CRISPR-Cas9 In Vivo Gene Editing for Transthyretin Amyloidosis

Key Information
Source
The New England Journal of Medicine
Year
2021
summary/abstract

Background:
Transthyretin amyloidosis, also called ATTR amyloidosis, is a life-threatening disease characterized by progressive accumulation of misfolded transthyretin (TTR) protein in tissues, predominantly the nerves and heart. NTLA-2001 is an in vivo gene-editing therapeutic agent that is designed to treat ATTR amyloidosis by reducing the concentration of TTR in serum.

Methods:
After conducting preclinical in vitro and in vivo studies, we evaluated the safety and pharmacodynamic effects of single escalating doses of NTLA-2001 in six patients with hereditary ATTR amyloidosis with polyneuropathy, three in each of the two initial dose groups within an ongoing phase 1 clinical study.

Results:
Preclinical studies showed durable knockout of TTR after a single dose. Serial assessments of safety during the first 28 days after infusion in patients revealed few adverse events, and those that did occur were mild in grade. Dose-dependent pharmacodynamic effects were observed. At day 28, the mean reduction from baseline in serum TTR protein concentration was 52% (range, 47 to 56) in the group that received a dose of 0.1 mg per kilogram.

Conclusions:
In a small group of patients with hereditary ATTR amyloidosis with polyneuropathy, administration of NTLA-2001 was associated with only mild adverse events and led to decreases in serum TTR protein concentrations through targeted knockout of TTR.

Abstract Source
https://www.nejm.org/doi/full/10.1056/NEJMoa2107454
DOI
10.1056/NEJMoa2107454
Authors
Julian D. Gillmore, Ed Gane, Jorg Taubel, Justin Kao, Marianna Fontana, Michael L. Maitland, Jessica Seitzer, Daniel O’Connell, Kathryn R. Walsh, Kristy Wood, Jonathan Phillips, Yuanxin Xu
Organisation
National Amyloidosis Centre, UCL, UK; Royal Free Hospital and Richmond Pharmacology, UK ; St. George’s University of London, UK; New Zealand Clinical Research, New Zealand ; University of Auckland, New Zealand ; Auckland City Hospital, New Zealand; Intellia Therapeutics, USA; Regeneron Pharmaceuticals, New York