Lee, Chang Woo; Krüger, Marie T; Akram, Harith; Zrinzo, Ludvic; Rubin, John; Birchall, Martin A; Fishman, Jonathan M; (2024) Central Mechanisms and Pathophysiology of Laryngeal Dystonia: An Up-to-Date Review. Journal of Voice 10.1016/j.jvoice.2024.07.007. (In press).

Text Lee et al. 2024 LD systematic review.pdf - Accepted Version Access restricted to UCL open access staff until 13 August 2025. Download (1MB)

Abstract

Objective: Laryngeal dystonia (LD), previously termed spasmodic dysphonia, is an isolated focal dystonia that involves involuntary, uncontrolled contractions of the laryngeal muscles during speech. It is a severely disabling condition affecting patients’ work and social lives through prevention of normal speech production. Our understanding of the pathophysiology of LD and available therapeutic options are currently limited. The aim of this short review is to provide an up-to-date summary of what is known about the central mechanisms and the pathophysiology of LD. // Methods: A systematic review of the literature was performed searching Embase, CINHAL, Medline, and Cochrane with the cover period January 1990–October 2023 with a search strategy ((“Laryngeal dystonia” OR “Spasmodic dysphonia”) AND (“Central Mechanism” OR “Pathophysiology”)). Original studies involving LD patients that discussed central mechanisms and/or pathophysiology of LD were chosen. // Results: Two hundred twenty-six articles were identified of which 27 articles were included to formulate this systematic review following the screening inclusion and exclusion criteria. LD is a central neurological disorder involving a multiregional altered neural network. Affected neural circuits not only involve the motor control circuit, but also the feedforward, and the feedback circuits of the normal speech production neural network, involving higher-order planning, somatosensory perception and integration regions of the brain. // Conclusion: Speech production is a complex process, and LD is a central neurological disorder involving multiregional neural network connectivity alteration reflecting this. Neuromodulation targeting the central nervous system could therefore be considered and explored as a new potential therapeutic option for LD in the future, and should assist in elucidating the underlying central mechanisms responsible for causing the condition.

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