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This paper describes the application of individual customized 3-dimensional (3D) printed macro-porous Ti6Al4 V shoulder prosthesis in the revision of total shoulder arthroplasty (TSA) for the patient with severe bone defects.A 47-year-old male had been under proximal humeral resection and TSA due to shoulder chondrosarcoma 6 years ago, but a second surgery to insert a new prosthesis was then performed because the prosthesis became loose 2 months ago leaving severe bone defects which conventional prosthesis was not suitable for revision of TSA. American Shoulder and Elbow Surgeons’ Form (ASES), Neer and Constant-Murley score were 36, 39, and 39, respectively.The patient was diagnosed with shoulder bone defects and restriction of the shoulder movement.3D printed shoulder model and computer-aided design (CAD) were used for prosthesis design and surgical simulation. The novel 3D printed titanium alloy shoulder prosthesis was customized subsequently to be used in the revision of TSA. The patient was followed up regularly after surgery. The ASES, Neer Shoulder score and Constant-Murley Shoulder score were evaluated during pre-operation, post-operation, and follow-up.Prosthesis was successfully implanted to complete anatomic reconstruction intraoperatively. The scores of ASES, Neer, and Constant-Murley were gradually increased after the operation. According to the X-ray, the bone healed satisfactorily without change of prosthetic position at 3rd and 12th months after the operation. The function of shoulder could meet the requirements of daily activities.The application of customized 3D printed titanium alloy shoulder prosthesis in the revision of TSA achieves satisfactory results. It provides a novel method for the similar revision surgery with severe bone defects.