Objective: Objectives: To report the use of spinal cord stimulation (SCS) in a patient with severe and refractory familial Restless Leg Syndrome (RLS).

Background: RLS is associated with decreased quality of life and can lead to significant sleep disruption[1]. Treatment of RLS can be challenging due to augmentation, and adverse effects of drugs, such as dopamine agonist, opioids, and alpha-2-delta ligands. There is a growing interest in the use of SCS for RLS [2-3].

Method: Case-report: The patient was a 65-year-old woman with longstanding history of RLS dating back to childhood. Her symptoms were described as discomfort affecting the lower limbs predominantly in the evening, with severe disruption of sleep. She had tried a combination of treatments including dopamine agonist (pramipexole, ropinirole, and rotigotine), levodopa, benzodiazepines, alpha-2-delta ligands (gabapentin and pregabalin), oral and intravenous iron supplementation, and opioids. Her current medication regimen for RLS is Clonazepam 0.75 mg, methadone 7.5 mg, and gabapentin enacarbil 600 mg in the evenings. She also uses a Relaxis device every night to alleviate her symptoms. Her past medical history is positive for gastroparesis (on treatment with domperidone), multiple back surgeries for scoliosis, including a T10-S1 spinal fusion, previous thyroid cancer, and thyroidectomy. She had a family history of RLS in her mother, maternal aunt, and identical twin sister. Her neurological exam is unremarkable and there is no evidence of neuropathy. Her last ferritin level was 115 ng/mL with normal iron studies and there has been no history of sleep apnea. Her restless leg rating scale score was 31 (severe). A spinal cord stimulator trial was performed.

Results: The patient noted a >50% improvement in her RLS symptoms. We proceeded with permanent implantation of a 10-kHz high-frequency spinal cord stimulator. Leads were placed at T4 level. Two weeks after implantation, her RLS rating scale score was 9. No medication changes were attempted at that point. Due to eventual lead erosion the device had to be explanted 1 month after the implantation. After explantation her RLS rating score returned to 33 (very severe).

Conclusion: Conclusions: Spinal cord stimulation may be an effective treatment option for medically refractory RLS. Prospective studies are necessary to define safety, efficacy, and long-term outcomes.

References: 1. Restless legs syndrome: diagnostic criteria, special considerations, and epidemiology. Sleep Med 4, 101–119 (2003). 2. De Vloo, P. et al. Successful spinal cord stimulation for severe medication-refractory restless legs syndrome. Mov. Disord. 4, 465–2 (2019). 3. Epidural Spinal Cord Stimulation: A Novel Therapy in the Treatment of Restless Legs Syndrome. 92, 582.e15–582.e18 (2016).

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