Syphilis is a spirochete disease that occurs by treponema pallidum. It can be sexually transmitted and can pass from mother to baby vertically. Neurological involvement is more common in late period. The use of common antibiotics can alter the natural course of disease. Because of this, syphilis can occur with different neurological findings. A 49-year-old male patient was admitted to our clinic with complaints of fluctuating double vision and low left eyelid for two months. His neurological examination; pupils middilate, bilateral direct and indirect pupillary light reflex were not available. In the left eye, there was movement limitation in all directions. In the right eye, there was movement restriction to the right. Also, bilateral ptosis was detected.
Brain magnetic resonance imaging (MRI) and electrophysiological studies including repetitive stimulation were normal. Syphilis was diagnosed with blood and cerebrospinal fluid (CSF) sample. For a long time, he was treated with high-dose intravenous penicillin. Ocular findings are an important indicator of neurosyphilis at central nervous system involvement. Ocular involvement due to primary effect of the disease is rare. If meningitis-related cerebrospinal fluid absorption defect develops, eye movement disorder and pupil abnormalities may occur. The diagnosis of neurosyphilis may be delayed due to different clinical manifestations. Neurosyphilis should also be considered in ocular findings.