Long-term hydromethylthionine treatment is associated with delayed clinical onset and slowing of cerebral atrophy in a pre-symptomatic P301S MAPT mutation carrier

Peter Bentham; Roger T. Staff; Bjoern O. Schelter; Helen Shiells; Charles R. Harrington; Claude M. Wischik

doi:10.3233/JAD-210390

Long-term hydromethylthionine treatment is associated with delayed clinical onset and slowing of cerebral atrophy in a pre-symptomatic P301S MAPT mutation carrier

Peter Bentham, Roger T. Staff, Bjoern O. Schelter, Helen Shiells, Charles R. Harrington, Claude M. Wischik^*

^*Corresponding author for this work

Research output: Contribution to journal › Article › peer-review

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Abstract

One of the mutations in the microtubule-associated protein tau, P301S, is causative for dominantly inherited frontotemporal dementia characterized by extensive tau pathology for which no licensed treatment is available. Hydromethylthionine is a potent tau aggregation inhibitor. We report treatment of an asymptomatic carrier of the P301S mutation using hydromethylthionine over a 5-year period beginning at the mean age of onset of clinical decline in the family. During the period of treatment, the rates of progression of cerebral atrophy were reduced by 61%-66% in frontal and temporal lobes, and the patient remained clinically asymptomatic.

Original language	English
Pages (from-to)	1017-1023
Number of pages	7
Journal	Journal of Alzheimer's Disease
Volume	83
Issue number	3
Early online date	28 Sept 2021
DOIs	https://doi.org/10.3233/JAD-210390
Publication status	Published - 28 Sept 2021

Bibliographical note

ACKNOWLEDGMENTS
We thank the patient for permitting publication of the present report.
Authors’ disclosures available online (https://www.j-alz.com/manuscript-disclosures/21-0390r1).

Keywords

Frontotemporal dementia
hydromethylthionine
leucomethylthioninium
microtubule-associated protein tau
P301S

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

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Bentham_et_al_Long_term_Journal_of_alzheimers_disease_vor
© 2021 – The authors. Published by IOS Press. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (CC BY 4.0). https://creativecommons.org/licenses/by/4.0/
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Long-term hydromethylthionine treatment is associated with delayed clinical onset and slowing of cerebral atrophy in a pre-symptomatic P301S MAPT mutation carrier. / Bentham, Peter; Staff, Roger T.; Schelter, Bjoern O. et al.
In: Journal of Alzheimer's Disease, Vol. 83, No. 3, 28.09.2021, p. 1017-1023.

Research output: Contribution to journal › Article › peer-review

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abstract = "One of the mutations in the microtubule-associated protein tau, P301S, is causative for dominantly inherited frontotemporal dementia characterized by extensive tau pathology for which no licensed treatment is available. Hydromethylthionine is a potent tau aggregation inhibitor. We report treatment of an asymptomatic carrier of the P301S mutation using hydromethylthionine over a 5-year period beginning at the mean age of onset of clinical decline in the family. During the period of treatment, the rates of progression of cerebral atrophy were reduced by 61%-66% in frontal and temporal lobes, and the patient remained clinically asymptomatic.",

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AU - Wischik, Claude M.

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Long-term hydromethylthionine treatment is associated with delayed clinical onset and slowing of cerebral atrophy in a pre-symptomatic P301S MAPT mutation carrier

Abstract

Bibliographical note

Keywords

UN SDGs

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