SummaryA patient With neurosyphilis, presenting with severe ocular impairment due to optic neuropathy, is described, in such as case, a low index of clinical suspicion and improper use of syphilitic serologic tests may delay diagnosis. However, specific tests of serum and cerebrospinal fluid are mandatory for the diagnosis. Treatment evaluation necessitates the follow-up of serology and cerebrospinal fluid cell count. Recent changes of therapy recommendations in subjects with neurosyphilis and in those syphilitic patients co-Infected with HIV are Mentioned.