Appendicitis is a common cause of acute abdomen in pediatrics. Periappendiceal abscesses are frequently found in the pediatric population. Acute appendicitis in children can, at times, be a difficult clinical diagnosis because of its highly variable history? and physical manifestations and its unpredictable course. Despite the uncertainty of the diagnosis, appendicitis demands prompt treatment because of the risk of perforation, which occurs in approximately one third of cases. Urological manifestations of appendicitis and appendiceal abscess can vary. Acute appendicitis presenting with ureteral stenosis and hydronephrosis is very rare. Here, we report a case of acute appendicitis with perforation and left hydronephrosis in a 3-year-old female. This case presents a 3-year-old girl with dysuria having hydronephrosis that originated from a perforated appendix.