Brucellosis may cause various clinical conditions. However, to our knowledge, there is no case report on the sublingual abscess caused by Brucella in the medical literature. In this case report, we present a 45-year-old male patient who presented with acute shortness of breath. A sublingual abscess was identified, and brucellosis was diagnosed based on Brucella species. The patient achieved full recovery after the lesion was drained with a catheter, and he was treated with rifampicin and doxycycline for 6 weeks. In regions where brucellosis is endemic, this rare disorder should be considered.