Genetics of amyotrophic lateral sclerosis: seeking therapeutic targets in the era of gene therapy

N Suzuki, A Nishiyama, H Warita, M Aoki - Journal of human genetics, 2023 - nature.com
Amyotrophic lateral sclerosis (ALS) is an intractable disease that causes respiratory failure
leading to mortality. The main locus of ALS is motor neurons. The success of antisense …

[HTML][HTML] Antisense Oligonucleotides for the Study and Treatment of ALS

BD Boros, KM Schoch, CJ Kreple, TM Miller - Neurotherapeutics, 2022 - Elsevier
Amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disease characterized by
motor neuron loss. ALS is now associated with mutations in numerous genes, many of which …

[HTML][HTML] Trial of Antisense Oligonucleotide Tofersen for SOD1 ALS

TM Miller, ME Cudkowicz, A Genge… - … England Journal of …, 2022 - Mass Medical Soc
Background The intrathecally administered antisense oligonucleotide tofersen reduces
synthesis of the superoxide dismutase 1 (SOD1) protein and is being studied in patients with …

Phase 1–2 Trial of Antisense Oligonucleotide Tofersen for SOD1 ALS

T Miller, M Cudkowicz, PJ Shaw… - … England Journal of …, 2020 - Mass Medical Soc
Background Tofersen is an antisense oligonucleotide that mediates the degradation of
superoxide dismutase 1 (SOD1) messenger RNA to reduce SOD1 protein synthesis …

[HTML][HTML] Design of a randomized, placebo-controlled, phase 3 trial of tofersen initiated in clinically presymptomatic SOD1 variant carriers: the ATLAS study

M Benatar, J Wuu, PM Andersen, RC Bucelli… - …, 2022 - Elsevier
Despite extensive research, amyotrophic lateral sclerosis (ALS) remains a progressive and
invariably fatal neurodegenerative disease. Limited knowledge of the underlying causes of …

Use of real‐world evidence to drive drug development strategy and inform clinical trial design

S Dagenais, L Russo, A Madsen… - Clinical …, 2022 - Wiley Online Library
Interest in real‐world data (RWD) and real‐world evidence (RWE) to expedite and enrich the
development of new biopharmaceutical products has proliferated in recent years, spurred by …

The SOD1-mediated ALS phenotype shows a decoupling between age of symptom onset and disease duration

S Opie-Martin, A Iacoangeli, SD Topp, O Abel… - Nature …, 2022 - nature.com
Superoxide dismutase (SOD1) gene variants may cause amyotrophic lateral sclerosis, some
of which are associated with a distinct phenotype. Most studies assess limited variants or …

Superoxide dismutase 1 in health and disease: how a frontline antioxidant becomes neurotoxic

BG Trist, JB Hilton, DJ Hare, PJ Crouch… - Angewandte Chemie …, 2021 - Wiley Online Library
Cu/Zn superoxide dismutase (SOD1) is a frontline antioxidant enzyme catalysing superoxide
breakdown and is important for most forms of eukaryotic life. The evolution of aerobic …

Antisense drugs make sense for neurological diseases

CF Bennett, HB Kordasiewicz… - Annual review of …, 2021 - annualreviews.org
The genetic basis for most inherited neurodegenerative diseases has been identified, yet
there are limited disease-modifying therapies for these patients. A new class of drugs …

Biomarkers in motor neuron disease: a state of the art review

NS Verber, SR Shepheard, M Sassani… - Frontiers in …, 2019 - frontiersin.org
Motor neuron disease can be viewed as an umbrella term describing a heterogeneous
group of conditions, all of which are relentlessly progressive and ultimately fatal. The …