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To describe the use and effectiveness of a novel intensive progressive resistance exercise (PRE) approach to address the functional goals of a 14-year-old adolescent with a myelomeningocele.The child had lower extremity weakness, knee and hip flexion contractures, impaired somatosensation, and cardiopulmonary deconditioning, affecting gait mechanics and functional ambulation. An 8-week intensive PRE-based intervention was designed to improve walking in the home by targeting both power-generating and stabilizing lower extremity musculature. Secondary intervention focused on cardiopulmonary endurance training.The child demonstrated improvements in gait speed, walking endurance, and functional lower extremity strength. Knee contracture was moderately responsive to sustained stretching and positioning.This intensive PRE training approach had been effective for improving function among youth with cerebral palsy, and to our knowledge it had not yet been applied to youth with other neurological conditions.