Infected cephalohematoma associated with meningitis is rarely reported. We report the case of 19- day-old female newborn with a cephalohematoma infected by Escherichia coli, and whose cerebrospinal fluid showed pleocytosis. Antibiotics alone could not eradicate the infection of the cephalohematoma, and surgical incision and drainage resulted in obvious clinical improvement. Three weeks of antibiotic usage completed the course of treatment.