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 left-sided than right-sided strokes were observed with post-stroke lingual movement disorders including dystonia; however, the data were not significant (p = 1). 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.

Keywords: Dystonia; magnetic resonance images; stroke.

Figure 1. Diffusion-weighted and Apparent Diffusion Coefficient Sequences of Magnetic Resonance Images of Brain. (A,B) Case 1: acute infarct in left basal ganglia. (C,D) Case 2: acute infarct in right basal ganglia, right corona radiata, and subcortical white matter. (E,F) Case 3: acute infarct in left basal ganglia, left corona radiata and subcortical white matter. (G,H) Case 4: acute infarct in the right basal ganglia and adjacent white matter. (I,J) Case 5: acute infarct in the left basal ganglia and adjacent white matter. (K,L) Case 6: acute infarct in the left corona radiata. (M,N) Case 7: left thalamic hemorrhage. Non-contrast computed tomography of head. (O) Case 8: chronic lacunar infarcts in the right basal ganglia and right parietal region. Axial T2 fluid-attenuated inversion recovery image. (P–R) Axial T2-weighted sequences of magnetic resonance images of brain. (P) Case 9: chronic infarcts in left posterior cerebral artery region, left thalamus and lacunar infarcts in bilateral basal ganglia). (Q) Case 10: chronic infarcts in left frontal subcortical region. (R) Case 11: chronic infarct of left middle cerebral artery region.

Figure 2. Lesions Causing Post-stroke Lingual Dystonia. This figure (top row) illustrates the imaging lesion localization in patients with post-stroke lingual dystonia described in the literature. Case 1 depicts acute post-stroke lingual dystonia, cases 2 and 3 are patients with chronic post-stroke oromandibular (lingual) dystonia, and case 4 is a patient with chronic post-stroke cranial lingual dystonia. The bottom two rows illustrate the imaging lesion localization in our patients with post-stroke abnormal lingual movements. Cases 1–7 are acute post-stroke lingual dystonia patients whereas cases 8–11 are chronic post-stroke lingual dystonia patients. Red lesions represent hemorrhagic stroke and blue lesions represent acute ischemic stroke whereas brown represents chronic ischemic stroke.