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Dissecting hepatocyte heterogeneity in liver growth to devise liver gene therapies for pediatric patients

Project description

Gene therapy for inherited metabolic liver diseases

Many metabolic disorders result from mutations in single genes that disrupt essential metabolic pathways. The liver is one of the most important organs of metabolism and hepatocytes are thus prime targets for therapeutic intervention. In vivo genetic engineering of hepatocytes holds great promise for treating these conditions, but long-term success depends on understanding liver cell dynamics. The ERC-funded HEPAGENE project aims to investigate postnatal liver maturation by identifying key proliferative cell subsets and mapping hepatocyte heterogeneity. Project results are expected to support the design of safe gene therapies for inherited metabolic diseases in paediatric patients.

Objective

In vivo genetic engineering of hepatocytes may represent a definitive cure for monogenic metabolic diseases. Integration of the therapeutic transgene into the target cell genome is essential for long-term expression after a single dose early in life and can be achieved by semi-randomly integrating lentiviral vectors or site-specific genome editing. Maintenance of the genetic modification upon hepatocytes proliferation in liver growth and turnover requires targeting the cells underlying these processes. Little is known about post-natal liver growth and how different hepatocyte subsets contribute to it. Unexpectedly, we found that most hepatocytes are quiescent during liver growth, and a fraction of them generate most of the adult tissue. The overall goal of HEPAGENE is to dissect hepatocyte heterogeneity in post-natal liver maturation and unravel its implications for in vivo gene engineering, to ultimately design and develop safe, effective, and durable gene therapies to treat diseases of hepatic metabolism in pediatric patients. To achieve this goal, we will: i) characterize molecular programs of proliferating and quiescent hepatocyte subsets; ii) assess their susceptibility to lentiviral gene transfer and nuclease-mediated gene editing, in both normal mice and in a disease model of methylmalonic acidemia, a severe early-onset disease, taken as paradigmatic of inherited metabolic diseases; iii) estimate clonal dynamics of genetically modified hepatocytes in vivo in non-human primates and analyze gene signatures in human liver samples, to establish a correspondence between murine and primate’s hepatocyte subsets. We will exploit state-of-the-art organoid, single-cell and multi-omic analyses including latest-generation spatial transcriptomics. HEPAGENE will lead to improved understanding of liver biology and gene engineering of hepatocytes, paving the way for novel genomic medicine products that offer hope to children affected by otherwise incurable metabolic diseases

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Topic(s)

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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

UNIVERSITA VITA-SALUTE SAN RAFFAELE
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.

€ 1 993 750,00
Address
VIA OLGETTINA 58
20132 Milano
Italy

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Region
Nord-Ovest Lombardia Milano
Activity type
Higher or Secondary Education Establishments
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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.

€ 1 993 750,00

Beneficiaries (1)

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