Background：Immune checkpoint inhibitors (ICIs) have been proven to be beneficial in multiple advanced malignancies. However, the widespread use of ICIs also occurred with various immune-related adverse events (irAEs). However, while various immune-related adverse events related to immune checkpoint inhibitors have been reported, there are few reports of lower urinary tract symptoms. Case presentation：A 42-year-old woman with lung cancer who was being treated with sintilimab, anlotinib, and denosumab presented to the nephrology department with frequent micturition, urgency, odynuria and gross hematuria. Initial laboratory tests did not reveal bacteria, but CT examination suggested cystitis. After empiric antiinfective therapy, the above symptoms did not improve significantly. The patient stopped taking sinlizumab and completed cystoscopy. Based on the combined clinical manifestations and laboratory findings, he was diagnosed with immune-related cystitis. Symptomatic relief was achieved via steroid treatment. Thereafter, the patient has been followed for 3 months without any symptoms or recurrence of immune-related cystitis Conclusions: immune-related cystitis is a commonly misdiagnosed disease. However, early diagnosis, treatment and prophylaxis through accumulated clinical data can help patients achieve a good prognosis. Therefore, clinicians need to be well aware of the variety of clinical characteristics and treatment options of this disease.

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