Abstract
INTRODUCTION: Restless legs syndrome (RLS) is a common sensorimotor disorder that primarily affects the lower extremities. This condition is characterized by unpleasant sensations and an irresistible urge to move the affected body regions, typically during periods of rest or at night. While RLS most commonly involves the legs, atypical variants affecting other body parts, including the arms, abdomen, face, and even the head, have increasingly been reported. This has led to the emergence of a broader diagnostic concept termed "restless X syndrome". However, no prior cases involving the ocular region have been documented. CASE DESCRIPTION: Herein, we report a novel case of a 57-year-old woman who experienced chronic, nocturnal ocular pain for over 10 years, which repeatedly woke her from sleep. The pain, described as heavy and crushing, was not associated with any known ocular or neurologic pathology, and could be consistently alleviated by eye movements or eyelid stimulation. Multiple treatments for presumed dry eye syndrome and atypical facial pain were unsuccessful. After extensive evaluation, the condition was identified as a novel variant of RLS affecting the eyes. The patient showed no response to a dopamine agonist (pramipexole), but experienced marked and sustained relief with gabapentin enacarbil. CONCLUSION: This case highlights the potential for RLS to present in non-traditional locations, including the eyes. Clinicians should maintain a high index of suspicion for RLS variants in patients with nocturnal sensory discomfort that can be relieved by movement, even in the absence of limb symptoms. LEARNING POINTS: Restless legs syndrome (RLS) variants can affect body parts beyond the lower limbs, including the eyes.Symptoms are typically nocturnal and relieved by voluntary movement or sensory stimulation.Gabapentin enacarbil may be effective in treating these atypical RLS presentations.