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A novel and empowered TARGETed gene addition approach at a relevant microglia locus for the treatment of inherited NeuroMetabolic Diseases

Description du projet

Une technologie de thérapie génique des cellules souches hématopoïétiques plus efficace pour traiter les maladies neurométaboliques

Au cours de la dernière décennie, la thérapie génique, notamment celle s’appuyant sur des cellules souches hématopoïétiques (CSH), a démontré un potentiel thérapeutique sans précédent grâce à des innovations dans la conception des vecteurs, la manipulation des cellules souches et l’administration. Récemment, le développement d’approches de thérapie génique CSH pour les maladies neurométaboliques (NMD), des maladies monogénétiques qui agissent sur l’activité métabolique des lysosomes ou des peroxysomes, a suscité un vif intérêt. Dans le cadre de ces maladies, les défaillances du système nerveux provoquent la mort des neurones, l’activation des cellules microgliales, la démyélinisation et la dégénérescence des axones. Le projet TARGETNMDs, financé par l’UE, entend développer un traitement efficace pour les NMD fondé sur une approche d’addition de gènes qui ciblent les CSH sur un locus microglial nouvellement identifié. La correction des défaillances sur ce locus CSH pourrait générer une progéniture de type microglial affichant un potentiel thérapeutique inédit.

Objectif

Hematopoietic stem cell (HSC) gene therapy based on self-inactivating integrating vectors has proven unprecedented therapeutic potential in inherited neurometabolic diseases (NMDs). However, phenotypic effects are delayed after treatment likely due to the slow replacement of resident microglia by transplant-derived cells that hampers the broad application of this approach. Moreover, unregulated gene expression driven by the in-use promoters could in the long term cause unwanted effects. Finally, recent events suggest that the treated patients might be at risk of developing side effects related to vector integration. Therefore, novel strategies anticipating therapeutic benefit and reducing these potential safety concerns are desirable to address the still unmet medical need of NMD patients.
Our long-term goal is to develop a novel, broadly effective and safe treatment platform for NMDs based on a newly empowered HSC targeted gene addition approach at a newly identified microglia locus. Our central hypothesis is that correcting the gene defect by targeted addition at this locus in HSCs of patients affected by NMDs could generate in a timely manner a microglia-like progeny endowed with unprecedented therapeutic potential. Indeed, based on our recent findings, gene editing and targeted integration at this locus are expected to uniquely favor the timely engraftment and efficient, rapid myeloid/microglia differentiation of transplanted, edited HSCs in the recipients’ brain, and to induce robust and regulated expression of the integrated transcript in transplant-derived microglia-like cells. Based on this hypothesis, we aim at developing a targeted gene addition approach at the newly selected microglia locus for correcting the underlying genetic defect in HSCs and obtaining proof of concept of its therapeutic potential in NMDs animal models. Thus, the proposed work could generate the basis for a novel treatment platform for these devastating conditions.

Régime de financement

HORIZON-ERC - HORIZON ERC Grants

Institution d’accueil

UNIVERSITA DEGLI STUDI DI PADOVA
Contribution nette de l'UE
€ 2 495 250,00
Coût total
€ 2 495 250,00

Bénéficiaires (1)