Introduction:Fungal urinary tract infections (UTIs) are becoming increasingly common in hospitalized patients andCandidaspecies are the most prevalent organisms. However, recurrent candiduria in young healthy outpatients is rare thus require further examination to find the etiologic factors.Case presentation:We described a case of recurrent asymptomatic c caused by azole-resistantC. glabratain a healthy young female who only had previous use of antibiotics without other risk factors. However, after removal of the predisposing factor and the use of sensitive antifungal agents, the patient’s urine cultures remained positive. This phenomenon indicated to us that the patient might have an immune-related genetic deficiency. We found a novel caspase-associated recruitment domain-containing protein 9 (CARD9) gene mutation (c.808-11G > T) which might be the cause of recurrent asymptomatic candiduria in this immune-competent young female without any underlying diseases.Conclusions:We report a case of recurrent asymptomatic candiduria caused by azole-resistantCandida glabratain a young healthy female with a novelCARD9mutation. A functional study of this mutation should be performed in the future to determine its effect on asymptomatic fungal UTIs.