Objective: We describe an unusual initial manifestation of Parkinson’s disease, consisting of oromandibular dystonia. We also discuss the role of DaTscan SPECT imaging in the diagnosis of uncommon presentations of Parkinson’s disease.

Background: Parkinson’s disease is a clinically heterogeneous disorder which typically features a combination of cardinal motor symptoms, classically rest tremor, bradykinesia, rigidity, and postural disturbances. However, patients can present with a broad range of symptoms that extend beyond these “classic” features.

Method: We describe 2 case reports seen recently at our institution. Neither patient had prior dopamine antagonist exposures.

Results: A 78 year old man presented for involuntary jaw opening, side-to-side movements of the tongue, and pharyngeal movements while speaking and swallowing for 9 years. He was previously treated with unsuccessful trials of trihexyphenidyl and tetrabenazine. His exam revealed subtle right hand rest tremor and bradykinesia. A DaTscan showed subtly decreased tracer uptake in the left putamen. We initiated him on levodopa, but he had no response to moderate-high dosages. Most recently, we treated him with botulinum toxin injections to the lateral pterygoids and genioglossus. A 71 year old woman presented for involuntary jaw opening and tongue movements for 6 years, which started after her lower teeth were extracted. The movements caused food to be expelled during chewing. Trials of botulinum toxin injection to the genioglossus, clonazepam, trihexyphenidyl, and amantadine were ineffective. Her symptoms slowly progressed and she also developed subtle right sided bradykinesia and rigidity. A DaTscan was performed, revealing decreased tracer uptake in the left putamen. She was started on low dose levodopa to which she has experienced a modest benefit.

Conclusion: Oromandibular dystonia is a rare form of focal dystonia, and can be either primary (idiopathic) or secondary, such as a tardive syndrome or resulting from certain dental cprocedures. However, Parkinson’s disease should be an important differential consideration, especially for an older patient with other asymmetric motor symptoms. In these clinical scenarios a DaTscan can be useful tool for differentiating parkinsonism from other etiologies, and is generally perceived as being clinically helpful for unusual or challenging clinical presentations.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/oromandibular-dystonia-as-an-initial-manifestation-of-parkinsons-disease-2-case-reports/