We read with interest the recent report by Jong and colleagues. 1 The authors highlighted a patient with primary lingual dystonia induced by speaking, with the problem relieved by something in the mouth and with anticholinergic treatment. Complete remission was noted 2 years later. The authors suggested that anticholinergic treatment may be the treatment of choice in such cases. We would like to highlight a patient with primary lingual dystonia, to draw attention to the clinical utility of sensory tricks, and the lack of effective pharmacologic therapy for this condition.
The patient is a 50-year-old woman, previously well with no significant medical or family history, presented with gradual difficulty in speaking over a period of a few months. Her tongue would curl up and occasionally pulled to one side of the mouth while speaking. At other times, her tongue would also protrude and press against her front teeth. She noticed that her tongue spasm and contortion only occurred during speaking but not with other tasks such as eating, drinking, whistling, and blowing. There was no history of exposure to neuroleptic medications, facial or oromandibular injuries, or recent infection.
Neurologic and mental state examinations were unremarkable, except for the presence of isolated tongue dystonia during speaking. Biochemical tests, including thyroid function, ceruloplasmin, and urea/electrolytes, were within normal limits. A magnetic resonance imaging scan of the brain was unremarkable. She was treated with anticholinergics, tetrabenazine, and benzodiazepines with little relief. Subsequently, the patient found that chewing bubble gum effectively relieved her symptoms. However, this sensory trick only lasted for a few months with gradual return of her symptoms. She then discovered that switching the nature of the food (such a sucking the seed of a fruit) in her mouth gave her renewed relief.
In contrast to the case by Jong and coworkers, 1 we highlight the observation that primary lingual dystonia may be intractable to pharmacologic therapy. As Jong and associates 1 pointed out, the etiology in the patient they described could be psychogenic in origin, possibly a contributory factor for the medical response and complete remission. Different sensory tricks as highlighted by our patient may be a useful adjunctive treatment. Our patient is considering botulinum toxin should her condition deteriorate. It would also be interesting to determine whether botulinum toxin, which works well for oromandibular dystonia, 2,3 is an effective treatment option for primary lingual dystonia.