We report a case of transverse myelitis as a complication of acute cytomegalovirus (CMV) infection in immunocompetent patients; and review the literature on the entity. Primary CMV infection was documented by CMV antigenemia and high serum titers of CMV IgM and IgG antibodies. Cerebrospinal fluid (CSF) pleocytosis indicated central nervous system inflammation; CSF polymerase chain reaction (PCR) for CMV, however, was negative. The results of magnetic resonance imaging of the myelon were normal. Although CMV-associated transverse myelitis has been well described in HIV-positive individuals, but is very rare in immunocompetent individuals. It remains unclear whether the neuronal damage is immune mediated or due to a cytotoxic effect of viral infection. The outcome is mainly favorable.