Objective: To investigate the clinical characteristics of painful legs moving toes (PLMT) spectrum disorders.

Background: Classic PLMT is a rare movement disorder characterized by continuous involuntary movements of the toes, accompanied by pain in the affected limbs. Less common variants of PLMT can affect other extremities or mouth, or be painless. Current evidence is limited to case reports and single center case series, restricting broader analysis.

Method: We conducted a systematic review of all cases with PLMT or its variants reported in published literature up to January 2026. Clinical characteristics, neuroimaging and electrodiagnostic testing results, presumed etiologies, and treatment responses were obtained from the reports.

Results: A total of 126 publications with 330 cases were included. The majority of cases (65.7%) were female, and the mean age at onset was 51.3 years (SD 18.0, range 9–85). Painful variants (n = 286, 87.2%) were far more common than painless variants (n = 42, 12.7%). Classic PLMT was the most common variant (73.2%), followed by painless legs moving toes (14.2%), whereas all other variants were rare (n ≤ 10 each). Overall, peripheral nerve injury / radiculopathy was the most common identified etiology (n = 129, 39.0%), and the etiology was unclear in 131 cases (40.0%). The etiology was dependent on the phenotype, as painful variants were significantly more often associated with peripheral nerve injury / radiculopathy (42.9% vs. 14.3%, p<0.001) and less often with brain lesions (0.9% vs. 7.1%, p = 0.033) compared to painless variants. Accordingly, painful variants were significantly more often associated with abnormal electroneuromyography (47.1% vs 22.7%, p = 0.034) and less often with abnormal brain MRI (11.4% vs. 37.5%, p = 0.025). Of the most often used treatments, botulinum toxin injections seemed to provide the best response (22 treated cases, 59.0% positive response), followed by baclofen (n = 26, 40.0%), clonazepam (n = 60, 36.7%), and pregabalin (n = 37, 29.7%).

Conclusion: This systematic review represents the largest synthesis of cases with painful legs moving toes spectrum disorders to date. Although PLMT has generally been considered a peripheral movement disorder, we identified several cases with likely central nervous system pathology, especially among patients with painless variants.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/painful-legs-moving-toes-spectrum-disorders-a-systematic-review/