ABSTRACT Introduction This article presents a unique case study of an individual with congenital limb loss and long-time (>56 years) body-powered prosthesis use, who was able to control a sophisticated robotic upper-limb prosthesis using surface electromyography signals and pattern recognition (PR) algorithms. This case demonstrates that individuals with congenital limb amputation are able to learn unique strategies to intuitively control a dexterous prosthetic limb. Materials and Methods After completing four training sessions using a virtual integration environment, a single subject participated in 12 in-laboratory clinical training sessions using the modular prosthetic limb (MPL)—a novel multiple–degree-of-freedom dexterous upper-limb prosthesis prototype. Baseline assessments were made with her body-powered prosthesis, as well as a two-site direct-control myoelectric Bebionic she had recently received. Functional assessments with the MPL were conducted during sessions 6 and 12. Outcome measures included timed box and blocks (BB) test, Assessment of Capacity for Myoelectric Control (ACMC), Jebsen-Taylor Hand Function Test (JTHFT), Trinity Amputation and Prosthesis Experience Scale, Upper Extremity Functional Scale (UEFS), and NASA Task Load Index. Results The subject was able to control two independent wrist degrees of freedom and up to three independent hand grasps of the MPL, using an array of surface electrodes. Improvements in the BB and ACMC were observed, although the total time to complete the JTHFT stayed relatively the same from weeks 6 to 12, using the MPL. While her enpoint perceived funcitonal ability with the MPL was 58% compared with 83% with her personal myoelectric prosthesis (12 hours of use vs 4–5 weeks of use as denoted on the UEFS); the subject reported short length of training, a long-term body-powered prosthetic user with congenital limb loss was able to demonstrate objective improvements in control of a dexterous prosthetic hand over a 12-week period of in-laboratory training, achieving intuitive independent control of a variety of simultaneous individual wrist motions and grasp patterns using PR. Conclusions This case demonstrates that even individuals with congenital amputation may be considered as candidates for upper-limb PR-controlled myoelectric prosthetic devices using surface electrodes.