BACKGROUND: Clinicians who perform radiation therapy (RT) are exposed to radiation, which may negatively affect their health. The present study reports a case of acute lymphoblastic leukemia in a healthcare provider who was exposed to radiation at work; we also present a literature review of this topic. CASE PRESENTATION: A 45-year-old patient, who had been a radiation oncologist and had been exposed to radiation while performing brachytherapy 10 years ago, complained of chest pain and was suspected of having leukemia based on the results of a blood test in an outpatient clinic. He was diagnosed with acute lymphoblastic leukemia, and subsequently underwent chemotherapy. However, the case died during treatment. Through epidemiological investigation, it was found that the case's cumulative exposure dose based on personal exposure and spatial dose measured during the work period was in the range of 6.08-12.15 mSv. CONCLUSIONS: Based on the following considerations, acute lymphoblastic leukemia was highly correlated with the level of radiation to which the case was exposed while performing brachytherapy on patients with cancer. Firstly, the latent period of acute lymphoblastic leukemia in the case closely matched the latency time reported in previous published studies (5-10 years). In addition, numerous studies have reported significantly higher relative risks of cancer among clinicians who perform RT compared with the general population. The case was also atypically exposed to radiation through his hands, despite wearing protective equipment. Lastly, the case's coworkers were also found to have been exposed to high levels of radiation. Investigation into the influence of radiation exposure through atypical routes during RT on the health of clinicians is recommended.