Osteosarcomas of the mandible: multidisciplinary management of a rare tumor of the young adult a cooperative study of the GSF-GETO, Rare Cancer Network, GETTEC/REFCOR and SFCE.

BACKGROUND Mandibular osteosarcomas (MOS) mostly affect young adults. Their treatment is extrapolated from that of extragnathic osteosarcomas. MATERIAL AND METHODS A retrospective multicooperative group study was conducted to determine the impact of chemotherapy, adjuvant radiation therapy and surgery on outcomes and to identify prognostic factors. This ethical committee-approved study included a centralized review of histology slides and operative reports. RESULTS Of 111 patients, 58.6% were male, median age 35 years (13%, ≤18 years). Histology was osteoblastic, chondroblastic, fibroblastic, conventional not otherwise specified and others in 39.6%, 30.6%, 8.1%, 12.6% and 8.0%, respectively. Pathological World Health Organisation grades were low, intermediate and high grade in 6.4%, 11.8% and 81.8%, respectively. Surgery was carried out for 94.5% of patients. Neoadjuvant chemotherapy (mixed protocols) was carried out in 93.1% of patients. Postoperative chemotherapy and radiotherapy were carried out in 54.7% and 23.8%, respectively. Median follow-up was 59.6 months (range). Five-year local control, metastasis-free, disease-free and overall survival rates were 64.6%, 68.9%, 53.2% and 69.2%, respectively. Survival was significantly associated with age, tumor size and surgery. Wide surgery with clear margins and free flap reconstruction was the strongest prognostic factor. Neoadjuvant chemotherapy improved disease-free and metastatic-free survival and increased clear margins rates from 50% to 68%. Intermediate grades behaved like high grades in terms of metastatic-free and disease-free survival. CONCLUSION This homogeneous series is the largest to date and emphasizes the major impact of clear margins and multidisciplinary management. Neoadjuvant chemotherapy improves disease-free survival and should be recommended for both high and intermediate grade MOS.

[1]  R. Mirimanoff,et al.  [RCN, Rare Cancer Network]. , 2014, Bulletin du cancer.

[2]  K. Rankin,et al.  Chemotherapy, En Bloc Resection, and Prosthetic Bone Replacement in the Treatment of Osteogenic Sarcoma , 2014 .

[3]  A. Italiano,et al.  Osteosarcomas of the mandible: are they different from other tumor sites? , 2012, Critical reviews in oncology/hematology.

[4]  S. Chaudhary,et al.  Osteosarcoma of jaws , 2012, Journal of oral and maxillofacial pathology : JOMFP.

[5]  S. Salas,et al.  Primary osteosarcomas of the mandible: Joint study of the Groupe Sarcome Francais (GSF), Rare Cancer Network, and Groupe d'Etude des Tumeurs de la Tete et du Cou (GETTEC). , 2011, Journal of clinical oncology : official journal of the American Society of Clinical Oncology.

[6]  N. Kalavrezos,et al.  Sarcomas of the head and neck: a 10-year retrospective of 25 patients to evaluate treatment modalities, function and survival. , 2011, The British journal of oral & maxillofacial surgery.

[7]  R. Padilla,et al.  The Spectrum of Gnathic Osteosarcoma: Caveats for the Clinician and the Pathologist , 2011, Head and neck pathology.

[8]  G. Nielsen,et al.  Well-differentiated Osteosarcoma of the Jaw Bones: A Clinicopathologic Study of 15 Cases , 2010, The American journal of surgical pathology.

[9]  B. H. Abdullah,et al.  Proliferative, apoptotic and angiogenic potentials in jaws and long bones osteosarcomas: a comparative immunohistochemical study. , 2010, Journal of oral pathology & medicine : official publication of the International Association of Oral Pathologists and the American Academy of Oral Pathology.

[10]  A. Morrison,et al.  Mandibular reconstruction using nonvascularized autogenous bone grafting , 2010, Current opinion in otolaryngology & head and neck surgery.

[11]  Ajura Aj,et al.  A retrospective clinicopathological study of 59 osteogenic sarcoma of jaw bone archived in a stomatology unit. , 2010 .

[12]  L. Qin,et al.  Do radiation-associated soft tissue sarcomas have the same prognosis as sporadic soft tissue sarcomas? , 2010, Journal of clinical oncology : official journal of the American Society of Clinical Oncology.

[13]  Yulan He,et al.  Computed tomography findings in radiation-induced osteosarcoma of the jaws. , 2010, Oral surgery, oral medicine, oral pathology, oral radiology, and endodontics.

[14]  A. Ajura,et al.  A retrospective clinicopathological study of 59 osteogenic sarcoma of jaw bone archived in a stomatology unit. , 2010, The Malaysian journal of pathology.

[15]  R. Benjamin,et al.  Pediatric and adult osteosarcoma: comparisons and contrasts in presentation and therapy. , 2009, Cancer treatment and research.

[16]  E. Sturgis,et al.  Osteosarcoma of the jaw/craniofacial region , 2009, Cancer.

[17]  Hye-Rim Park,et al.  Expression of Ezrin and Metastatic Tumor Antigen in Osteosarcomas of the Jaw , 2009, Tumori.

[18]  P. Casali,et al.  Osteosarcoma: ESMO clinical recommendations for diagnosis, treatment and follow-up. , 2008, Annals of oncology : official journal of the European Society for Medical Oncology.

[19]  G. Ottaviani,et al.  The epidemiology of osteosarcoma. , 2009, Cancer treatment and research.

[20]  C. Brunelli,et al.  Decreased occurrence of osteonecrosis of the jaw after implementation of dental preventive measures in solid tumour patients with bone metastases treated with bisphosphonates. The experience of the National Cancer Institute of Milan. , 2009, Annals of oncology : official journal of the European Society for Medical Oncology.

[21]  K. Freier,et al.  Interdisciplinary combined treatment of craniofacial osteosarcoma with neoadjuvant and adjuvant chemotherapy and excision of the tumour: a retrospective study. , 2008, The British journal of oral & maxillofacial surgery.

[22]  J. L. L. Pendás,et al.  Radiation-induced sarcomas of the head and neck. , 2008 .

[23]  S. Pai,et al.  Osteosarcoma of the head and neck region: Lessons learned from a single‐institution experience of 50 patients , 2008, Head & neck.

[24]  M. Ballo,et al.  Excellent local control rates and distinctive patterns of failure in myxoid liposarcoma treated with conservation surgery and radiotherapy. , 2008, International journal of radiation oncology, biology, physics.

[25]  S. Bielack,et al.  Craniofacial osteosarcoma Experience of the cooperative German-Austrian-Swiss osteosarcoma study group. , 2008, Oral oncology.

[26]  N. Nikitakis,et al.  Osteogenic sarcoma of the jaw: a 10-year experience , 2007, BDJ.

[27]  S. Salas,et al.  Ezrin immunohistochemical expression in cartilaginous tumours: a useful tool for differential diagnosis between chondroblastic osteosarcoma and chondrosarcoma , 2008, Virchows Archiv.

[28]  M. van Glabbeke,et al.  Improvement in histologic response but not survival in osteosarcoma patients treated with intensified chemotherapy: a randomized phase III trial of the European Osteosarcoma Intergroup. , 2007, Journal of the National Cancer Institute.

[29]  G. Nielsen,et al.  Radiation therapy for control of soft-tissue sarcomas resected with positive margins. , 2005, International journal of radiation oncology, biology, physics.

[30]  W. Tilakaratne,et al.  Clinicopathological analysis of osteosarcoma of jaw bones. , 2007, Oral diseases.

[31]  Dafydd G. Thomas,et al.  Primary versus radiation‐associated craniofacial osteosarcoma , 2006, Cancer.

[32]  J. Aguilar-Ponce,et al.  Free osseous and soft tissue surgical margins as prognostic factors in mandibular osteosarcoma. , 2006, Oral oncology.

[33]  W. Adeyemo,et al.  Osteogenic sarcoma of the jaw bones: a single institution experience over a 21-year period. , 2006, Oral surgery, oral medicine, oral pathology, oral radiology, and endodontics.

[34]  T. Delaney,et al.  Radiotherapy for local control of osteosarcoma. , 2003, International journal of radiation oncology, biology, physics.

[35]  Osteogenic sarcoma of the mandible and maxilla: a Canadian review (1980-2000). , 2004, The Journal of otolaryngology.

[36]  L. Karnell,et al.  National Cancer Data Base report on osteosarcoma of the head and neck , 2003, Cancer.

[37]  P. Bradley,et al.  Management of sarcomas of the head and neck in adults. , 2003, Oral oncology.

[38]  J. Shah,et al.  Improved outcomes in patients with osteogenic sarcoma of the head and neck , 2002, Cancer.

[39]  M. Chindia,et al.  Osteosarcoma of the jaw bones. , 2001, Oral oncology.

[40]  Y. Talmi,et al.  Osteosarcoma of the jaw: The Chaim Sheba Medical Center experience , 2001 .

[41]  P. Speight,et al.  Osteosarcoma of the jaws: a 30-year retrospective review. , 2000, Oral surgery, oral medicine, oral pathology, oral radiology, and endodontics.

[42]  F. Frassica,et al.  Osteosarcoma of the head and neck: A review of the johns hopkins experience , 1999, The Laryngoscope.

[43]  G. Snow,et al.  OSTEOSARCOMA OF THE HEAD AND NECK: META‐ANALYSIS OF THE NONRANDOMIZED STUDIES , 1998, The Laryngoscope.

[44]  R. Souhami,et al.  Randomised trial of two regimens of chemotherapy in operable osteosarcoma: a study of the European Osteosarcoma Intergroup , 1997, The Lancet.

[45]  E. Conrad,et al.  Head and neck osteosarcoma at the University of Washington , 1997, Head & neck.

[46]  M. August,et al.  Osteogenic sarcoma of the jaws: factors influencing prognosis. , 1997, International journal of oral and maxillofacial surgery.

[47]  R. Koole,et al.  Osteosarcoma of the jaw bones. Long-term follow up of 48 cases. , 1997, International journal of oral and maxillofacial surgery.

[48]  G. Snow,et al.  Effect of chemotherapy on survival of craniofacial osteosarcoma: a systematic review of 201 patients. , 1997, Journal of clinical oncology : official journal of the American Society of Clinical Oncology.

[49]  P. V. van Diest,et al.  Radical surgical treatment in craniofacial osteosarcoma gives excellent survival. A retrospective cohort study of 14 patients. , 1994, European journal of cancer. Part B, Oral oncology.

[50]  F. Sim,et al.  Postradiation sarcoma of bone. , 1972, Orthopedics.

[51]  F. Bertoni,et al.  The istituto rizzoli‐beretta experience with osteosarcoma of the jaw , 1991, Cancer.

[52]  G. Gardner,et al.  Family cancer syndrome: A study of the kindred of a man with osteogenic sarcoma of the mandible , 1990, The Laryngoscope.

[53]  B. Colmenero,et al.  Osteogenic sarcoma of the maxilla and mandible. , 1986, Oral surgery, oral medicine, and oral pathology.

[54]  P. Slootweg,et al.  Osteosarcoma of the jaw bones. Analysis of 18 cases. , 1985, Journal of maxillofacial surgery.

[55]  W. Taylor,et al.  A controlled pilot study of high-dose methotrexate as postsurgical adjuvant treatment for primary osteosarcoma. , 1984, Journal of clinical oncology : official journal of the American Society of Clinical Oncology.

[56]  K. D. Devine,et al.  Osteosarcoma of the jaw , 1983, Cancer.

[57]  J. Smith,et al.  Radiation-induced sarcoma of bone: clinical and radiographic findings in 43 patients irradiated for soft tissue neoplasms. , 1982, Clinical radiology.

[58]  T. D. Briant,et al.  Osteogenic sarcoma of the mandible. , 1981, The Journal of otolaryngology.

[59]  S. Leibel,et al.  Treatment of osteogenic sarcoma of the mandible. , 1979, Archives of otolaryngology.

[60]  G. Rosen,et al.  Chemotherapy, en bloc resection, and prosthetic bone replacement in the treatment of osteogenic sarcoma , 1976, Cancer.

[61]  R. Marcove,et al.  Radiation‐induced sarcoma of bone , 1971, Cancer.

[62]  J. Cassady,et al.  Radiation induced neoplasia following external beam therapy for children with retinoblastoma. , 1969, The American journal of roentgenology, radium therapy, and nuclear medicine.

[63]  G. Garrington,et al.  Osteosarcoma of the jaws. Analysis of 56 cases , 1967, Cancer.

[64]  L. Triedman.  Osteogenic sarcoma of the mandible. , 1965, American journal of surgery.

[65]  R. G. Chambers,et al.  Osteogenic sarcoma of the mandible. A method of treatment. , 1960, American journal of surgery.

[66]  Митрополита Василя Липківського.  National , 1897, The Journal of comparative medicine and veterinary archives.