Lymphogranuloma venereum (LGV) is a sexually transmitted infection caused by an invasive form ofChlamydia trachomatis, typically affecting the anogenital area with regional lymphatic involvement. Extragenital manifestations are rare but can occur, particularly if the bacterium is inoculated out of the genital tract. To our knowledge, only 13 cases of symptomatic neck LGV have been published so far in the medical literature. We describe a case of a young man living with HIV with bilateral, rapid-growing and painful neck swelling, whose definite diagnosis was challenging. The patient underwent extensive investigation, including repeated ultrasound-guided fine-needle aspirations, microbiological exams and histological characterisation. Genotyping revealedC. trachomatisL2 serovar, confirming LGV as a final diagnosis, affecting neck lymph nodes. Treatment with doxycycline combined with surgical drainage led to clinical resolution. This case emphasises the importance of a comprehensive testing strategy to identify LGV, especially in individuals reporting unprotected oral sex, with unexplained neck lymphadenitis.