CRISPR Gene editing in Neuroscience

Introduction
Since it was first used to edit the mammalian genome,1 the targetable gene editing tool CRISPR (clustered regularly interspaced short palindromic repeats) has become widely accessible to researchers. Compared to older gene editing technologies, such as zinc-finger nucleases (ZFNs) and transcription activator-like effector nucleases (TALENs), CRISPR has significant advantages: it is more efficient, faster to set up, and can be multiplexed: several DNA loci can be targeted in one experiment.2 CRISPR’s potential in neuroscience ranges from investigating fundamental processes underlying brain function and development, to modelling neurological diseases in both animals and cells, and perhaps to CRISPR-based therapies. This review discusses CRISPR’s current applications in cell and animal models aiming to clarify brain function and dysfunction, and some of the challenges that currently limit CRISPR’s use in neuroscience.

What is CRISPR?
The CRISPR gene editing system has been identified in and is derived from part of the prokaryote adaptive immune system, which defends against invading viruses or plasmids by specifically cleaving exogenous DNA. Adapting CRISPR for gene editing exploits the ability of CRISPR nucleases to make predictable DNA breaks at specifically targeted sequences. There are three types of CRISPR system (I, II and III) of which type II is most widely used in gene editing (see figure 1 below).

Figure 1. Using the Cas9 CRISPR system for gene editing. Guide RNA (gRNA) is composed of a scaffold sequence (required for binding between Cas9 and the gRNA) and a 20 base pair sequence which is designed complementary to the DNA target. The DNA target must be upstream of a proto-spacer adjacent motif (PAM) sequence, which is required for DNA cleavage. Cas9 nuclease is directed to the target DNA by base pairing between the target DNA and gRNA, resulting in a double-stranded DNA break. The double-stranded break can be repaired by non-homologous end-joining (NHEJ) or homologous recombination (HR). NHEJ results in either restoration of the wild-type sequence, insertions or deletions (indels). Indels vary in length and can cause frame-shift mutations, leading to premature stop codons and gene knockout. HR requires a DNA template with homologous regions up- and down-stream of the break. Therefore, a DNA repair template can be designed in order to introduce precise insertions, deletions or point mutations.
Figure 1. Using the Cas9 CRISPR system for gene editing.
Guide RNA (gRNA) is composed of a scaffold sequence (required for binding between Cas9 and the gRNA) and a 20 base pair sequence which is designed complementary to the DNA target. The DNA target must be upstream of a proto-spacer adjacent motif (PAM) sequence, which is required for DNA cleavage. Cas9 nuclease is directed to the target DNA by base pairing between the target DNA and gRNA, resulting in a double-stranded DNA break. The double-stranded break can be repaired by non-homologous end-joining (NHEJ) or homologous recombination (HR). NHEJ results in either restoration of the wild-type sequence, insertions or deletions (indels). Indels vary in length and can cause frame-shift mutations, leading to premature stop codons and gene knockout. HR requires a DNA template with homologous regions up- and down-stream of the break. Therefore, a DNA repair template can be designed in order to introduce precise insertions, deletions or point mutations.
First, guide RNA is designed complementary to a DNA target. Next, the guide RNA complexes with Cas9 nuclease (the CRISPR effector), followed by base-pairing between the guide RNA and its DNA target, which directs Cas9 to cleave the DNA. Finally, double stranded DNA breaks are repaired by either non-homologous end-joining (NHEJ) or homologous recombination (HR). NHEJ makes insertion or deletion mutations (indels) of varying lengths, usually resulting in a premature stop codon and gene knockout. Alternatively, DNA with the desired insertion, deletion or point mutation can be introduced to act as a repair template during HR, leading to precise mutations in the DNA.3

Cas9 is a nuclease, but CRISPR is not limited to nuclease activity. Inactivation of both catalytic domains in dead (d)Cas9 renders the nuclease unable to cleave DNA, but it can prevent transcription by steric hindrance in CRISPR interference (CRISPRi). CRISPRi can be enhanced by complexing dCas9 to repressors, for reversible gene knockdown, whereas dCas9-activator complexes can be used for reversible overexpression. In addition, dCas9 complexed to epigenetic modifiers can be used for methylation or histone modifications, and Cas9 with a fluorescent molecule can tag genomic loci.4 The range of functional domains coupled to Cas9 is expanding, linking CRISPR to advances in our understanding of genetic processes and our ability to manipulate them. One area where CRISPR has been readily adopted is in modelling neurological disease with human induced pluripotent stem cells (hiPSCs).

More at

http://www.acnr.co.uk/2017/02/crispr-for-gene-editing-in-neuroscience-and-neurological-disease/

Here’s an email I got from the researchers responsible for this work:

In theory, CRISPR gene editing could be used to fix the polyglutamine expansion in SCA1. The Doudna lab (and others) are researching how CRISPR might be applied to treat a similar disorder, Huntington’s disease. The nature of these disorders suggests that insight gained about one may very well be applicable to the other. In my opinion, there are two main roadblocks to achieving CRISPR-based treatments in humans: delivery and accuracy.

Delivery: We know what changes we need to make in DNA to alleviate disease, but getting the editing molecules into the affected tissues to actually edit the DNA is a huge challenge. Direct injection into the brain may end up being the best option, or we may be able to develop less invasive molecular shuttles that bring editing components from the bloodstream to the brain. This is an area of intense, active research in the gene editing community right now.

Accuracy (safety): Once CRISPR editing components get into disease-affected cells, we need to make sure they only change DNA in the intended place. Right now, the Cas9 protein sometimes cuts in the wrong spot, which could cause cancer. This problem is slowly being solved by researchers, but it remains to be seen how accurate the CRISPR system will be in humans. Any therapy must therefore be tested rigorously to ensure that the side effects are not worse than the condition it aims to treat.

Thus, even for illnesses with a well-understood genetic cause (like SCA1), developing a treatment or cure requires much more scientific research, extensive safety and efficacy studies, clinical trials, and more. Therapeutic gene editing is still in the very early stages of development. The IGI is years away from turning our current CRISPR research into a real treatment option for any disorder. We are committed to seeing this through, but are not there yet.

Please visit https://innovativegenomics.org/ to learn more and follow our updates. You may wish to get in touch with patient advocacy and support groups for more personal guidance. For example, MyGene2 and the Rare Genomics Institute are excellent organizations that connect patients and their families with both patient advocates and scientific researchers who are studying their specific condition.

Thank you for taking the time to write to us. We wish you and your family well.

Sincerely,
Megan Hochstrasser, PhD

Yes.
Another potential step forward that needs more research !