The reports on Inverted Follicular Keratosis (IFK) onset since childhood were rare. Here, we reported 15-y-old male patient with a facial papule occurred since childhood. He was diagnosed as wart and accepted an excision 10 months before this treatment, but the histopathology examination has been ignored. The uniform lesion was recurrent soon. After fusiform excision was performed for the recurred lesion, a histopathology examination showed the feature of IFK. No recurrence has been observed during the follow-up visits for 1 y for this patient after surgical resection. The reports on IFK onset since childhood were rare. This case experience strongly suggests that histopathology examination should not be ignored after the excision of the skin lesion, even though the disease is easy to be acknowledged by clinical diagnosis.