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Discover the physiological and developmental functions of X chromosome dosage using new genetic and system models

Project description

X chromosome dosage in development and disease

Sex chromosomes play a crucial role in determining the phenotypic differences between males and females. However, their unequal gene copy distribution can lead to gene product imbalances. To address this, many species have evolved dosage compensation mechanisms, which equalise gene expression between the sexes. When this process fails, it can result in sex-specific disorders or lethality. In this context, the ERC-funded XDose project seeks to understand the mechanisms behind X monosomy-associated lethality by studying sex chromosome dosage in flies. By leveraging fly genetics and genomic techniques, the project will explore the roles of X chromosome dosage in development and its evolutionary significance across insect species, ultimately shedding light on its impact on health and disease.

Objective

Sex chromosomes are critical for controlling the phenotypic differences between males and females. However, they also create an imbalance in the gene products of sex-linked genes, with males having only one copy and females having two copies. To counteract this imbalance, many species evolved a chromosome-wide mechanism known as dosage compensation, which helps equalise gene expression between the sexes. This process is vital, as failed dosage compensation can lead to sex-specific disorders and lethality in several species including humans. Although researchers have made significant progress in understanding the mechanistic basis of dosage compensation, the core questions remain unanswered: why is the lack of X-chromosome compensation lethal and what are the physiological consequences of its absence?
The proposed research aims to identify the mechanisms underlying X monosomy-associated lethality by studying the genes, cells, and developmental processes sensitive to X chromosome doses. This question has been poorly investigated partly due to the difficulties of studying sex chromosome effects. To overcome this barrier, we will investigate flies as these permit the remarkable possibility of generating mosaic animals where sex chromosomes can be genetically manipulated in defined organs, and consequently, functional and molecular analysis.

Here, I will combine classical fly genetics, two novel model systems and cutting-edge genomic techniques to:
1. Identify the functions of X chromosome dosage in fly development.
2. Characterise the ancestral X chromosome compensatory system of all insects.
3. Discover the general shared functions of sex chromosome dosage in development using new insect model species.

Thus, results from this research should have a major impact on our understanding of the importance of sex chromosome dosage in physiology and disease.

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HORIZON-ERC - HORIZON ERC Grants

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Call for proposal

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(opens in new window) ERC-2024-COG

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Host institution

CENTRE NATIONAL DE LA RECHERCHE SCIENTIFIQUE CNRS
Net EU contribution

Net EU financial contribution. The sum of money that the participant receives, deducted by the EU contribution to its linked third party. It considers the distribution of the EU financial contribution between direct beneficiaries of the project and other types of participants, like third-party participants.

€ 1 983 769,00
Address
RUE MICHEL ANGE 3
75794 PARIS
France

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Region
Ile-de-France Ile-de-France Paris
Activity type
Research Organisations
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Total cost

The total costs incurred by this organisation to participate in the project, including direct and indirect costs. This amount is a subset of the overall project budget.

€ 1 983 769,00

Beneficiaries (1)

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