Restless mouth syndrome

Abstract

60-year-old man was referred to our headache clinic for evaluation of atypical facial pain. He reported a 1-year history of oral discomfort that began 2 to 3 months after a fall with trauma to the posterior neck. He described an uncomfortable but nonpainful sensation of swelling and numbness at the roof of the mouth that gradually spread to the tongue, bilateral buccal surfaces, and eventually the whole mouth. This was particularly prominent when his mouth was in a relaxed open position and was associated with a strong urge to move his jaw. Movement of the jaw and tongue provided temporary relief. He chewed gum for hours at a time to alleviate the symptoms, although the relief was sometimes only partial or intermittent. Keeping the jaw completely closed or clenching the teeth also provided improvement in his symptoms. Thus, he used a headband, pillow, or hand to keep the jaw closed. His symptoms began midmorning and persisted throughout the day, but were worse in the evening. The symptoms did not interfere with sleep as long as he was able to keep his jaw closed with a pillow. He denied xerostomia or xerophthalmia. He had a history of basal cell carcinoma of the left cheek status post resection 2 to 3 years ago without residual numbness. He had no personal or family history of trigeminal neuralgia, headaches, neuropathy, or restless legs syndrome (RLS). He had no history of having been on neuroleptics, antiemetics, or other dopamine antagonists. Before seeing us, he tried abstaining from caffeine and spicy food, and tried cinnamon, increased hydration, and vitamin B 12 without benefit. Trials of gabapentin 200 to 300 mg 3 times a day and pregabalin 200 mg twice a day also had not provided improvement. Oxcarbazepine, at an unknown dose, was ineffective and associated with anxiety and nervousness. Physical examination, including general and neurologic examination, was normal. There was no dystonic posturing observed in the tongue at rest, during speaking, or with rapid alternating movements.

Motivation States for Physical Activity and Sedentary Behavior: Desire, Urge, Wanting, and Craving

“…Urges to move, most cited in work on Restless Legs Syndrome, are highly specific in this context to muscular movement but are clearly gauged as a negative tension. Patients with Restless Leg Syndrome, other variants such as Restless Arm or Mouth Syndrome, and Tourette's Syndrome report pressing, involuntary and bothersome urges to move and/or stretch that are often temporarily suppressed but eventually released (Cavanna and Nani, 2013;Jung et al, 2017;Ruppert, 2019). Appetence is highly specific to movement, but neutral in feeling in the same sense that appetite may be either present or lacking but distinct from the pangs of hunger (Ferreira et al, 2006).…”

Measurement of Motivation States for Physical Activity and Sedentary Behavior: Development and Validation of the CRAVE Scale

“…A preponderance of information about desires for movement exists in clinical and psychiatric studies. Urges to move, especially at night, are a defining characteristic of Restless Leg Syndrome, which may affect other parts of the body as well ( Garcia-Borreguero et al, 2011 ; Jung et al, 2017 ; Ruppert, 2019 ). Exercise dependence/addiction is a “craving for leisure-time physical activity, resulting in uncontrollable excessive exercise behavior” and showing a variety of physical and psychological symptoms ( Hausenblas and Downs, 2002 ).…”

Measurement of motivation states for physical activity and sedentary behavior: Development and validation of the CRAVE scale

“…Despite the lack of data in this area, there is a preponderance of information about desire for movement in other relevant literatures. Urges to move, especially at night, are a defining characteristic of Restless Leg Syndrome, which may affect other parts of the body as well (20,37,63). Urges to move the entire body, particularly for the purpose of energy expenditure are also a key symptom of exercise dependence (24).…”

“…A preponderance of information about desires for movement exists in clinical and psychiatric literatures. Urges to move, especially at night, are a defining characteristic of Restless Leg Syndrome, which may affect other parts of the body as well (31-33). Exercise dependence/addiction is a “craving for leisure-time physical activity, resulting in uncontrollable excessive exercise behavior” and showing a variety of physical and psychological symptoms (34).…”