Restless Jaw Syndrome: A Novel Possible Regional Anatomic Variant of Restless Leg Syndrome.
Sir, Restless leg syndrome (RLS) is a common neurological disorder characterized by an uncontrollable urge to move the legs, often accompanied by discomfort, especially during periods of rest or inactivity in the evening or at night.[1] RLS affects roughly 5–10% of the global population, with a female-to-male ratio of 2:1. While RLS traditionally involves the legs, it can also manifest in other regions, leading to diagnostic challenges. We present a rare case of restless jaw syndrome (RJS), a regional variant of RLS that is confined to the jaw. A 36-year-old male presented with a three-year history of intermittent burning sensations in the right jaw extending to the anterior neck, accompanied by a strong urge to move his jaw. Symptoms worsened during periods of rest but improved with activities like chewing, talking, or singing. To alleviate his discomfort, the patient developed a habit of chewing substances such as cardamom. A neurological examination was unremarkable, and an MRI of the brain revealed an incidental arachnoid cyst in the right temporal region without significant mass effect. An MRI of the face, including a trigeminal nerve protocol, was not performed. Comprehensive laboratory tests, including serum ferritin levels, were normal (serum ferritin: 75 μg/L). Trials of gabapentin (300 mg twice daily) and carbamazepine (200 mg twice daily) resulted in minimal improvement. Given the symptom pattern, RJS—a potential isolated variant of RLS was considered. The patient was started on low-dose pramipexole (0.125 mg twice daily), with a gradual increase to three times daily. Over three months, the patient reported a 70–80% reduction in symptoms, as assessed subjectively and using the patient global impression of change (PGIC) scale, without adverse effects. RLS is primarily diagnosed clinically, with secondary causes ruled out through targeted investigations. While it predominantly affects the legs, isolated regional variants involving other areas have been documented.[2] This case met the International RLS Study Group diagnostic criteria, and the significant improvement following dopaminergic therapy supports the diagnosis. Differential diagnoses such as trigeminal neuralgia and burning mouth syndrome were ruled out based on clinical features, absence of intraoral sensory disturbances, and poor response to non-dopaminergic treatments. The distinction from burning mouth syndrome is critical, as the latter primarily involves intraoral dysesthesia rather than an urge to move the jaw.[3] The limited improvement with gabapentin and carbamazepine suggests that the underlying pathophysiology of RJS may be more closely linked to dopaminergic dysfunction rather than neuropathic mechanisms. While dopaminergic therapy was effective in this case, long-term follow-up is needed to assess the durability of symptom relief and potential relapses. This report underscores the need for heightened awareness of atypical presentations of RLS, such as RJS. Dopaminergic dysfunction and brain iron dysregulation are key mechanisms in the pathophysiology of RLS, and these regional variants likely share similar underlying pathways.[4] Recognizing such variants can facilitate early intervention with dopaminergic agents, providing substantial symptom relief. We recommend further research into isolated RLS variants to improve diagnostic criteria and management strategies, ensuring timely and appropriate care for affected patients. 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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