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Maxillofacial diagnostic features of the enigmatic metastasizing pleomorphic adenoma
Institution:1. Department of Oral and Maxillofacial Surgery, Hebrew University–Hadassah, Jerusalem, Israel;2. Department of Pathology, Faculty of Medicine, Hebrew University–Hadassah, Jerusalem, Israel;3. Department of ENT, Faculty of Medicine, Hebrew University–Hadassah, Jerusalem, Israel;4. Arab American University–AAUP, Palestinian Authority;1. Center for TMD & Orofacial Pain, Peking University School & Hospital of Stomatology, Beijing, China;2. Department of Oral & Maxillofacial Radiology, Peking University School & Hospital of Stomatology, Beijing, China;3. National Clinical Research Center for Oral Diseases, Beijing, China;4. National Engineering Laboratory for Digital and Material Technology of Stomatology, Beijing, China;5. Beijing Key Laboratory of Digital Stomatology, Beijing, China;6. Department of Dentistry, Ng Teng Fong General Hospital, National University Health System, Singapore;7. Faculty of Dentistry, National University of Singapore, Singapore;8. National Dental Centre Singapore, Singapore;1. Department of Otolaryngology, Stanford University School of Medicine, Stanford, California, USA;2. Department of Oral and Maxillofacial Surgery (Dentistry), International St. Mary’s Hospital, Catholic Kwandong University, Incheon, South Korea;3. Center of Excellence in Otolaryngology Head and Neck Surgery, Rajavithi Hospital, College of Medicine, Rangsit University, Bangkok, Thailand;4. Department of Craniomaxillofacial and Oral Surgery, University Hospital, Medical University of Vienna, Vienna, Austria;1. Department of Oral and Maxillofacial Surgery, University of Groningen, University Medical Centre Groningen, Groningen, The Netherlands;2. Department of Implant Dentistry, University of Groningen, University Medical Centre Groningen, Dental School, Groningen, The Netherlands;1. Department of Clinical Oral Oncology, Unit of Translational Medicine, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan;2. Department of Oral and Maxillofacial Surgery, Division of Surgery, Tokai University School of Medicine, Isehara, Japan;3. Department of Oral and Maxillofacial Surgery, Kobe University Graduate School of Medicine, Kobe, Japan;4. First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, Osaka, Japan;5. Department of Dentistry and Oral Surgery, Shinshu University School of Medicine, Matsumoto, Japan;6. Department of Oral and Maxillofacial Surgery, School of Medicine, Nara Medical University, Kashihara, Japan;7. Department of Oral Surgical Oncology, Hokkaido Cancer Center, Sapporo, Japan;1. Craniofacial Center, Division of Oral and Maxillofacial Surgery, Seattle Children’s Hospital, Seattle, WA, USA;2. Craniofacial Center, Division of Plastic and Craniofacial Surgery, Seattle Children’s Hospital, Seattle, WA, USA;3. Craniofacial Center, Division of Craniofacial Orthodontics, Seattle Children’s Hospital, Seattle, WA, USA
Abstract:Considering the confusion in the literature regarding local recurrence, spread, or metastases of pleomorphic adenoma (PA) in the head and neck region, the aim of this study was to enhance understanding of the characteristics of metastasizing pleomorphic adenoma (MPA) by reviewing the literature and presenting a case. English language articles with proof of metastases were included in the literature review. Of the 80 cases in the literature with MPA, 46 were female and 33 were male (sex missing for one case). Thirty-five percent of the neoplasms affected the bones; the maxilla was affected in five cases and the mandible in three. The parotid was the primary site in 72.5% of cases and the submandibular gland in 16.2% of cases. The local recurrence rate was 70%. The mean interval between primary PA and MPA was 15.52 years. The total mortality rate was 8.7%. A case of PA of the submandibular gland that recurred after surgical excision and metastasized (confirmed by the presence of intact cortical borders) to the ipsilateral mandibular body, upper lip, and neck is described. The high mortality rate in a histologically defined benign disease that metastasizes demands that management include careful primary excision and long-term clinical follow-up.
Keywords:pleomorphic adenoma  dissemination  metastasizing pleomorphic adenoma
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