Thumb hypoplasia and radial dysplasia are two of the most common hand therapy requiring conditions among the congenital problems. However, any cases with congenital agenesis of bilateral thumb extensors, and their related rehabilitatitive therapy were not reported in the literature. The aim of this case report is to make the first contribution to the restoration of thumb extension function in a child with congenital absence of bilateral extensor pollicis longus (EPL) and extensor pollicis brevis (EPB) The case was a 5-year old girl and her family noticed that she had no thumb extension. Results of the tests performed to eliminate the other systemic problems were all in normal ranges. The patient underwent immobilization in a forearm cast with 20° wrist extension and full thumb extension and abduction for 6 weeks. During this period, early dynamic motion protocol was applied by a wire-rubber system splint. After removal of the splint, therapeutic ultrasound was applied and passive and active-assisted exercises gradually proceeded to resistive exercises through the 8 th week. By the 8 th week, occupational therapy with gradually increasing resistance was initiated while the patient was permitted to use both hands in daily activities after 4 th month. Assessments on the postoperative 6 th , 8 th and 12 th weeks revealed significant improvements in range of motion in the thumbs, grip and pinching strengths and hand functional level. Restoration of thumb extension with surgery and hand rehabilitation before school age provided optimal hand function and quality of life of this child.