Painful limbs/moving extremities (PLME) is a disorder characterized by spontaneous, complex, slow (1-2 Hz) involuntary toe or finger movements. The movements that can be bilateral or unilateral are usually accompanied by pain in the affected limbs. Painless variants are less common. PLME has been associated with peripheral and central nervous system disease although idiopathic cases have been reported. Its etiopathogenesis is unknown and treatment approaches remain largely empirical. Nerve blocks and botulinum toxin type A injections as well as oral medication have had some measure of success. Current theories suggest that central oscillator(s) at the spinal or supraspinal levels may be involved. Future research in PLME should include prospective electrophysiological and functional imaging studies as well as clinical trials with botulinum toxin injections and oral pharmacological agents.