Work

Lingual Dystonia Following Thalamic Infarction in a Patient on Methotrexate Therapy for Hidradenitis Suppurativa

Year: 2025

Type: article

Abstract: Lingual dystonia is a rare form of focal dystonia involving involuntary, repetitive, and often painful muscle contractions of the tongue, which can lead to abnormal tongue posturing or protrusion, dysarthria, and/or dysphagia. Lingual dysotnia can be primary or secondary to various conditions. The thalamus is a key relay center between the motor cortex and basal ganglia, and damage here can disrupt motor signaling pathways, possibly leading to focal dystonia, like lingual dystonia. Methotrexate (MTX) can cause acute to subacute neurological complications, which are linked with high doses of MTX, while chronic MTX neurotoxicity develops more slowly, resulting in persistent focal neurological deficits. We present a 56-year-old male patient on MTX therapy who developed an isolated lingual dystonia as a chronic presentation of a small infarction in the posterolateral thalamus. The finding of thalamic infarction on brain CT and the lack of clinical improvement in dystonia symptoms after prolonged MTX withdrawal from therapy indicate that lingual dystonia could be directly causally related to thalamic insult, while it is less likely that lingual dystonia is a side effect of MTX therapy. This case provides us with new knowledge in determining the etiology of lingual dystonia in the light of thalamic infarction and MTX neurotoxicity.

Source: Cureus

DOI: 10.7759/cureus.82974

Authors: Iva Sarac, Fran Borovecki, Liborija Lugovic Mihic, Hanna Pasic, Helena Sarac

Institutions: University of Zagreb, University Hospital Centre Zagreb

Language: English

Topic: Neurological and metabolic disorders

Subfield: Endocrinology, Diabetes and Metabolism

Field: Medicine

Domain: Health Sciences

SDG: Good health and well-being

Open Access status: diamond

OpenAlex ID: W4409812287