Transverse myelitis is a rare and serious complication of systemic lupus erythematosus (SLE). A longitudinal involvement of the spinal cord with lupus-related transverse myelitis is more unusual. Only 7 cases have been reported. We describe a 53-year-old woman presenting with short-term paraplegia as an initial manifestation of SLE with longitudinal myelitis. She had a partial response to treatment with pulse cyclophosphamide and high-dose corticosteroids after follow-up more than 2 years. To the best of our knowledge, this is the first case report of "longitudinal" myelitis as an initial presentation of SLE. Magnetic resonance imaging typically shows increased signal intensity in T2-weighted images, cord swelling, and contrast enhancement over several spinal segments. The possibility of SLE should be kept in mind in women presenting with paraplegia with no apparent cause.