Introduction
Levodopa‐induced dyskinesia (LID) is a recognized motor response complication affecting 30% to 80% of patients with Parkinson's disease on chronic levodopa (l‐dopa) therapy.1, 2 It is typically choreiform, and involves all parts of the body, especially the head and neck. Besides chorea, rarer types of LIDs include myoclonic dyskinesia, ocular dyskinesia, and respiratory dyskinesia.1 Oromandibular dystonia, following l‐dopa therapy, typically raises the suspicion of an underlying diagnosis of multiple system atrophy (MSA). Herein, we report a case of idiopathic Parkinson's disease (IPD), in which the patient developed a unique oromandibular dystonia following l‐dopa therapy.
Case Report
A 59 year‐old‐man was diagnosed with idiopathic Parkinson's disease (IPD) at 53 years of age, after having experienced bilateral hand tremors and slowness of movement in the preceding year. There was no family history of Parkinson's disease (PD). Clinical examination revealed asymmetric parkinsonism with resting hand tremor, limb rigidity, and bradykinesia that were more prominent on the right. There were no cerebellar signs, gaze palsy, or autonomic dysfunction. Magnetic resonance imaging (MRI) of his brain was normal. He was treated with l‐dopa and selegiline, with excellent l‐dopa response for four years, after which he started to experience neck and jaw dyskinesia. The dyskinesia occurred one hour after each l‐dopa dose (100 mg) and lasted for one hour. It involved predominantly his jaw leading to an almost rhythmic jaw opening and closing dyskinesia. This movement was associated with similar rhythmic vertical neck movements (yes‐yes) and frequent eye‐blinking. There was no dyskinesia in other body parts (Supporting Video 1). Owing to this troublesome dyskinesia, he self‐adjusted the l‐dopa dose to once daily, resulting in him being OFF throughout the day. Multiple adjustments of l‐dopa dose (to as low as 50 mg daily) and frequency, and the addition of amantadine and clonazepam, were ineffective in ameliorating the dyskinesia. Because of his disabling dyskinesia, he was planned for deep brain stimulation surgery given his excellent response to l‐dopa with a 52% improvement in the Unified Parkinson's Disease Rating Scale (UPDRS) part III motor examination score (OFF score 65 and ON score 31). While awaiting surgery, he was given a trial of botulinum toxin injections into the lateral pterygoids. The severity of jaw opening dyskinesia was ameliorated by 40% with improvement in his masticatory function and pain (Supporting Video 2).
Discussion
Facial and jaw dystonias are recognized side effects of l‐dopa therapy in patients with multiple system atrophy (MSA),3 and more recently, a patient with progressive supranuclear palsy (PSP).4 Except for one case report,5 prominent oromandibular dystonia as a manifestation of LID is rare in PD. Our patient had a unique peak‐dose dyskinesia‐dystonia with jaw opening spasms in an almost rhythmic manner.5 It is interesting to note that our case is similar to a case by Pfeiffer et al.,5 in which the jaw dystonia occurred during the peak‐dose. Furthermore, there was an improvement in dystonia with botulinum toxin injections in both our cases. In conclusion, prominent l‐dopa‐induced oromandibular dystonia may also occur in patients with IPD and thus expands the phenomenology of LID in PD.
Author Roles
1. Research Project: A. Conception, B. Organization, C. Execution; 2. Statistical Analysis: A. Design, B. Execution, C. Review and Critique; 3. Manuscript Preparation: A. Writing the First Draft, B. Review and Critique.
T.Y.T.: 1C, 3A, 3B
C.S.K.: 3A, 3B
N.M.I.: 1A, 1B, 1C, 3A, 3B
Disclosures
Ethical Compliance Statement: We conformed to the local ethical guidelines by obtaining verbal and written consent for educational publication as a case report/series. Written and verbal consent was obtained during clinic follow up, the patient has signed the consent form prepared by our hospital for video recording for publication and education purpose. We confirm that we have read the Journal's position on issues involved in ethical publication and affirm that this work is consistent with those guidelines.
Funding Sources and Conflict of Interest: No specific funding was received for this work. The authors declare that there are no conflicts of interest relevant to this work.
Financial Disclosures for the previous 12 months: The authors declare that there are no additional disclosures to report.
Supporting information
Supporting Video 1. This video was taken approximately 1 hour after his Madopar 62.5 mg (levodopa 50 mg/benserazide 12.5 mg). The patient had rhythmic wide jaw‐opening with facial grimacing and contraction of the platysmal muscles. This was associated with lingual contraction with side‐to‐side movement.
Supporting Video 2. This was captured 1 month after botulinum injection therapy. He had less rhythmic and reduced jaw opening movements. Facial grimacing was absent. Neck muscle contraction was remarkably reduced. Lingual movement was still observed, but less.
Relevant disclosures and conflicts of interest are listed at the end of this article.
References
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Associated Data
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Supplementary Materials
Supporting Video 1. This video was taken approximately 1 hour after his Madopar 62.5 mg (levodopa 50 mg/benserazide 12.5 mg). The patient had rhythmic wide jaw‐opening with facial grimacing and contraction of the platysmal muscles. This was associated with lingual contraction with side‐to‐side movement.
Supporting Video 2. This was captured 1 month after botulinum injection therapy. He had less rhythmic and reduced jaw opening movements. Facial grimacing was absent. Neck muscle contraction was remarkably reduced. Lingual movement was still observed, but less.