Early neurological Lyme disease (neuroborreliosis) typically presents with well-recognised neurological syndromes. Spinal myoclonus is however a rare manifestation of neuroborreliosis. We present the case of a man who developed spinal myoclonus 3 weeks after returning from the Czech Republic where he developed erythema migrans on his arm following multiple tick exposures. Spinal fluid analysis showed a pleocytosis and MRI showed enhancement at C5-C6. His serology was positive for IgM antibodies to
Borrelia afzelli. He was successfully treated with ceftriaxone and doxycycline with improvement of his spinal myoclonus and radiculitis. We conclude that early Lyme neuroborreliosis may present with uncommon neurological manifestations and so a high degree of suspicion is needed.