Post-Stroke Lingual Dystonia: Clinical Description and Neuroimaging Findings

Abstract

Background: Lingual dystonia is extremely rare following stroke. We describe clinical features and neuroimaging findings in a series of 11 patients (seven acute and four chronic) with post-stroke lingual dystonia and review the literature. Methods: This was a case series using a preformed structured proforma and review of literature using a PubMed search. Results: In our case series, all patients had dysarthria as a presenting symptom. Seven patients had acute presentation (six had an ischemic infarct and one had thalamic hemorrhage) and four had chronic presentation (all had infarct). All patients except one had small infarcts, with the majority of them in the basal ganglia and subcortical white matter regions. Additional chronic ischemic lesions were seen in all patients with acute presentation. The majority of the patients with acute (five out of seven; 71.42%) presentation had left-sided involvement on imaging. We could identify only one case of acute post-stroke lingual dystonia following the PubMed search. Three other cases of post-stroke lingual dystonia with chronic presentation have been described; however, these were associated with oromandibular or cranial dystonia. Discussion: Our results, based on brain lesions, suggest that all lingual dystonia patients with acute infarcts had underlying chronic infarcts. Overall, more leftsided than right-sided strokes were observed with post-stroke lingual movement disorders including dystonia; however, the data were not significant (p51). All patients had dysarthria, with only one having mild tongue weakness and only four having facial weakness. This suggests that the lingual dystonia was responsible for the dysarthria rather than weakness in these patients.

A microstructural neural network biomarker for dystonia diagnosis identified by a DystoniaNet deep learning platform

“…Structural abnormalities in the corpus callosum were previously reported within the dystonia spectrum, including laryngeal dystonia, cervical dystonia, and blepharospasm, as well as focal hand dystonia, musician’s dystonia, X-linked dystonia-parkinsonism, and poststroke lingual dystonia (e.g., refs. 19 , 20 , 29 , and 33 – 45 ). These alterations were related to the broader spread of cortical changes in dystonia and attributed to aberrant interhemispheric information transfer.…”

Gaps, Controversies, and Proposed Roadmap for Research in Poststroke Movement Disorders

“…Some MDs have been reported almost exclusively in PSMDs, such as “jerky hand” or “dyskinetic hand” defined as a mixed complex presentation of myoclonus, dystonia, tremor, or choreoathetotic movements, 69 and represent a small fraction of PSMD 3 . Also, some MDs such as lingual dystonia have been poorly defined 5 . There is currently no consensus on the definition of acute, subacute, and chronic/delayed‐onset PSMD, with different authors using different time cutoffs 2,3 .…”

“…However, it is also possible that PSMDs are underrecognized 1 . Although previous studies have highlighted the clinical spectrum, phenomenology, and neuroimaging findings of PSMDs, there are many important knowledge gaps and controversies 2‐6 . In this article, we aim to highlight these gaps and to resolve the controversies with the proposal of a tentative roadmap to advance knowledge and optimize the care of these patients.…”

The Frequency, Predictors and Health Impact of Post-Stroke Extrapyramidal Disorders (Clinical and Epidemiological Study)

“…They affect deep brain regions, including the thalamus (37%), basal ganglia (44%), and occasionally multiple locations across the motor circuit [2] . Examples of post-stroke extrapyramidal disorders (PSEDs) include dystonia, chorea with or without hemiballismus, tremor, parkinsonism, segmental or focal myoclonus, athetosis, choreoathetosis, and asterixis [3][4][5] .…”