Despite the recent advances in microsurgical techniques, reconstruction of extensive skull base defects using free flaps in pediatric patients presents a surgical challenge, and reports on skull base reconstruction in infants is quite limited. We present a case of reconstruction of an extensive anterior skull base defect using a rectus abdominis (RA) myocutaneous flap in a 1 year-old (14 months) infant. Sufficient coverage of the intracranial contents, good aesthetic results, and minimal growth disturbance at the donor site were achieved by the muscle-sparing RA flap transfer. To the best of our knowledge, this was among the youngest case of skull base reconstruction using a free flap. The feasibility of free flap transfer and flap selection in pediatric skull base reconstruction is discussed. © 2012 Wiley Periodicals, Inc.