ArticleBrain : a journal of neurology2013
Beneficial effects of bumetanide in a CaV1.1-R528H mouse model of hypokalaemic periodic paralysis.
Article in Brain : a journal of neurology, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT02582476 (A Randomised, Double-blind, Placebo-controlled, Phase II Clinical Trial With a Cross-over Design Assessing Efficacy of a Single Dose of Bumetanide in Reducing Focal Attack Severity in Hypokalaemic Periodic Paralysis Assessed Using the McManis Protocol), which is not on this map. Cited by 20 papers, 1 of them a synthesis that pooled it.
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The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
A Randomised, Double-blind, Placebo-controlled, Phase II Clinical Trial With a Cross-over Design Assessing Efficacy of a Single Dose of Bumetanide in Reducing Focal Attack Severity in Hypokalaemic Periodic Paralysis Assessed Using the McManis Protocol
Who cites it
20 citing papers in PubMed, 1 synthesis or guideline pooled it.
- Targeted Therapies for Skeletal Muscle Ion Channelopathies: Systematic Review and Steps Towards Precision Medicine.Journal of neuromuscular diseases · 2021Pooled it
- Article
- Review
- Molecular genetics of skeletal muscle channelopathies.Journal of human genetics · 2025Review
- Periodic paralysis.Handbook of clinical neurology · 2024Review
- Optical measurement of gating pore currents in hypokalemic periodic paralysis model cells.Disease models & mechanisms · 2023Article
- Novel γ-sarcoglycan interactors in murine muscle membranes.Skeletal muscle · 2022Article
- Autophagy is affected in patients with hypokalemic periodic paralysis: an involvement in vacuolar myopathy?Acta neuropathologica communications · 2021Article
- New Challenges Resulting From the Loss of Function of NaFrontiers in pharmacology · 2021Review
- Treatment Updates for Neuromuscular Channelopathies.Current treatment options in neurology · 2020Review
- Myotonia in a patient with a mutation in an S4 arginine residue associated with hypokalaemic periodic paralysis and a concomitant synonymous CLCN1 mutation.Scientific reports · 2019Article
- Recovery from acidosis is a robust trigger for loss of force in murine hypokalemic periodic paralysis.The Journal of general physiology · 2019Article
- IncreasedNeurology. Genetics · 2018Article
- When muscle CaThe Journal of physiology · 2018Review
- Review of the Diagnosis and Treatment of Periodic Paralysis.Muscle & nerve · 2018Review
- Phospholemman, a major regulator of skeletal muscle NaExperimental and therapeutic medicine · 2017Article
- Therapeutic Approaches to Genetic Ion Channelopathies and Perspectives in Drug Discovery.Frontiers in pharmacology · 2016Review
- Channelopathies of skeletal muscle excitability.Comprehensive Physiology · 2015Review
- Treatment and management of neuromuscular channelopathies.Current treatment options in neurology · 2014Article
- Ageing contributes to phenotype transition in a mouse model of periodic paralysis.JCSM rapid communicationsArticle
Corrections and comments
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Authors and funding
3 authors.
Funding
Abstract
Transient attacks of weakness in hypokalaemic periodic paralysis are caused by reduced fibre excitability from paradoxical depolarization of the resting potential in low potassium. Mutations of calcium channel and sodium channel genes have been identified as the underlying molecular defects that cause instability of the resting potential. Despite these scientific advances, therapeutic options remain limited. In a mouse model of hypokalaemic periodic paralysis from a sodium channel mutation (NaV1.4-R669H), we recently showed that inhibition of chloride influx with bumetanide reduced the susceptibility to attacks of weakness, in vitro. The R528H mutation in the calcium channel gene (CACNA1S encoding CaV1.1) is the most common cause of hypokalaemic periodic paralysis. We developed a CaV1.1-R528H knock-in mouse model of hypokalaemic periodic paralysis and show herein that bumetanide protects against both muscle weakness from low K+ challenge in vitro and loss of muscle excitability in vivo from a glucose plus insulin infusion. This work demonstrates the critical role of the chloride gradient in modulating the susceptibility to ictal weakness and establishes bumetanide as a potential therapy for hypokalaemic periodic paralysis arising from either NaV1.4 or CaV1.1 mutations.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.