Introduction: Gorham-Stout disease is a rare idiopathic condition of the bone that is characterized by a massive and spontaneous osteolysis, with a vascular or lymphatic proliferation in bone, which is then replaced by fibrous tissue. Observation: a 16-year-old patient was referred to the maxillofacial surgery department to remove ectopic teeth bilaterally in the ramus. He had Gorham-Stout disease, managed for many years in orthopedic surgery department for a lower limb lesion and in neurosurgery department for a breach of the meninges. He was treated for 4 years with bisphosphonates. The removal of the ectopic teeth went well, with a simple postoperative course. Discussion: Gorham-Stout disease physiopathology remains unknown. Facial bones are often involved, especially the mandible. There are many possible treatments, but, due to the rarity of the disease, no therapeutic consensus exists. Bisphosphonates seem to be a good way to control this condition. So far, no case of bisphosphonates related osteonecrosis of the jaw has been reported in children. Conclusion: Gorham-Stout disease can involve the mandible and may lead to ectopic teeth.

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Zygomatic implants have been used for several years for the treatment of extremely resorbed maxilla. Indications were extended for oral rehabilitations after maxillectomy in oncologic patients. A 24-year old patient with a triple A syndrome who underwent a left maxillectomy due to a spinocellular tumour was addressed for prosthetic rehabilitation. As his obturator prosthesis failed, surgical closure of the defect combined with 2 zygomatic implants to support the prosthesis was proposed. Despite a small persistent oro-antral fistula, the new obturator prosthesis restored the patient's functions and esthetics and improved his quality of life. The literature reports less than 40 cases of maxillectomy patients rehabilitated with zygomatic implants (with or without flap closure of the defect). Regardless of implant placement, there is no significant difference between reconstructive surgery and obturator prosthesis. Thus, zygomatic implants seem to be a reliable method for the stabilization of obturator prosthesis, without complex surgical procedure. Nonetheless, reservations should be expressed given the lack of data in terms of long-term follow up.

BackgroundOsteonecrosis of the jaw is a known complication of antiresorptive treatment, like bisphosphonate. More recently, denosumab was validated as a treatment in the osteoporosis and bone metastasis. Its mechanism is different from bisphosphonate but induces also a decrease of bone resorption and a risk of osteonecrosis of the jaw. In case of treatment failure by a dental surgeon or in complex cases, patients could be addressed to a bone and joint infection (BJI) reference center. The aim of this study was to analyze microbiology, as well as surgical and medical care of patients who present denosumab-related osteonecrosis of the jaw (DRONJ) and who were treated in a bone and join reference center.MethodsAll patients managed in our BJI reference center between January 2013 and December 2018 for a DRONJ were included in our retrospective observational monocentric cohort.ResultsTwelve patients (median age 71; ratio M/W 0.7) with a DRONJ (metastatic cancer, n = 10 (83%)) in grade 3 (n = 5), 2 (n = 4), 1 (n = 3) were included. Only 3 patients (25%) had a dental health control before initiating the treatment by denosumab and 7 patients (58%) had a dental surgical procedure done before the DRONJ. Eleven patients had a bone exposure, treated at least with a scaling and mucosal closure at the same time. All infections with bacterial cultures (n = 11 (91%)) were polymicrobial, including 8 (72%) with Streptococcus spp; 8 (72%) with anaerobia including 2 (18%) with Actinomyces; 5 (45%) with Staphylococcus spp; 5 (45%) with enterobacteria; 3 (27%) with Candida spp; 2 (17%) with a non-fermentative Gram-negative bacilli and 7 (64%) with others bacteria. All patients (n = 12) received a betalactam, 8 (66%) a lincosamide or a synergistin, 5 (41%) an antifungal, 5 (41%) metronidazole, 4 (33%) a fluoroquinolone, 3 (25%) a glycopeptide and 2 (17%) other antibiotics. The median follow-up was 6 months. Eight patients were cured after a medico-surgical care and a median duration of antibiotics of 97 days (including 28.5 days in intravenous). 2 patients required a suppressive antibiotic treatment, 1 relapsed at a distance of the treatment and 1 died from some other causes.ConclusionDRONJ is a potential complex BJI, for which some patients could benefit from medical care in a BJI reference center.Disclosures All authors: No reported disclosures.