Haloperidol-induced isolated lingual dystonia

A 28-year-old woman presenting with agitation and mania with psychotic features developed symptoms of isolated lingual dystonia shortly after the initiation of a haloperidol concentrate regimen.

Case Report: Isolated Lingual Dystonia

Oromandibular dystonia is defined as a focal dystonia that manifests as forceful contractions of the face, jaw, and/or tongue. Lingual dystonia is a rare subtype of oromandibular dystonia that specifically affects the tongue. Multiple etiologies are thought to attribute to oromandibular dystonia, including brain damage, the use of neuroleptic medications, neurodegenerative disorders, metabolic disorders, neurodevelopmental disorders, and viral infections. Idiopathic cases of isolated lingual dystonia are rare and seldom reported in the literature. This report describes a 35-year-old female patient with lingual dystonia that was present at rest and aggravated during speech. Despite detailed history taking and a thorough examination, along with multiple imaging and laboratory studies, no cause could be established and her case was classified as being that of an idiopathic etiology.

Bilateral Pallidal Stimulation with Directional Leads for Primary Focal Lingual Dystonia

There have been limited studies regarding stereotactic and functional neurosurgery for lingual dystonia. Here, we report a patient with primary lingual dystonia who showed significant improvement after bilateral deep brain stimulation (DBS). A 42-year-old woman presented with a 5- to 6-year history of tongue protrusion; however, she lacked a significant medical or medication history before onset. She presented with gradually worsening symptoms and was diagnosed with idiopathic lingual dystonia. Her tongue was injected with botulinum toxin on 6 occasions; however, it had a limited effect. Oral medications were ineffective. She underwent DBS since her involuntary tongue movements were causing nocturnal breathing problems. Directional leads were bilaterally inserted into the internal segment of the globus pallidus (GPi). The directional part of each lead was inserted at the GPi bottom on both sides. The posteromedial contacts were used to deliver stimulation. After 1.5 years, the patient’s Burke-Fahn-Marsden dystonia rating scale score improved from 9 to 1.5 and 2 to 1 for movement and disability, respectively. This case demonstrated the effectiveness of bilateral GPi-DBS. Placing the directional part of the lead in the GPi bottom could improve the stimulation effects.

Case Report: Isolated lingual dystonia

Oromandibular dystonia is defined as a focal dystonia that manifests as forceful contractions of the face, jaw, and/or tongue. Lingual dystonia is a rare subtype of oromandibular dystonia that specifically affects the tongue. Multiple etiologies are thought to attribute to oromandibular dystonia, including brain damage, the use of neuroleptic medications, neurodegenerative disorders, metabolic disorders, neurodevelopmental disorders, and viral infections. Idiopathic cases of isolated lingual dystonia are rare and seldom reported in the literature. This report describes a 35-year-old female patient with lingual dystonia that was present at rest and aggravated during speech. Despite detailed history taking and a thorough examination, along with multiple imaging and laboratory studies, no cause could be established and her case was classified as being that of an idiopathic etiology.

Isolated Lingual Dystonia as the Manifestation of Acute Extrapyramidal Syndrome Induced by Metoclopramide

ABSTRACTOromandibular dystonia is characterized by persistent contractions and repetitive spasms of masticatory muscles surrounding the oral cavity and lingual muscles. The lingual dystonia is rare hyperkinetic movement disorder, often occurring secondary to drugs. The common drugs which can cause acute focal dystonia are neuroleptics, antiemetics, and antipsychotics. Isolated lingual dystonia is a rare form of disabling focal dystonias. We report a young female, who developed tongue dystonia in isolation and responding favorably to antihistaminics. The physicians are advised to make inquiry about administered drugs in isolated lingual dystonia.

Botulinum Neurotoxin Therapy for Lingual Dystonia Using an Individualized Injection Method Based on Clinical Features

Lingual dystonia is a debilitating type of oromandibular dystonia characterized by involuntary, often task-specific, contractions of the tongue muscle activated by speaking or eating. Botulinum neurotoxin (BoNT) has been used to treat lingual dystonia; however, it is known to cause serious complications, such as dysphasia and aspiration. The purpose of this study was to evaluate the efficacy and adverse effects of individualized BoNT therapy for lingual dystonia. One-hundred-and-seventy-two patients (102 females and 70 males, mean age: 46.2 years) with lingual dystonia were classified into four subtypes based on symptoms of involuntary tongue movements: protrusion (68.6%), retraction (16.9%), curling (7.6%), and laterotrusion (7.0%). Patients were treated with BoNT injection into the genioglossus and/or intrinsic muscles via individualized submandibular and/or intraoral routes. Results were compared before and after BoNT therapy. Botulinum neurotoxin was injected in 136 patients (mean: 4.8 injections). Clinical sub-scores (mastication, speech, pain, and discomfort) in a disease-specific rating scale were reduced significantly (p < 0.001) after administration. Comprehensive improvement after BoNT injection, assessed using the rating scale, was 77.6%. The curling type (81.9%) showed the greatest improvement, while the retraction type showed the least improvement (67.9%). Mild and transient dysphasia occurred in 12.5% of patients (3.7% of total injections) but disappeared spontaneously within several days to two weeks. No serious side effects were observed. With careful diagnosis of subtypes and a detailed understanding of lingual muscle anatomy, individualized BoNT injection into dystonic lingual muscles can be effective and safe.