Alkaline-encrusted cystitis (AEC) has been reported sporadically during the past 90 years. It occurs mainly in immunocompromised or debilitated patients after urologic procedures. However, we report two unexpected cases of AEC in patients who were young and not under conditions of transplantation, immunocompromise or debilitation. Both had undergone a ureteroscopic lithotripsy procedure before being diagnosed as having encrusted cystitis. Although no information on such atypical patients is available, at least to our knowledge, we undertook a further report and discussion on the imaging characteristics of this atypical AEC.
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