Late-Onset Oral Cenesthopathy With Dopaminergic Dysfunction: Therapeutic Response to Pramipexole in a Case With Suspected Prodromal Lewy Body Disease.

Abstract

An 85-year-old woman with late-onset depression subsequently developed persistent oral cenesthopathy. As antidepressant augmentation, low-dose aripiprazole improved both mood and oral symptoms, but oversedation and parkinsonism necessitated tapering and discontinuation. After discontinuation, oral cenesthopathy recurred without clear depressive worsening. Dopamine transporter single-photon emission computed tomography showed reduced bilateral striatal uptake and ¹²³I-metaiodobenzylguanidine cardiac scintigraphy showed decreased uptake, with preserved cognition. In light of the clinical and imaging findings suggestive of dopaminergic dysfunction, and after an informed discussion of off-label use, pramipexole 0.25 mg daily was initiated. Oral discomfort lessened within 1 week and oral intake normalised by 2 weeks, without adverse effects; remission of oral symptoms and mood persisted for 6 months. Overall, the presentation was conceptually compatible with the psychiatric-onset phenotype described in the prodromal Lewy body disease research framework, but this remains an interpretive consideration rather than a diagnosis. Assessing possible dopaminergic involvement in similar cases of oral cenesthopathy may aid individualised management; however, the therapeutic role of dopamine agonists requires confirmation in prospective studies.