A Gal4-expressing driver line, T76 [19] that strongly drives expression in male germ line [20] was used to drive expression of the siRNAs.…”

Section: Inhibition Of Vrs By Rnaimentioning

confidence: 99%

“…Flies were created bearing the T76 Gal4 driver [19] and a UASRNAi transgene targeting either CG5538(CG5538 P{KK106939} ) or desat1(desat1 P{KK107747} ) mRNAs. We chose T76 as the driver due to its expression in male germ line at the earliest stages of primary spermatocytes [20]. We tested both T76/CG5538 P{KK106939} and T76/desat1 P{KK107747} males for chromosome loss among progeny.…”

Section: Verifying Thatvrs Is Cg5538 Sirna Knockdownmentioning

confidence: 99%

Versager is expressed at the histone-to-protamine transition during spermiogenesis and is required for embryonic chromosome transmission in Drosophila melanogaster

Binder¹,

Wakimoto²,

Davis³

et al. 2017

Res J Dev Biol

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Background: Chromatin remodeling is one of the most intriguing features of spermiogenesis, during which nuclei undergo drastic morphological changes leading to extensive chromatin compaction. Genetic and cytological accessibility make Drosophila melanogaster a powerful model to study this process. In fruit flies, paternal histones are largely replaced with sperm specific nuclear basic proteins in a highly coordinated manner. This remodeling is essential not only for sperm function but also for proper behavior of paternal chromosomes in the embryo. Our understanding of the changes associated with sperm chromatin and their role in embryonic chromosome behavior is incomplete, and will depend on the identification and characterization of additional components. One such newly identified gene, versager (vrs), is described here. Methods: Chromosome transmission was genetically monitored from vrs Z2566 males using chromosomespecific visible mutations. Recombination and deletion mapping was used to localize the mutation, and DNA sequencing was used to identify the causative lesion. Both in vivo RNAi expression knockdown and rescue by transgene expression of EGFP-tagged protein were used to verify the gene identity. The developmental expression pattern of Vrs was defined based on the EGFP signal in testis relative to RFP-tagged H2Av expression. Behavior of DAPI-stained chromosomes in early embryos from vrs Z2566 males was examined using confocal microscopy. Results: Genetic observations indicated that vrsZ2566 is required in males for high fidelity transmission of paternally derived chromosomes. DNA sequencing revealed that the vrs Z2566 mutation is a missense mutation in Celera predicted gene CG5538 and results in a D2V amino acid substitution. This residue was found to be conserved in Drosophila species and related Diptera. RNAi knockdown of vrs resulted in paternal-effect chromosome loss, and a vrs + -EGFP transgene fully rescued the mutant phenotype. Confocal microscopy of testis revealed nuclear localization of Vrs-EGFP specifically at the canoe stage of spermiogenesis, overlapping with the time of removal of H2Av-RFP at the histone-to-protamine transition. Examination of early stage embryos revealed micronuclei, isolated chromosomes and bridges indicative of chromosome loss events during the first three divisions. Approximately a quarter of later stage embryos arrested with an abnormal number of metaphase and fragmented nuclei. Conclusions: A novel sperm-specific paternal-effect gene, vrs, was identified that is expressed at the histone-to-protamine transition and is important for embryonic chromosome behavior and development. The histone-to-protamine transition may be a developmental period sensitive to perturbations that may lead to embryonic mitotic errors, aneuploidy and developmental arrest.

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“…Homolog conjunction in male flies depends on three proteins, MNM, stromalin-in-meiosis (SNM) (Thomas et al 2005) and Tef (Arya et al 2006), that have been proposed to interact in a conjunction complex (Thomas et al 2005). There were two suggestions that the dtopors-induced anaphase bridges might be dependent on the activity of this conjunction complex.…”

Section: Regulation Of Homolog Separationmentioning

confidence: 99%

Nuclear Structure and Chromosome Segregation inDrosophilaMale Meiosis Depend on the Ubiquitin Ligase dTopors

et al. 2011

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In many organisms, homolog pairing and synapsis at meiotic prophase depend on interactions between chromosomes and the nuclear membrane. Male Drosophila lack synapsis, but nonetheless, their chromosomes closely associate with the nuclear periphery at prophase I. To explore the functional significance of this association, we characterize mutations in nuclear blebber (nbl), a gene required for both spermatocyte nuclear shape and meiotic chromosome transmission. We demonstrate that nbl corresponds to dtopors, the Drosophila homolog of the mammalian dual ubiquitin/small ubiquitin-related modifier (SUMO) ligase Topors. We show that mutations in dtopors cause abnormalities in lamin localizations, centriole separation, and prophase I chromatin condensation and also cause anaphase I bridges that likely result from unresolved homolog connections. Bridge formation does not require mod(mdg4) in meiosis, suggesting that bridges do not result from misregulation of the male homolog conjunction complex. At the ultrastructural level, we observe disruption of nuclear shape, an uneven perinuclear space, and excess membranous structures. We show that dTopors localizes to the nuclear lamina at prophase, and also transiently to intranuclear foci. As a role of dtopors at gypsy insulator has been reported, we also asked whether these new alleles affected expression of the gypsy-induced mutation ct 6 and found that it was unaltered in dtopors homozygotes. Our results indicate that dTopors is required for germline nuclear structure and meiotic chromosome segregation, but in contrast, is not necessary for gypsy insulator function. We suggest that dtopors plays a structural role in spermatocyte lamina that is critical for multiple aspects of meiotic chromosome transmission.A SSOCIATIONS between chromosomes and the nuclear envelope during meiotic prophase are a widely conserved phenomenon important for proper chromosome transmission. In many species, such interactions are required for bouquet formation, an arrangement in which telomeres cluster in association with the nuclear envelope to facilitate homolog pairing and synapsis (reviewed in Scherthan 2007). In Caenorhabditis elegans, analogous interactions between nuclear envelope proteins and chromosomes are mediated by zinc finger proteins that connect chromosomespecific pairing sites to integral nuclear membrane proteins ). These connections establish bridges across the nuclear envelope and allow for interactions between meiotic chromosomes and cytoskeletal actin. Chromosome movements dependent on these connections are important for homolog pairing (Sato et al. 2009).The association of meiotic chromosomes with the nuclear periphery is particularly striking in Drosophila males, in which paired homologs occupy discreet domains closely apposed to the nuclear membrane. The relevance of this organization to meiotic chromosome segregation in this organism, however, has not been explored. Drosophila males have an unconventional meiosis in which homologs pair but do not assemble synapto...

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Molecular Characterization of teflon, a Gene Required for Meiotic Autosome Segregation in Male Drosophila melanogaster

Cited by 25 publications

References 35 publications

The Drosophila fragile X mental retardation protein participates in the piRNA pathway

The Drosophila fragile X mental retardation protein participates in the piRNA pathway

Versager is expressed at the histone-to-protamine transition during spermiogenesis and is required for embryonic chromosome transmission in Drosophila melanogaster

Nuclear Structure and Chromosome Segregation inDrosophilaMale Meiosis Depend on the Ubiquitin Ligase dTopors

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