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Background Moving-Toe-Syndrome (MTS) is a rare movement disorder characterized by involuntary toe movements. Vitamin B12 deficiency can lead to a variety of movement disorders. To our knowledge, this is the first reported case of painless MTS associated with B12 deficiency.Case history A 24-year-old Caucasian male with autism presented with a 7-month history of involuntary movements of the left second and third toes, initially considered functional. Unlike typical MTS, there was no pain. The movements persisted despite distraction and did not show entrainment. He had no history of trauma. He was a vegetarian. Neurological examination was unremarkable, and there was no neurophysiological evidence of neuropathy. The electroencephalogram did not show epileptiform activity during these movements. Laboratory tests revealed a significant vitamin B12 deficiency (B12 level: 112 pg/mL). Additionally, the patient had elevated methylmalonic acid (MMA) levels (2.4 µmol/L), elevated homocysteine levels (19.5 µmol/L), and low active vitamin B12 levels (150 pmol/L). Magnetic resonance imaging of the brain and spine was unremarkable.This was diagnosed as MTS potentially attributed to severe vitamin B12 deficiency. His symptoms resolved completely within 4 weeks of B12 replacement, and the active B12 level normalized.Conclusion This suggests that vitamin B12 deficiency may be a rare cause of MTS. The complete resolution following B12 replacement supports this hypothesis. Further studies are required to explore this association.sathyajith.ambawatte@gmail.com