Objective: Efforts to understand laryngeal dystonia (LD) pathophysiology using neuroimaging techniques have demonstrated altered brain structure and function in patients compared to healthy controls^1-5. However, the interpretation of prior findings is limited by differences in imaging modalities, scanning protocols, analytical techniques, and patient inclusion criteria across studies. Our objective was to consolidate reported findings across neuroimaging studies of LD and identify a robust set of abnormal brain regions contributing to the pathophysiological signature of the disorder.

Background: LD is an isolated, task-specific, focal dystonia characterized by intermittent spasms of laryngeal muscles impairing speech production⁶. Although recent studies have demonstrated neural alterations in LD, the consistency of findings across studies is not well-established, limiting their translational applicability.

Method: We conducted a systematic literature search to identify studies reporting stereotactic coordinates of peak structural and functional abnormalities in LD patients compared to healthy controls, followed by a coordinate-based activation likelihood estimation meta-analysis.

Results: A total of 21 functional and structural neuroimaging studies, including 31 experiments in 521 LD patients and 448 healthy controls, met the study inclusion criteria. The multimodal meta-analysis of these studies identified abnormalities in the bilateral primary motor cortices, the left inferior parietal lobule and striatum, the right insula and supplementary motor area in LD patients compared to healthy controls.

Conclusion: The identified meta-analytical findings reinforce the current view of dystonia as a neural network disorder and consolidate evidence for future investigations probing these targets with new therapies.

References: 1. Battistella G, Fuertinger S, Fleysher L, Ozelius LJ, Simonyan K. Cortical sensorimotor alterations classify clinical phenotype and putative genotype of spasmodic dysphonia. European Journal of Neurology 2016;23(10):1517-1527.
2. Hanekamp S, Simonyan K. The large-scale structural connectome of task-specific focal dystonia. Hum Brain Mapp 2020;41(12):3253-3265.
3. Haslinger B, Erhard P, Dresel C, Castrop F, Roettinger M, Ceballos-Baumann AO. “Silent event-related” fMRI reveals reduced sensorimotor activation in laryngeal dystonia. Neurology 2005;65(10):1562-1569.
4. Mantel T, Dresel C, Welte M, et al. Altered sensory system activity and connectivity patterns in adductor spasmodic dysphonia. Scientific Reports 2020;10(1):10179-10179.
5. Simonyan K, Ludlow CL. Abnormal Activation of the Primary Somatosensory Cortex in Spasmodic Dysphonia: An fMRI Study. Cerebral Cortex 2010;20(11):2749-2759.
6. Guiry S, Worthley A, Simonyan K. A separation of innate and learned vocal behaviors defines the symptomatology of spasmodic dysphonia. Laryngoscope 2019;129(7):1627-1633.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/structural-and-functional-brain-alterations-in-laryngeal-dystonia-a-coordinate-based-activation-likelihood-estimation-meta-analysis/