HDR Gene Editing in HSPCs for Precise Haploinsufficient-Gene Knock-In

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Solution Overview

Problem

Current gene editing technologies for hematopoietic stem cells (HSPCs) face low efficiency and potential genotoxicity due to low cell cycle phases and non-physiological transgene expression, necessitating improved safety and efficiency for therapeutic applications.

Innovation Solution

A method involving targeted integration by homology-directed repair (HDR) in haploinsufficient genes, using a nuclease and guide RNA to introduce a double-strand break, followed by a donor template with homology arms for precise knock-in cassette integration, purging cells with undesired editing outcomes.

Engineering Contradictions & Design Principles

VSEngineering Contradiction Analysis

1Manufacturing precision

If gene editing by HDR is applied in HSPCs, then precise modification of endogenous genes is achieved, but editing efficiency is low

Engineering Contradiction:
Improveprecision of gene editingVSAvoidediting efficiency
Core Design Contradiction:
Manufacturing precisionVSProductivity

Solution Approach 1:

The patent changes the cellular parameter by inducing cells to exit quiescence and enter active cell cycle phases (S/G2) using growth factors and cytokines, thereby increasing the proportion of cells permissive to HDR and improving editing efficiency while maintaining precision

Inventive Principle:
Principle #35Parameter changes

Solution Approach 2:

The patent performs preliminary actions by pre-treating HSPCs with growth factors and cytokines before gene editing to activate cell cycle progression, ensuring cells are in the optimal state for efficient HDR when the editing agent is introduced

Inventive Principle:
Principle #10Preliminary action

2Productivity

If viral vectors with strong promoters are used for gene therapy, then therapeutic efficacy is improved, but risk of insertional mutagenesis increases

Engineering Contradiction:
Improvetherapeutic efficacyVSAvoidinsertional mutagenesis risk
Core Design Contradiction:
ProductivityVSObject-affected harmful factors

Solution Approach 1:

The patent extracts and removes the harmful element (strong viral promoters) from the gene therapy construct, replacing them with endogenous promoters that drive expression only at the target locus, thereby eliminating insertional mutagenesis risk while maintaining therapeutic efficacy

Inventive Principle:
Principle #2Taking out (Extraction)

Solution Approach 2:

The patent introduces an intermediary mechanism (HDR-based precise integration) that allows therapeutic genes to be inserted at specific safe harbor loci rather than randomly, mediating between the need for strong expression and the need to avoid mutagenesis

Inventive Principle:
Principle #24Intermediary (Mediator)

3Productivity

If selection strategies are used to enrich HDR-edited cells, then efficacy is improved, but genotoxic events may still persist

Engineering Contradiction:
Improveproportion of edited cellsVSAvoidgenotoxic events
Core Design Contradiction:
ProductivityVSObject-affected harmful factors

Solution Approach 1:

The patent converts the potentially harmful effect of selective pressure into a benefit by using it to purge out cells with genotoxic editing outcomes (indels, large deletions, translocations) while enriching for cells with correct HDR outcomes, thereby improving both efficacy and safety

Inventive Principle:
Principle #22Blessing in disguise (Convert harm into benefit)

Applied Scientific Principles

This section explains which scientific principles are used to turn an abstract innovation direction into a practical engineering solution.

Function Achieved in This Case

Enhances the efficiency and safety of gene editing in HSPCs by enriching cells with uniform editing outcomes and preserving genome integrity, ensuring long-term survival and engraftment.

Implementation Method 1

targeted integration by homology-directed repair (HDR) in haploinsufficient genes, using a nuclease and guide RNA to introduce a double-strand break, followed by a donor template with homology arms for precise knock-in cassette integration

Methodology Applied
Scientific EffectHomology-directed repair:

Data Source

PatentEP4640830A1Means and methods for safe and efficient gene editing in cells
Publication Date: 2025.10.29 FOND AZIONE TELETHON
  • EP4640830A1 patent drawingFigure 1A~1B
  • EP4640830A1 patent drawingFigure 1C~1D
  • EP4640830A1 patent drawingFigure 2A~2C

AI summary

The invention is directed to means and methods for efficient and safe genetic manipulation of a target cell, wherein a knock-out/knock in strategy is performed via HDR in a target gene that is haploinsufficient (and/or in single copy) in the cell.