The fly eye: Through the looking glass

Kumar, Justin P.

doi:10.1002/dvdy.24585

Cited by 52 publications

(63 citation statements)

References 176 publications

(213 reference statements)

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“…The degree of eye-to-antenna transformation ranged from some antennal tissue protruding from the eye to the complete transformation of an eye into an antenna ( Figure 5D). Similar homeotic fate transformation phenotypes have been previously described in mutants causing cell proliferation defects in the eye imaginal disc and when eye specification genes such as ey, twin of eyeless (toy), eyes absent (eya), sine oculis (so) or dachshund (dac) are downregulated or misregulated (Duong et al, 2008;Kumar, 2018;Kumar and Moses, 2001).…”

Section: The Roles Of Dtgs1 and Smn In Eye Developmentsupporting

confidence: 70%

Intimate functional interactions between TGS1 and the Smn complex revealed by an analysis of the Drosophila eye development

Maccallini

Bavasso

Scatolini

et al. 2020

Preprint

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Trimethylguanosine synthase 1 (TGS1) is a conserved enzyme that mediates formation of the trimethylguanosine cap on several RNAs, including snRNAs and telomerase RNA. Previous studies have shown that TGS1 binds the Survival Motor Neuron (SMN) protein, whose deficiency causes spinal muscular atrophy (SMA). In addition, TGS1 depletion results in increased hTR levels and telomere elongation in human cells. Here, we analyzed the roles of the Drosophila orthologs of the human TGS1 and SMN genes.We show that the Drosophila TGS1 protein (dTgs1) physically interacts with all subunits of the Drosophila Smn complex (Smn, Gem2, Gem3, Gem4 and Gem5), and that a human TGS1 transgene rescues the mutant phenotype caused by dTgs1 loss. We demonstrate that both dTgs1 and Smn are required for viability of retinal progenitor cells and that downregulation of these genes leads to a reduced eye size. Importantly, overexpression of dTgs1 partially rescues the eye defects caused by Smn depletion, and vice versa. These results suggest that the Drosophila eye model can be exploited for screens aimed at the identification of genes and drugs that modify the phenotypes elicited by Tgs1 and Smn deficiency. These modifiers could help to devise new therapies for SMA and diseases caused by telomerase insufficiency.

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Section: The Roles Of Dtgs1 and Smn In Eye Developmentsupporting

confidence: 70%

Intimate functional interactions between TGS1 and the Smn complex revealed by an analysis of the Drosophila eye development

Maccallini

Bavasso

Scatolini

et al. 2020

Preprint

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“…Compound eye development is understood in great detail in Drosophila melanogaster (WOLFF and READY 1993;DOMINGUEZ and CASARES 2005;KUMAR 2011;KUMAR 2012;CASARES and ALMUDI 2016;KUMAR 2018). In this fly, the eyes as well as the head capsule tissue, ocelli, antennae and maxillary palps develop from the eye-antennal disc.…”

Section: Introductionmentioning

confidence: 99%

Characterization of the genetic architecture underlying eye size variation withinDrosophila melanogasterandDrosophila simulans

Gaspar

Arif

Sumner‐Rooney

et al. 2019

Preprint

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1865 484191 Fax: +44 (0)1865 483242 Running title: Evolution of Drosophila eye size AbstractThe compound eyes of insects exhibit striking variation in size, reflecting adaptation to different lifestyles and habitats. However, the genetic and developmental bases of variation in insect eye size is poorly understood, which limits our understanding of how these important morphological differences evolve. To address this, we further explored natural variation in eye size within and between four species of the Drosophila melanogaster species subgroup. We found extensive variation in eye size among these species, and flies with larger eyes generally had a shorter inter-ocular distance and vice versa. We then carried out quantitative trait loci (QTL) mapping of intra-specific variation in eye size and inter-ocular distance in both D. melanogaster and D. simulans. This revealed that different genomic regions underlie variation in eye size and inter-ocular distance in both species, which we corroborated by introgression mapping in D. simulans. This suggests that although there is a trade-off between eye size and inter-ocular distance, variation in these two traits is likely to be caused by different genes and so can be genetically decoupled. Finally, although we detected QTL for intra-specific variation in eye size at similar positions in D. melanogaster and D. simulans, we observed differences in eye fate commitment between strains of these two species. This indicates that different developmental mechanisms and therefore, most likely, different genes contribute to eye size variation in these species. Taken together with the results of previous studies, our findings suggest that the gene regulatory network that specifies eye size has evolved at multiple genetic nodes to give rise to natural variation in this trait within and among species.2014; KEESEY et al. 2019;RAMAEKERS et al. 2019). Like other drosophilids and other dipterans, this variation in eye size is negatively correlated with face width (inter-ocular distance) and/or antennal size, suggesting trade-offs during the development of eyeantennal tissues that contribute to their final size (NORRY et al. 2000;DOMINGUEZ and CASARES 2005;SUKONTASON et al. 2008;POSNIEN et al. 2012;KEESEY et al. 2019;RAMAEKERS et al. 2019).Studying the genetic basis of eye size variation in Drosophila offers an excellent opportunity to better understand the regulation and evolution of the development of eyes and other tissues, and ultimately how these morphological changes can cause functional differences in vision. For example, strains of D. mauritiana have larger eyes than either D. melanogaster or D. simulans, caused mainly by differences in ommatidial diameter, which is wider in D. mauritiana (POSNIEN et al. 2012; ARIF et al. 2013). QTL mapping has shown that while the larger eyes of D. mauritiana is caused by an X-linked locus of large effect, the reciprocal shorter inter-ocular distance of this species is caused by non overlapping autosomal loci (POSNIEN et al. 2012; ARIF et al. 2013...

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“…There are many other causes involved in the development of pink eyes, such as tightly regulated cell divisions and mechanical processes such as the movement of the morphogenetic furrow in the eye imaginal disc of the larva of D. melanogaster . [ 22,23 ] Moreover, genetic determinants of eye color causally depend on enzymes like RNA polymerase and functional cell metabolism to exert their effect. However, these other causes remain largely constant in populations (or vary in ways not correlated with eye color), and therefore cannot be said to cause the observed phenotypic differences.…”

Section: Genes As Specific Actual Difference‐makersmentioning

confidence: 99%

Genetic Causation in Complex Regulatory Systems: An Integrative Dynamic Perspective

DiFrisco

Jaeger

2020

BioEssays

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The logic of genetic discovery has changed little over time, but the focus of biology is shifting from simple genotype–phenotype relationships to complex metabolic, physiological, developmental, and behavioral traits. In light of this, the traditional reductionist view of individual genes as privileged difference‐making causes of phenotypes is re‐examined. The scope and nature of genetic effects in complex regulatory systems, in which dynamics are driven by regulatory feedback and hierarchical interactions across levels of organization are considered. This review argues that it is appropriate to treat genes as specific actual difference‐makers for the molecular regulation of gene expression. However, they are often neither stable, proportional, nor specific as causes of the overall dynamic behavior of regulatory networks. Dynamical models, properly formulated and validated, provide the tools to probe cause‐and‐effect relationships in complex biological systems, allowing to go beyond the limitations of genetic reductionism to gain an integrative understanding of the causal processes underlying complex phenotypes.

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The fly eye: Through the looking glass

Cited by 52 publications

References 176 publications

Intimate functional interactions between TGS1 and the Smn complex revealed by an analysis of the Drosophila eye development

Intimate functional interactions between TGS1 and the Smn complex revealed by an analysis of the Drosophila eye development

Characterization of the genetic architecture underlying eye size variation withinDrosophila melanogasterandDrosophila simulans

Genetic Causation in Complex Regulatory Systems: An Integrative Dynamic Perspective

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