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CASE REPORT
Year : 2019  |  Volume : 67  |  Issue : 4  |  Page : 1110--1111

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

Ali S Shalash1, Abdelrahman Ibrahim Abushouk2, Mohammed Yasser Elsherbeny3, Hanan Elrassas4, Taha Kamel1,  
1 Department of Neurology, Faculty of Medicine, Ain Shams University, Cairo, Egypt
2 Medical Research Center, Faculty of Medicine, Ain Shams University, Cairo, Egypt
3 Internal Medicine Department, Faculty of Medicine, Mansoura University, Mansoura, Egypt
4 Department of Psychiatry, Faculty of Medicine, Ain Shams University, Cairo, Egypt

Correspondence Address:
Prof. Ali S Shalash
Faculty of Medicine, Ain Shams University, Ramsis St, 11591, Abbasia, Cairo
Egypt

Abstract

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.



How to cite this article:
Shalash AS, Abushouk AI, Elsherbeny MY, Elrassas H, Kamel T. Refractory Open Jaw Oromandibular Tardive Dystonia with a Sensory Trick, Treated with Botulinum Toxin: A Case Report.Neurol India 2019;67:1110-1111


How to cite this URL:
Shalash AS, Abushouk AI, Elsherbeny MY, Elrassas H, Kamel T. Refractory Open Jaw Oromandibular Tardive Dystonia with a Sensory Trick, Treated with Botulinum Toxin: A Case Report. Neurol India [serial online] 2019 [cited 2019 Oct 15 ];67:1110-1111
Available from: http://www.neurologyindia.com/text.asp?2019/67/4/1110/266235


Full Text



Oromandibular dystonia (OMD) is defined as involuntary, repetitive, twisting spasms of masticatory, lingual, and pharyngeal muscles. Its clinical manifestations may include jaw closing, jaw opening, jaw deviation, facial grimacing, tongue movements, lip pursing, or a combination of these movements.[1] It could be primary (idiopathic) or secondary to several etiologies, especially neuroleptic-induced or tardive dystonia (TD).[2] Botulinum toxin (BTX) can be used to treat refractory OMD cases after exhausting medical treatment options [such as benzodiazepines (e.g., diazepam and clonazepam), skeletal muscle relaxants (e.g., baclofen), and anticholinergic agents (e.g., benzhexol and trihexyphenidyl)]; however, it has different response rates according to OMD type.[2],[3] The jaw-opening OMD (O-OMD) is a distressing form that is caused by different etiologies and is more challenging to treatment.[4]

Several articles described idiopathic OMD cases that were associated with characteristic sensory tricks, such as putting pens or cigarettes between teeth and successfully treated with BTX injection.[2],[5] However, fewer reports described prominent tardive O-OMD and their response to the same treatment.[4] Here, we report a patient with tardive O-OMD, associated with a characteristic sensory trick and successfully treated after two sessions of extraoral, high-dose BTX injection.

 Case History



A 34-year-old male Saudi patient presented to the outpatient clinic at Ain Shams University Hospitals with O-OMD with inability to close his mouth, producing a marked dysarthria of 4-year duration. He had a history of substance-induced psychosis, receiving risperidone (2 mg daily) and mirtazapine (30 mg daily) for over a year. The condition was also associated with involuntary protrusion of the tongue, especially during talking, as well as mild cervical dystonia (of sternomastoid and trapezius muscles). The patient reported improvement of mouth closure and ability to talk after putting something between his teeth, such as cigarettes [Supplementary video 1]. He received anticholinergic drugs (benzhexol 2 mg twice daily for more than 6 months) with no satisfactory response.[MULTIMEDIA:1]

The patient later presented to our clinic and received bilateral electromyography (EMG)-guided onabotulinum toxin A (Botox) injection to the tongue (25 units), lateral pterygoids (LPs) (with an extraoral approach, 75 units for each side), sternocleidomastoids (25 units for each side), trapezius (25 units for each side), and platysma (25 units) muscles. Following BTX injection, he reported a remarkable response with improved ability to close his mouth and talk without using his previous sensory trick [Supplementary video 1]. A mild and transient nasal intonation was noted after the second BTX injection (interval between the two sessions: 3 months). The Abnormal Involuntary Movement Scale score improved from 19 prior to BTX injection to 9, one month after the injection (52.6% improvement).

 Discussion



TD is a complex movement disorder, caused by dopamine receptor blocking drugs, mainly antipsychotics. The most frequent form of TD is oro-facial-lingual dyskinesia, while prominent O-OMD is less frequently described in these patients and is usually associated with tongue stereotyped movements.[6] In this report, we described a patient who presented with prominent O-OMD, following a course of antipsychotic medications. The case was associated with lingual dystonia and a remarkable sensory trick (putting a cigarette between his teeth) that improved his dystonia. The patient's dystonia responded remarkably to extraoral injection of LP muscles with high-dose BTX.

Idiopathic OMD can be relieved by sensory tricks as touching the face, chin, or lips, pinching the neck, bending the neck forward, chewing a gum, or biting something, such as a cigarette or a toothpick.[7] Fewer reports described similar characteristic sensory tricks with tardive O-OMD.[4] A previous study by Schramm et al. suggested that using sensory trick maneuvers in O-OMD reduces agonistic/antagonistic muscle activity and can improve articulation and behavioral activity.[5]

Compared to jaw closing OMD, O-OMD is a more difficult type to manage and can be associated with a significantly higher complication rate.[4] Here, we reported that BTX injection of LP muscles can produce a significant improvement in jaw closure, as well as articulation, with minimal side effects. The LP muscle injection can be done internally or externally; however, the extraoral approach is preferred to minimize the risk of injury to the mandibular nerve and the maxillary artery and to provide an appropriate depth for injection. In our study, we opted for a relatively high dose of BTX (75 units) during LP injection due to the severity of the condition and the younger age of the patient. Most studies in the literature reported using lower doses of BTX for LPs (up to 40 units).[8],[9] The rapid patient response (after two sessions only) and the treatment safety (no development of dysphagia despite using relatively high doses) could be attributed to the use of EMG guidance and avoidance of injection into the submentalis complex. A sustained long-term efficacy has been reported for BTX injection in O-OMD management.[2],[10] Furthermore, an extended follow-up is planned for the current case report.

To recapitulate, this report described a distinct clinical phenomenon of a characteristic sensory trick in a patient with tardive O-OMD. Moreover, it showed that using an extraoral approach for LP muscle injection with high-dose BTX (under EMG guidance) improved jaw closure and articulation and abolished the sensory trick.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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