In view of the difficulties encountered in diagnosing neurosyphilis, we report an atypical case of neurosyphilis with unusual clinical presentation and MRI features.

Syphilis is an infectious disease caused by Treponema pallidum. The term of neurosyphilis refers to infection of central nervous system by this bacterium.

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A 57 year-old man, with a past history of risky sexual behavior, bilateral hearing loss and right eye redness since four months, was admitted in our department for an acute horizontal diplopia in the binocular vision since one month.  Neurological examination showed a palsy of the left sixth nerve. General examination showed a right scleritis and bilateral serum-mucous otitis with a right cervical lymphadenopathy. Brain MRI showed both bilateral supra and infra tentorial pachymeningitis more marked on the left side and bilateral lacrimal gland enlargement with mastoid filling and pansinusitis. Cerebrospinal fluid (CSF) examination showed elevated protein levels with no pleocytosis and normal glucose level. Results of laboratory and radiology screening tests including infectious, inflammatory and neoplastic testing showed positive serum TPHA test and reactive CSF VDRL. The diagnosis of neurosyphilis with ocular and otosyphilis was retained and treatment with penicillin G in collaboration with otolaryngologists and ophthalmologists was started.

Neurosyphilis diagnosis is relatively difficult because of the clinical polymorphism and differential diagnosis of pachymeningitis on brain imaging. The diagnosis is based on laboratory tests. The occurrence of ocular symptoms and otosyphilis is unusual and requires multidisciplinary management.