A lack of premature termination codon read-through efficacy of PTC124 (Ataluren) in a diverse array of reporter assays.

The drug molecule PTC124 (Ataluren) has been described as a read-through agent, capable of suppressing premature termination codons (PTCs) and restoring functional protein production from genes disrupted by nonsense mutations. Following the discovery of PTC124 there was some controversy regarding it...

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Main Authors: Stuart P McElroy, Toshifumi Nomura, Leah S Torrie, Emma Warbrick, Ulrike Gartner, Gavin Wood, W H Irwin McLean
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2013-01-01
Series:PLoS Biology
Online Access:https://doi.org/10.1371/journal.pbio.1001593
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author Stuart P McElroy
Toshifumi Nomura
Leah S Torrie
Emma Warbrick
Ulrike Gartner
Gavin Wood
W H Irwin McLean
author_facet Stuart P McElroy
Toshifumi Nomura
Leah S Torrie
Emma Warbrick
Ulrike Gartner
Gavin Wood
W H Irwin McLean
author_sort Stuart P McElroy
collection DOAJ
description The drug molecule PTC124 (Ataluren) has been described as a read-through agent, capable of suppressing premature termination codons (PTCs) and restoring functional protein production from genes disrupted by nonsense mutations. Following the discovery of PTC124 there was some controversy regarding its mechanism of action with two reports attributing its activity to an off-target effect on the Firefly luciferase (FLuc) reporter used in the development of the molecule. Despite questions remaining as to its mechanism of action, development of PTC124 continued into the clinic and it is being actively pursued as a potential nonsense mutation therapy. To thoroughly test the ability of PTC124 to read through nonsense mutations, we conducted a detailed assessment comparing the efficacy of PTC124 with the classical aminoglycoside antibiotic read-through agent geneticin (G418) across a diverse range of in vitro reporter assays. We can confirm the off-target FLuc activity of PTC124 but found that, while G418 exhibits varying activity in every read-through assay, there is no evidence of activity for PTC124.
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institution Kabale University
issn 1544-9173
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language English
publishDate 2013-01-01
publisher Public Library of Science (PLoS)
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spelling doaj-art-71bb01af95ae43f989b1c9563f2c37412025-08-20T03:46:12ZengPublic Library of Science (PLoS)PLoS Biology1544-91731545-78852013-01-01116e100159310.1371/journal.pbio.1001593A lack of premature termination codon read-through efficacy of PTC124 (Ataluren) in a diverse array of reporter assays.Stuart P McElroyToshifumi NomuraLeah S TorrieEmma WarbrickUlrike GartnerGavin WoodW H Irwin McLeanThe drug molecule PTC124 (Ataluren) has been described as a read-through agent, capable of suppressing premature termination codons (PTCs) and restoring functional protein production from genes disrupted by nonsense mutations. Following the discovery of PTC124 there was some controversy regarding its mechanism of action with two reports attributing its activity to an off-target effect on the Firefly luciferase (FLuc) reporter used in the development of the molecule. Despite questions remaining as to its mechanism of action, development of PTC124 continued into the clinic and it is being actively pursued as a potential nonsense mutation therapy. To thoroughly test the ability of PTC124 to read through nonsense mutations, we conducted a detailed assessment comparing the efficacy of PTC124 with the classical aminoglycoside antibiotic read-through agent geneticin (G418) across a diverse range of in vitro reporter assays. We can confirm the off-target FLuc activity of PTC124 but found that, while G418 exhibits varying activity in every read-through assay, there is no evidence of activity for PTC124.https://doi.org/10.1371/journal.pbio.1001593
spellingShingle Stuart P McElroy
Toshifumi Nomura
Leah S Torrie
Emma Warbrick
Ulrike Gartner
Gavin Wood
W H Irwin McLean
A lack of premature termination codon read-through efficacy of PTC124 (Ataluren) in a diverse array of reporter assays.
PLoS Biology
title A lack of premature termination codon read-through efficacy of PTC124 (Ataluren) in a diverse array of reporter assays.
title_full A lack of premature termination codon read-through efficacy of PTC124 (Ataluren) in a diverse array of reporter assays.
title_fullStr A lack of premature termination codon read-through efficacy of PTC124 (Ataluren) in a diverse array of reporter assays.
title_full_unstemmed A lack of premature termination codon read-through efficacy of PTC124 (Ataluren) in a diverse array of reporter assays.
title_short A lack of premature termination codon read-through efficacy of PTC124 (Ataluren) in a diverse array of reporter assays.
title_sort lack of premature termination codon read through efficacy of ptc124 ataluren in a diverse array of reporter assays
url https://doi.org/10.1371/journal.pbio.1001593
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