Purpose: To report a case of unilateral incipient syphilitic papillitis. Case summary: A 53-year-old female visited for a routine ocular examination. Best-corrected visual acuities were 20/20 in both eyes and there was no abnormality in the anterior segment, pupillary response, ocular motility examination, color vision test, and there were no inflammatory cells in the anterior chamber and vitreous cavity of both eyes. However, optic disc edema and corresponding retinal nerve fiber layer (RNFL) thickening in her left eye were observed in a fundoscopic examination and by optical coherence tomography. In orbital magnetic resonance imaging, there was no abnormal enhancement of the optic nerve or optic nerve sheath in both eyes. Serological tests were positive for fluorescent treponemal antibody absorbance (FTA-ABS) immunoglobulin M (IgM) and IgG, the treponema pallidum hemagglutination test, venereal disease research laboratory (VDRL) testing, and a rapid plasma reagin test, but there was no abnormal finding in cerebrospinal fluid analyses including the VDRL and FTA-ABS tests. With the diagnosis of unilateral incipient syphilitic papillitis in her left eye, she was treated with intravenous penicillin for 14 days and high dose systemic steroids for 3 days with complete resolution of the optic disc edema and RNFL thickening. Conclusions: Syphilis can present as a unilateral incipient papillitis. Therefore, in case with unilateral papillitis, serological test for syphilis, appropriate antibiotics and steroid treatment should be considered.
