DISCUSSION: Lyme disease involves multisystems with dermatological, rheumatological, neurological and cardiac manifestations. It is caused by Borrelia burgdorferi and transmitted by Ixodes. Several neurological syndromes had been described including lymphatic meningitis, cranial neuropathy (commonly unilateral or bilateral Bell's palsy), painful radiculoneuritis, optic neuritis, mononeuritis multiplex, Guillain-Barre syndrome, encephalomyelitis, peripheral neuropathy and encephalopathy. Myelitis has recently been reported as an unusual complication of Lyme disease. Our case represents one of the few cases of this type. The unique feature in our case is that Lyme disease was first presented as myelitis.