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Painful Legs and Moving Toes syndrome (PLMT) is a rare neurological movement disorder characterized by involuntary and irregular movements of one or more toes, often together, with moderate to severe diffuse pain at the foot and leg. It can appear on one or both sides and in the upper limbs as well as lower limbs. The etiology of PLMT is still unclear; therefore, the main symptoms of PLMT, including pain in limbs as well as involuntary movements of toes, can be treated only symptomatically, and the outcomes are often unsatisfactory. Central or peripheral nerve injuries have been assumed to be the possible causes of irregular involuntary movements, often accompanied by chronic pain; however, its etiology is unknown in most cases. The management of PLMT is very difficult because of its rare prevalence and obscure pathogenesis, and there have been no clear guidelines for the treatment of PLMT. Oral medications (e.g., GABAergic agents, benzodiazepines, antiepileptics, antidepressants, atypical antipsychotics, beta-blockers, corticosteroids, cannabis, calcitonin, and analgesics) and several therapeutic interventions (e.g., local botulinum neurotoxin-A injection, spinal cord stimulation, anesthetic, and surgical interventions) have been recommended, based on individual experiences from several cases. Its etiology, pathogenesis, and effective treatments should be further discussed.
Painful Legs and Moving Toes syndrome (PLMT), one of the spinal-generated movement disorders [1], is a rare movement disorder with involuntary irregular flexion/extension of one or more toes, which is often accompanied with pain felt deeply in the foot and leg. It therefore reduces the quality of life as well as activities in social community and the workplace [2][3]. PLMT was firstly reported as a pain syndrome in the feet or lower limbs with spontaneous movements of the toes by Spillane and colleagues in 1971 [2]; since then, PLMT has been described in case or cases series reports rarely [4][5][6][7][8][9][10][11][12][13]. Until now, about 130 cases with PLMT have been reported, according to our literature search on PubMed and other public open-access databases (screening on 31 October 2021) and patients’ ages ranged from 11 to 86 [13][14][15]. The definition of PLMT has expanded to the upper limbs (painful hands and moving fingers) [7][8][11][12][16]. It has been also called “painful limbs and moving extremities” involving more than toes or fingers, e.g., feet or hands and limbs [8][12][17]. The affected locations have extended from toes or fingers proximally to ipsilateral foot or hands and limbs, as well as contralateral limbs in some cases, and possibly from the lower limb to the upper [16]. Several atypical cases with painful movement disorder have been reported [9][10][18][19][20][21]. One case with PLMT extended to the tongue, called “painful moving tongue”, but detailed information was not available [22].