Oropharyngeal lymphogranuloma venereum (LGV) is an uncommon presentation and mainly consists of either asymptomatic infections or enlarged cervical lymphadenopathy. According to cross-sectional studies, the prevalence of pharyngeal LGV among men who have sex with men (MSM) seems to be very low. Tissue tropism could explain the difference between rectal and oral mucosa infection frequencies. This paper reports the singular case of an oral ulcer caused by LGV in an MSM who had also an asymptomatic rectal infection. Given the increasing number of cases of LGV, unusual presentations such as oral ulceration may be seen more frequently.