OBJECTIVE: To report a rare case of drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome caused by antituberculosis (TB) drugs, which progressed to acute generalized exanthematous pustulosis (AGEP) after moxifloxacin treatment. CASE SUMMARY: A 25-year-old female was hospitalized for dyspnea and dizziness. She had a history of TB and experienced rifampin-induced skin rash. She was treated for TB with moxifloxacin, isoniazid, ethambutol, and pyrazinamide. Upon admission, she had a fever of 39.2 degrees C, and aspartate aminotransferase (AST), alanine aminotransferase (ALT) levels, and eosinophil count increased during the first 10 days after admission. The patient had a rash all over the body with itching, pain, and a burning sensation. Diagnosis of DRESS syndrome was made. Immunoglobulin and prednisolone administration improved the DRESS symptoms. After the first DRESS syndrome diagnosis, anti-TB medications were changed to isoniazid, ethambutol, pyrazinamide, cycloserine, and streptomycin, which also caused a skin rash, itching, and elevated AST/ALT levels, and eosinophil count. Then, the anti-TB treatment was changed to cycloserine, streptomycin, ethionamide, and para-aminosalicylic acid. The rash and itching persisted and eosinophil count increased further. All TB medications were discontinued except streptomycin. Due to the flushing and a burning sensation by streptomycin at the injection site, it was replaced with moxifloxacin. The patient experienced erythematous pustules and patches on skin with desquamation, fissures, and swelling. Therefore, a diagnosis of moxifloxacin-induced AGEP was made. CONCLUSION: DRESS syndrome induced by anti-TB drugs developed in a 25-year-old woman with moxifloxacin-related subsequent AGEP..