Lyme disease is the most commonly reported vector-borne illness in the United States, but it is relatively rare in Taiwan. Lyme disease can be treated with antibiotic agents, but approximately 20% of these patients experience persistent or intermittent subjective symptoms, so-called chronic Lyme disease (CLD). The mechanisms of CLD remain unclear and the symptoms related to CLD are difficult to manage. Hyperbaric oxygen therapy (HBOT) was applied in CLD therapy in the 1990s. However, reported information regarding the effectiveness of HBOT for CLD is still limited. Here, we present a patient with CLD who was successfully treated with HBOT.