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Background The need for whole mandibular bone reconstruction and bilateral joint replacement is fortunately rare, but it is an extremely challenging topic in maxillofacial surgery, due to its functional implications. CAD-CAM techniques development has opened new broad horizons in the surgical planning of complex maxillofacial reconstructions, in terms of accuracy, predictability, and functional cosmetic results. The review of the literature has revealed a small number of scientific reports on total mandibulectomy including the condyles, with only eleven cases from 1980. Most of the works describe reconstructions secondary to dysplastic or inflammatory diseases affecting the lower jaw. The aim of this work, reporting a rare case of massive fibrous dysplasia of the whole mandible, is to share our experience in the management of extended mandibular and bilateral joint reconstruction, using porous titanium patient-specific implants. Case presentation The authors present a 20-year-old male patient suffering from massive bone fibrous dysplasia of the mandible. The mandibular body and both the rami and the condylar processes had been involved, causing severe functional impairment, tooth loss, and facial deformation. The young patient, after repeated ineffective conservative surgical treatments, has required a biarticular mandibular replacement. Using virtual surgical planning (VSP) software, the authors, in collaboration with medical engineers, have created a custom-made original titanium porous mandibular implant, suspended from a bilateral artificial temporomandibular joint. The mandibular titanium implant body has been specifically designed to support soft tissues and to fix, in the alveolar region, a free fibular bone graft, for delayed dental implant prosthetic rehabilitation. Conclusion The surgical and technical details, as well as the new trends in mandibular reconstructions using porous titanium implants, are reported, and discussed, reviewing literature reports on this topic. Satisfactory functional and cosmetic restorative results have been obtained, and no major complications have occurred. The patient, currently in the 18th month clinical and radiological follow-up, has recently completed the functional restoration program by an implant-supported full-arch dental prosthesis.