Bone fibrous dysplasia (BFD) in most cases demonstrates to be a self-limiting disease during patient aging but, progressive growth into the third and fourth decades of life, has been reported. Malignant degeneration has been reported in 0.4% of cases [
14]. Jawbones BFD may cause either facial deformity or severe malocclusion, alveolar bone deformation, tooth loss or migration, bone pain, and severe TMJ functional impairment [
24]. Treatment of BFD in adults remains challenging, and long-term success is inherently unpredictable. Medical treatments using bisphosphonates, or other bone antiresorptive drugs, are proven to reduce pain, but seem to be ineffective on dysplastic bone growth. Immune target therapies are still off-label, experimental but promising treatments [
25]. If conservative or minor surgical procedures are not successful, a major surgical approach with extended bone resection/reconstruction becomes mandatory. The case reported in this paper demonstrates, in our opinion, that satisfactory rehabilitation can be achieved even after a bicondylar mandibulectomy using a free bone flap combined to patient-specific mandibular and articular titanium implants. Reviewing the literature, from 1980 to date, we have found only eleven reported cases of bicondylar mandibular replacement (Tables
1 and
2) [
12‐
23]. One of them, reported by Jeremic in 2010, was a bicondylar-sparing mandibular resection, so the bone flaps were attached directly to the native joints by plates and screws [
15]. In two cases, prosthetic joints have been used to connect the bone flaps to the skull, reporting good functional results, one by Paley in 2005 with a Christensen joint [
14], the other by Rustemeyer in 2019 with a Biomet joint [
21]. In the remaining seven cases, the new mandible has been resuspended. In three cases only, the disk has been preserved to allow close resuspension of the bone flap extremity to the glenoid fossa. The most used bone flap was the FFF (six cases), while just in one case a bilateral iliac crest flap was performed. Motlagh described, in 2017, the use of a freeze-dried human allogenic mandible as a transplantation [
12], while Poukens, in 2012, has replaced the whole mandible by a total prosthetic implant and no bone has been used [
18]. The report by Pouken, available on the official website of Xilloc ™, a biomedical implant manufacturer, has been described as the “very first world’s 3D-printed total jaw reconstruction,” but to our knowledge, has never been scientifically published on a peer-reviewed medical journal. Recently Chernohorskyi [
22], has published a clinical report on the reconstruction of a young female patient mandible using the titanium custom-made implant described by Poukens. All the Authors, anyway, reported good functional and stable outcomes, without major complications, considering a medium follow-up of 28 months, ranging from a minimum of 7 months to a maximum of 5 years. In all the cases of this review, the alveolar branch of the mandibular nerve has been resected bilaterally, and none have reported nerve reconstruction. In most cases, the disease affecting the mandible was osteomyelitis or low-grade dysplastic degeneration. Making a comparison to the reports of this literature review, the case we are presenting reveals some original aspects that, in our opinion, must be outlined and discussed. Our reconstruction project is based on a new original concept of creating a hybrid (bone/metallic) mandible. The basal mandibular bone and the rami have been replaced by a microporous surface and a macro-porous body titanium implant, designed to be integrated by connective soft and hard tissues, and to reattach muscular insertions, reducing at the same time the plate exposure risk. The mandibular body implant has been designed and shaped to support a screwed-free fibula flap to recreate the alveolar bone process of the mandible, as recently suggested by some surgeons [
26‐
28]. The VSP procedure planned the ideal bone position for a delayed dental implant insertion. CAD-CAM techniques have revealed to be very effective, predictable and surgically timesaving in the treatment of this complex case. Even if we did not insert dental implants during the primary reconstruction, the bone position has allowed a satisfactory dental restoration. Porous titanium bone implants or porous patient-specific plates appear to produce better results instead of commercial plates, in term of healing times, tissue integration, and plate exposure rate [
29,
30]. The implanted mandibular body has been secured to its prosthetic condylar heads by a Morse’s cone attachment, either to facilitate intraoperative fitting to the glenoid prosthetic component or to allow future joint prosthetic replacement. The introduction of CAD/CAM technology for the design and manufacturing of patient-specific mandibular reconstruction plates, in combination with cutting and drill guide printing, has created a broad range of new opportunities for the planning and implementation of mandibular reconstruction. This concept can be applied not only to the reconstruction with bone grafts or bone flaps but also to stand-alone alloplastic reconstructions [
22]. The need for condylar reconstruction while performing a mandibular replacement is a high debated topic in literature [
31], due to postoperative pain and dysfunctional outcomes. One of the most used techniques reported in this literature review, is the articular disk sparing to prevent glenoid fossa resorption or bone surface anchylosis, sassociated with the shaping and the resuspension of the bone flap extremity, used as a new condyle (Table
2). In other case reports the articular prosthesis has been designed to be the replica of the condylar head. In the case we are reporting, the left condyle has been removed 5 years before during a previous surgical debulking, the right condyle was deformed, and the articular space was so thin revealing that the disk was malacic and could not be preserved. We decided that both condyles needed to be replaced by a bilateral TMJ patient-specific prosthesis, to obtain a good mandibular function and stability. We choose a round-shaped condyle. The patient has undergone radical surgery in 2021, and no major intra- or post-operative complications occurred, presenting good functional and aesthetic outcomes. On the 12
th-month follow-up, the patient has undergone dental implant restoration revealing an optimal preservation and viability of the fibular bone flap, even if the free bone flap has no contact with other bones. To date, at the 18
th-month follow-up, the occlusion has been restored by a dental implant-supported full arch prosthesis. The review of the literature on real total mandibular reconstructions demonstrates that the need for a total replacement of the mandible is fortunately a rare condition, mainly caused by osteomyelitis or by low-grade malignancies, but it's still a challenging topic in maxillofacial surgery for its functional and structural implications. The technological development in the field of maxillo-mandibular reconstruction, VSP and CAD-CAM techniques evolution, to date, justify a more extensive reconstructive approach, even towards benign diseases to achieve better cosmesis, improved function, and to reduce the disease recurrence rate.