Refractory Open Jaw Oromandibular Tardive Dystonia with a Sensory Trick, Treated with Botulinum Toxin: A Case Report

• Published in: Neurology India Vol. 67; no. 4; pp. 1110 - 1111
• Main Authors: Shalash, Ali, Abushouk, Abdelrahman, Elsherbeny, Mohammed, Elrassas, Hanan, Kamel, Taha
• Format: Journal Article
• Language: English
• Published: India Wolters Kluwer India Pvt. Ltd 01.07.2019; Medknow Publications and Media Pvt. Ltd; Medknow Publications & Media Pvt. Ltd
• Subjects: Adult; Botulinum toxin; Botulinum toxins; Botulinum Toxins - administration & dosage; Botulinum Toxins - pharmacology; Care and treatment; Case reports; Cigarettes; Drug dosages; Drug resistance; Dystonia; Dystonic Disorders - drug therapy; Electromyography; Facial Muscles - drug effects; Humans; Jaw; Male; Medical research; Mouth Diseases - drug therapy; Neuromuscular Agents - administration & dosage; Neuromuscular Agents - pharmacology; Torticollis - drug therapy; Treatment resistance; oromandibular dystonia; case report; sensory trickKey Message: This report described a distinct clinical phenomenon of a characteristic sensory trick in a patient with tardive O-OMD. Moreover; Botulinum toxin; it showed that using an extraoral approach for lateral pterygoid muscle injection with high-dose BTX (for two sessions under electromyography guidance) improved jaw closure and articulation and abolished the sensory trick; sensory trick
• ISSN: 0028-3886; 1998-4022
• DOI: 10.4103/0028-3886.266235

Jaw-opening oromandibular dystonia (O-OMD) is a clinical subtype of OMD, commonly resistant to treatment. Here, we report a distinct case of tardive O-OMD with a characteristic sensory trick, successfully treated with high-dose botulinum toxin (BTX) injection. A 34-year-old male patient presented with involuntary jaw opening, tongue protrusion, dysarthria, and mild cervical dystonia. The patient reported improved abilities to talk and close his mouth after putting something, like a cigarette, between his teeth. After an unsuccessful treatment with anticholinergic medications, the patient received electromyography-guided BTX injection to the lateral pterygoids (through an extraoral approach), sternocleidomastoids, trapezius, tongue, and platysma muscles. Following the injection, the patient reported marked improvements in his ability to talk and close his mouth without using his sensory trick. One month later, we detected a 58.2% improvement in the Abnormal Involuntary Movement Scale score. Therefore, high-dose BTX injection may be an effective alternative in refractory O-OMD.