Longitudinally extensive transverse myelitis presenting a relapse after oral contraceptive therapy

Abstract

A 25–year–old female suddenly developed paraparesis and bowel bladder dysfunction. Magnetic resonance imaging (MRI) at the onset revealed longitudinally extensive lesions (LETM) at the vertebral height of Th5–Th10, centrally located within the spinal cord. She was treated with intravenous methylprednisolone therapy, followed by oral prednisolone (PSL) of 40mg/day for 2 weeks. Her symptoms completely resolved, and did not show any relapses for 10 years in the absence of PSL. However, 6 months after oral contraceptive therapy was initiated at the age of 34, she developed a clinical relapse. The attack caused paraparesis, bowel bladder dysfunction, and sensory disturbances of the lower limbs. Lumbar MRI revealed centrally located swollen lesion at the vertebral height of Th10–Th11. Laboratory blood tests were negative for both anti–aquaporin–4, and anti–myelin oligodendrocyte glycoprotein antibodies. She was treated with intravenous methylprednisolone therapy, and her neurological symptoms completely resolved. She was started on oral PSL of 15mg/day. LETM is the core feature of neuromyelitis optica spectrum disorders (NMOSD). Despite the fact that for the fulfillment of NMOSD diagnostic criteria announced in 2015, 2 core symptoms are required, the centrally located lesions of the spinal cord is a typical characteristic suggesting NMOSD. Moreover, in contrast to the favorable effect of oral contraceptive therapy on multiple sclerosis, autoantibody–mediated disease such as systemic lupus erythematosus, is well known to be exacerbated. Our case is unique in that after 10 years of remission, she suddenly presented a clinical relapse after the induction of oral contraceptive therapy. This report highlights the possibility that oral contraceptive therapy may induce relapse of seronegative LETM.