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Zhao Z, Xiong G, Wang C, Cao W. From pathogenesis to precision medicine: Transformative advances in research and treatment of ameloblastoma. Cancer Lett 2025; 612:217448. [PMID: 39800213 DOI: 10.1016/j.canlet.2025.217448] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/09/2024] [Revised: 01/02/2025] [Accepted: 01/04/2025] [Indexed: 01/15/2025]
Abstract
Odontogenic neoplasms of the jaw are dominated by ameloblastoma (AM), a locally aggressive epithelial tumor with a significant propensity for recurrence. The World Health Organization's 2022 update to the AM classification system underscores recent progress in comprehending its underlying mechanisms and refining clinical approaches. Contemporary research has yielded significant insights into the genetic underpinnings of AM, paving the way for the development of precision-based treatment strategies. Advanced genetic profiling has revealed a significant frequency of BRAF (V-raf murine sarcoma viral oncogene homolog) V600E and SMO (Smoothened) gene alterations in AM. Importantly, therapeutic interventions specifically designed to target these genetic aberrations, including BRAF and MEK pathway blockers, have shown encouraging results in terms of both effectiveness and tolerability, as documented in individual case reports and small-scale clinical investigations. This comprehensive review summarizes the recent modifications to the World Health Organization's categorization of AMs, explores progress in elucidating their underlying molecular pathways, and evaluates emerging targeted treatment modalities. Our objective is to present a thorough synthesis of contemporary scientific discoveries and therapeutic interventions, potentially paving the way for more efficacious and individualized clinical management protocols for this complex neoplasm.
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Affiliation(s)
- Zhang Zhao
- Department of Oral and Maxillofacial & Head and Neck Oncology, Shanghai Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China; National Center for Stomatology, National Clinical Research Center for Oral Diseases, Shanghai Key Laboratory of Stomatology, Shanghai, China
| | - Gan Xiong
- Hospital of Stomatology, Sun Yat-sen University, Guangzhou, China; Guangdong Provincial Key Laboratory of Stomatology, Sun Yat-sen University, Guangzhou, China; Guanghua School of Stomatology, Sun Yat-sen University, Guangzhou, China
| | - Cheng Wang
- Hospital of Stomatology, Sun Yat-sen University, Guangzhou, China; Guangdong Provincial Key Laboratory of Stomatology, Sun Yat-sen University, Guangzhou, China; Guanghua School of Stomatology, Sun Yat-sen University, Guangzhou, China.
| | - Wei Cao
- Department of Oral and Maxillofacial & Head and Neck Oncology, Shanghai Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China; National Center for Stomatology, National Clinical Research Center for Oral Diseases, Shanghai Key Laboratory of Stomatology, Shanghai, China.
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Marxgut L, Fourneret P, Waissi W. Exploring treatment complexity in maxillary ameloblastoma: A case study on the efficacy of radiotherapy with transcriptomic and literature insights. Cancer Radiother 2025; 29:104591. [PMID: 40043527 DOI: 10.1016/j.canrad.2025.104591] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/18/2024] [Revised: 09/22/2024] [Accepted: 09/24/2024] [Indexed: 04/01/2025]
Abstract
In this report, we describe the case of a patient initially experiencing symptoms including nasal obstruction and rhinorrhoea, leading to surgeries and a diagnosis of ameloblastoma. Despite complete surgical resections, MRI follow-ups indicated tumour regrowth, prompting a shift to radiotherapy. The treatment plan involved a high-dose (60Gy in 30 fractions of 2Gy) volumetric modulated arc therapy. During treatment, the patient experienced minimal side effects and showed significant clinical improvement, including tumour size reduction and resolution of facial paralysis. Follow-up MRI confirmed shrinkage of the tumour, with some persistent symptoms like diplopia. To better understand the radiosensitivity of ameloblastomas, transcriptomic analysis highlighted the potential of a radiosensitivity index to predict treatment response, indicating ameloblastomas exhibit higher radiosensitivity compared to odontogenic keratocysts. Finally, literature analysis showed a small number of cases with wide range of radiation dose. In conclusion, the case illustrates the complex diagnostic journey of ameloblastomas and underscores the efficacy of radiotherapy for inoperable cases, and the importance of predictive biomarkers to enhance treatment outcomes.
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Affiliation(s)
- Léa Marxgut
- Département de radiothérapie, hôpital universitaire Grenoble Alpes, 38000 Grenoble, France.
| | - Philippe Fourneret
- Département de radiothérapie, hôpital de Chambéry, 73000 Chambéry, France
| | - Waisse Waissi
- Département de radiothérapie, centre Léon-Bérard, 69008 Lyon, France; Équipe Génétique, épigénétique et biologie des sarcomes, Centre de recherche en cancérologie de Lyon, Inserm U1052 - CNRS UMR5286, centre Léon-Bérard, université Claude-Bernard Lyon-1, 69008 Lyon, France
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Lynch MM, Hermida-Viveiros P, Stencel S, Knott H, Al-Maryati R, Obeidin F, Alexiev BA, Abbinanti S, Damodaran S, Agulnik M, Pollack SM. Durable disease regression with copanlisib treatment in PI3K-mutated metastasizing ameloblastoma: A case report. Rare Tumors 2025; 17:20363613241309961. [PMID: 39790867 PMCID: PMC11707780 DOI: 10.1177/20363613241309961] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/24/2024] [Revised: 12/02/2024] [Accepted: 12/10/2024] [Indexed: 01/12/2025] Open
Abstract
Ameloblastoma is a rare tumor arising from odontogenic cells that is benign, yet locally aggressive. Metastasizing ameloblastoma (METAM) is an ultra-rare ameloblastoma variant in which both primary and secondary tumors have histological features of benign ameloblastoma. This is a case report of a patient who presented with a jaw mass and subsequent lung metastases, later diagnosed as METAM. Initial treatments, including carboplatin, etoposide, and taxane-based chemotherapy, were ineffective. Molecular profiling revealed mutations including PIK3CA H1047R and BRAF V600E. The patient was enrolled in a tumor-agnostic trial and began treatment with copanlisib, a PI3K inhibitor, which resulted in a partial response and durable disease regression. After 76 cycles, she continues to tolerate therapy well with minimal adverse events. This case highlights the potential of targeted therapies such as copanlisib for treating METAM, providing a promising therapeutic option for patients with PIK3CA mutations.
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Affiliation(s)
- Meghan M Lynch
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
| | - Pedro Hermida-Viveiros
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
| | - Sean Stencel
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
| | - Hannah Knott
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
| | - Rusul Al-Maryati
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
| | - Farres Obeidin
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
| | - Borislav A Alexiev
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
| | - Susan Abbinanti
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
| | | | - Mark Agulnik
- Department of Medicine, City of Hope Cancer Center, Duarte, USA
| | - Seth M Pollack
- Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago, USA
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Raemy A, May L, Sala N, Diezi M, Beck-Popovic M, Broome M. Anti-MAPK Targeted Therapy for Ameloblastoma: Case Report with a Systematic Review. Cancers (Basel) 2024; 16:2174. [PMID: 38927880 PMCID: PMC11201667 DOI: 10.3390/cancers16122174] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/09/2024] [Revised: 05/30/2024] [Accepted: 06/06/2024] [Indexed: 06/28/2024] Open
Abstract
Ameloblastoma, a benign yet aggressive odontogenic tumor known for its recurrence and the severe morbidity from radical surgeries, may benefit from advancements in targeted therapy. We present a case of a 15-year-old girl with ameloblastoma successfully treated with targeted therapy and review the literature with this question: Is anti-MAPK targeted therapy safe and effective for treating ameloblastoma? This systematic review was registered in PROSPERO, adhered to PRISMA guidelines, and searched multiple databases up to December 2023, identifying 13 relevant studies out of 647 records, covering 23 patients treated with MAPK inhibitor therapies. The results were promising as nearly all patients showed a positive treatment response, with four achieving complete radiological remission and others showing substantial reductions in primary, recurrent, and metastatic ameloblastoma sizes. Side effects were mostly mild to moderate. This study presents anti-MAPK therapy as a significant shift from invasive surgical treatments, potentially enhancing life quality and clinical outcomes by offering a less invasive yet effective treatment alternative. This approach could signify a breakthrough in managing this challenging tumor, emphasizing the need for further research into molecular-targeted therapies.
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Affiliation(s)
- Anton Raemy
- Department of Maxillofacial Surgery, Lausanne University Hospital, 1011 Lausanne, Switzerland; (L.M.); (M.B.)
| | - Laurence May
- Department of Maxillofacial Surgery, Lausanne University Hospital, 1011 Lausanne, Switzerland; (L.M.); (M.B.)
| | - Nathalie Sala
- Institute of Pathology, Lausanne University Hospital, 1011 Lausanne, Switzerland;
| | - Manuel Diezi
- Department of Paediatric Oncology, Lausanne University Hospital, 1011 Lausanne, Switzerland; (M.D.); (M.B.-P.)
| | - Maja Beck-Popovic
- Department of Paediatric Oncology, Lausanne University Hospital, 1011 Lausanne, Switzerland; (M.D.); (M.B.-P.)
| | - Martin Broome
- Department of Maxillofacial Surgery, Lausanne University Hospital, 1011 Lausanne, Switzerland; (L.M.); (M.B.)
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Chujan S, Vajeethaveesin N, Satayavivad J, Kitkumthorn N. Identification of Molecular Mechanisms of Ameloblastoma and Drug Repositioning by Integration of Bioinformatics Analysis and Molecular Docking Simulation. Bioinform Biol Insights 2024; 18:11779322241256459. [PMID: 38812739 PMCID: PMC11135093 DOI: 10.1177/11779322241256459] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/29/2023] [Accepted: 05/02/2024] [Indexed: 05/31/2024] Open
Abstract
Background Ameloblastoma (AM) is a benign tumor locally originated from odontogenic epithelium that is commonly found in the jaw. This tumor makes aggressive invasions and has a high recurrence rate. This study aimed to investigate the differentially expressed genes (DEGs), biological function alterations, disease targets, and existing drugs for AM using bioinformatics analysis. Methods The data set of AM was retrieved from the GEO database (GSE132474) and identified the DEGs using bioinformatics analysis. The biological alteration analysis was applied to Gene Ontology (GO) and Kyoto Encyclopedia of Genes and Genomes (KEGG) pathways. Protein-protein interaction (PPI) network analysis and hub gene identification were screened through NetworkAnalyst. The transcription factor-protein network was constructed via OmicsNet. We also identified candidate compounds from L1000CDS2 database. The target of AM and candidate compounds were verified using docking simulation. Results Totally, 611 DEGs were identified. The biological function enrichment analysis revealed glycosaminoglycan and GABA (γ-aminobutyric acid) signaling were most significantly up-regulated and down-regulated in AM, respectively. Subsequently, hub genes and transcription factors were screened via the network and showed FOS protein was found in both networks. Furthermore, we evaluated FOS protein to be a therapeutic target in AMs. Candidate compounds were screened and verified using docking simulation. Tanespimycin showed the greatest affinity binding value to bind FOS protein. Conclusions This study presented the underlying molecular mechanisms of disease pathogenesis, biological alteration, and important pathways of AMs and provided a candidate compound, Tanespimycin, targeting FOS protein for the treatment of AMs.
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Affiliation(s)
- Suthipong Chujan
- Laboratory of Pharmacology, Chulabhorn Research Institute, Bangkok, Thailand
- Center of Excellence on Environmental Health and Toxicology (EHT), Office of the Permanent Secretary (OPS), Ministry of Higher Education, Science, Research and Innovation (MHESI), Bangkok, Thailand
| | | | - Jutamaad Satayavivad
- Laboratory of Pharmacology, Chulabhorn Research Institute, Bangkok, Thailand
- Center of Excellence on Environmental Health and Toxicology (EHT), Office of the Permanent Secretary (OPS), Ministry of Higher Education, Science, Research and Innovation (MHESI), Bangkok, Thailand
| | - Nakarin Kitkumthorn
- Department of Oral Biology, Faculty of Dentistry, Mahidol University, Bangkok, Thailand
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Gates JC, Clark AP, Cherkas E, Shreenivas AV, Kraus D, Danzinger N, Huang RSP, Johnson J, Ross JS. Genomic profiling and precision medicine in complex ameloblastoma. Head Neck 2023; 45:816-826. [PMID: 36645099 DOI: 10.1002/hed.27294] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/14/2022] [Revised: 12/01/2022] [Accepted: 12/27/2022] [Indexed: 01/17/2023] Open
Abstract
BACKGROUND Ameloblastoma may present a significant treatment challenge in the locally advanced, recurrent and metastatic setting. Comprehensive genomic profiling (CGP) can identify targetable genomic alterations to aid in treatment. METHODS Ameloblastoma samples were sequenced using hybrid-capture based sequencing. A systematic literature review was performed to examine outcomes in studies employing targeted treatment in ameloblastoma. RESULTS We reviewed 14 cases of Ameloblastoma using CGP. There were six patients with activating BRAF mutations, five with PIK3CA, five with SMO, four with FGFR2, one with EGFR, and one with ROS1. All cases were MSI stable and the median TMB was 2.5 mutations/Mb. A separate literature review of clinical outcomes in ameloblastoma showed a predominance of at least partial response to targeted treatment (7/12 cases). CONCLUSION CGP is helpful in identifying specific driver mutations in patients with complex ameloblastoma. Targeted treatment has been employed with success in achieving treatment response.
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Affiliation(s)
- James C Gates
- Department of Oral and Maxillofacial Surgery, Hospital of the University of Pennsylvania, Penn Medicine, Philadelphia, Pennsylvania, USA
| | | | - Elliot Cherkas
- Sidney Kimmel Medical College, Thomas Jefferson University, Philadelphia, Pennsylvania, USA
| | - Aditya V Shreenivas
- Department of Medical Oncology, Medical College of Wisconsin, Milwaukee, Wisconsin, USA
| | - Dennis Kraus
- Director of Oncology, Centura Health, Centennial, Colorado, USA
| | | | | | - Jennifer Johnson
- Department of Medical Oncology, Thomas Jefferson University, Philadelphia, Pennsylvania, USA
| | - Jeffrey S Ross
- Foundation Medicine, Inc., Cambridge, Massachusetts, USA
- Department of Pathology, Upstate Medical University, Syracuse, New York, USA
- Department of Urology, Upstate Medical University, Syracuse, New York, USA
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Limpin ET, Lasap-Go EL, David-Paloyo FP, Paloyo SR, Bisquera OC, Ayuste EC. Metastasizing ameloblastoma of the breast: A case report. Int J Surg Case Rep 2022; 101:107800. [PMID: 36436420 PMCID: PMC9691435 DOI: 10.1016/j.ijscr.2022.107800] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/27/2022] [Revised: 11/21/2022] [Accepted: 11/22/2022] [Indexed: 11/27/2022] Open
Abstract
INTRODUCTION Ameloblastomas are slow growing and locally aggressive odontogenic tumors with a high propensity for recurrence. It frequently arises in the mandible and has been reported to metastasize commonly in the lungs. An updated World Health Organization classification re-categorized metastasizing ameloblastomas under benign tumors. Other rare metastatic sites include the skull, maxilla, kidney, and liver. CASE PRESENTATION We present a 53-year-old female with a gradually enlarging right breast mass for 2 years. She previously underwent right hemimandibulectomy with clavicular bone grafting 15 years ago for a primary ameloblastoma. Preoperative imaging showed a resectable, heterogenous right breast mass with a biopsy revealing spindle cell neoplasm. She subsequently underwent radical mastectomy with a latissimus dorsi myocutaneous flap as a reconstructive procedure. Histopathologic findings were consistent with a metastasizing ameloblastoma. The patient remains disease-free as of most recent follow-up. DISCUSSION There are several proposed mechanisms for metastasizing ameloblastomas. Based on the history and location of the tumor, we surmised that tumor seeding from the first surgery done 15 years ago may explain this rare occurrence. Preoperative imaging and biopsy determine resectability and surgical approach. Radical surgery is frequently performed which largely depends on the site of the tumor. Complete primary resection with adequate margins remains to be the treatment of choice to prevent recurrence or metastasis. The role of adjuvant radiotherapy or chemotherapy are still to be established. CONCLUSION This case highlights the value of history-taking and having a high-index of suspicion for metastasis several years after primary resection of ameloblastomas.
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Affiliation(s)
- Emmanuel T. Limpin
- Department of Surgery, University of the Philippines-Philippine General Hospital, Philippines
| | - Edna L. Lasap-Go
- Department of Laboratories, University of the Philippines-Philippine General Hospital, Philippines,College of Medicine, University of the Philippines-Manila, Philippines
| | - Ferri P. David-Paloyo
- Department of Surgery, University of the Philippines-Philippine General Hospital, Philippines,College of Medicine, University of the Philippines-Manila, Philippines
| | - Siegfredo R. Paloyo
- Department of Surgery, University of the Philippines-Philippine General Hospital, Philippines,College of Medicine, University of the Philippines-Manila, Philippines,Corresponding author at: Department of Surgery, University of the Philippines-Philippine General Hospital, Philippines.
| | - Orlino C. Bisquera
- Department of Surgery, University of the Philippines-Philippine General Hospital, Philippines,College of Medicine, University of the Philippines-Manila, Philippines
| | - Eduardo C. Ayuste
- Department of Surgery, University of the Philippines-Philippine General Hospital, Philippines,College of Medicine, University of the Philippines-Manila, Philippines
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Gene Expression and Immunochemistry Analysis of ADAMTS-1 and Versican in Ameloblastoma. Int J Dent 2022; 2022:5235376. [PMID: 36338393 PMCID: PMC9629950 DOI: 10.1155/2022/5235376] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/10/2022] [Revised: 09/23/2022] [Accepted: 10/11/2022] [Indexed: 11/23/2022] Open
Abstract
Background Ameloblastoma is a benign but locally invasive odontogenic epithelial tumor, associated with a high recurrence rate after treatment. The action of enzymes of the metalloproteinase family is important to the degraded extracellular matrix, contributing to invasion. Thus, this study aimed to investigate the gene and protein expression of ADAMTS-1 and versican in ameloblastoma. Materials and Methods Twenty cases of ameloblastoma (n = 20) and ten dental follicles (DF) (n = 10) were used as a source for immunochemistry and quantitative RT-PCR for determining the protein and mRNA expressions of the concerned genes, respectively. Moreover, western blot and indirect immunofluorescence analysis were performed in AME cells. Results ADAMTS-1 and versican were overexpressed in DF than ameloblastoma by RT-PCR. However, in the immunolocalization analysis, ADAMTS-1 was expressed in ameloblastoma more than in DF and versican immunostaining obtained a similar pattern between ameloblastoma and DF. Indirect immunofluorescence detected the ADAMTS-1 and versican expression in cell lines derived from ameloblastoma. Western blot from cell lysate and conditioned medium detected ADAMTS-1 bands representing full-length and different processed forms. Monensin treatment confined ADAMTS-1 in the cell cytoplasm. Versican fragments also were detected in different compartments, intracellular and conditioned medium, allowing the versican process by ADAMTS-1. Conclusion This study showed a distinct expression of ADAMTS-1 and versican in ameloblastoma and DF, with ADAMTS-1 protein higher expression observed in ameloblastoma and possibly cleaved versican. These findings suggested that ADAMTS-1 may participate in tumor invasion, especially for the degradation of substrates (versican) in the ECM.
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Park JU, Jung JW. Metastatic ameloblastoma with postoperative accelerated tumor growth treated with carboplatin and paclitaxel: a case report. KOSIN MEDICAL JOURNAL 2022. [DOI: 10.7180/kmj.21.030] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022] Open
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Treatment efficacy and prognosis of pulmonary metastasizing ameloblastoma: a systematic review. Int J Oral Maxillofac Surg 2021; 51:579-590. [PMID: 34462177 DOI: 10.1016/j.ijom.2021.07.016] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/02/2021] [Revised: 05/01/2021] [Accepted: 07/23/2021] [Indexed: 11/23/2022]
Abstract
The aim of this review was to integrate the current literature into a comprehensive evaluation of pulmonary metastasizing ameloblastoma (MA). Related articles, published since January 2000, were reviewed. The PubMed, Embase, Cochrane Database of Systematic Reviews, and Web of Science databases were searched based on the PRISMA guidelines. Twenty-four studies, including 28 case reports, met the eligibility criteria. The mean ± standard deviation disease-free interval after primary treatment was 12.1 ± 9.3 years. Adjuvant therapy (surgery and chemoradiotherapy) is considered appropriate for pulmonary MA patients presenting with rapid progression or apparent pulmonary symptoms. The proportion of patients undergoing simple follow-up (observation) has increased rapidly in the past 10 years. Meanwhile, among the three treatment groups (observation, surgery, and adjuvant therapy), no significant difference was found in the efficacy (P = 0.081) or progression-free survival (P = 0.263). The 5-year and 10-year overall survival rates were 72.4% and 52.8%, respectively. This study provides insights that could help surgeons with the management of patients presenting with pulmonary MA.
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Collins AP, Mubarak N, Hemaidan HS, Hemaidan SM, Hemaidan A. Malignant Ameloblastoma with Hepatic Metastasis in a 38-Year-Old Haitian Woman. AMERICAN JOURNAL OF CASE REPORTS 2021; 22:e929422. [PMID: 34175884 PMCID: PMC8255087 DOI: 10.12659/ajcr.929422] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/23/2022]
Abstract
BACKGROUND Primary malignant ameloblastoma is a very rare tumor of the dental lamina epithelium. Similar to the benign ameloblastoma, the mass is without significant histological atypia, but the malignant type may present with metastases, most commonly to the lungs. The average age of diagnosis is 34 years, and the malignancy affects men and women equally. The tumors often present with an insidious growth and have a median survival from time of diagnosis of 17.6 years. Due to the rarity of this lesion, a standard of care has not yet been established. CASE REPORT A 38-year-old Haitian woman, who initially presented with a large primary malignant ameloblastoma of the angle of the mandible, experienced a recurrence in the floor of the mouth 30 months after surgical resection. In 2018, 2 years after the removal of the recurrent tumor, the patient presented with ascites, right-sided abdominal pain, weight loss, and a palpable liver mass. Laparoscopic exploration demonstrated a complex lateral right liver lobe cyst, suspicious for parasitic infection. Cytological analysis showed positive staining for cytokeratin 5/6, P63, and CD56, indicative of metastatic ameloblastoma of the liver. Consistent cell morphology from the primary tumor and liver cyst was also noted. Following drainage of the cyst, the patient returned to Haiti, where she died in 2020. In Haiti, she lacked appropriate local medical care, leading to the severe progression of her initial primary ameloblastoma and disease recurrence. CONCLUSIONS Malignant ameloblastoma accounts for less than 2% of all odontogenic tumors, as the benign variant is much more common. Distant metastases of these lesions are rare; to date, few cases have presented with hepatic metastases.
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Affiliation(s)
- Andrew P Collins
- College of Medicine, University of Central Florida, Orlando, FL, USA
| | - Naser Mubarak
- College of Medicine, University of Central Florida, Orlando, FL, USA
| | - Hadi S Hemaidan
- Department of Biological Sciences, University of Florida, Gainesville, FL, USA
| | - Sami M Hemaidan
- Department of Biological Sciences, University of Florida, Gainesville, FL, USA
| | - Ammar Hemaidan
- Department of Gastroenterology, Florida State University College of Medicine, Tallahassee, FL, USA
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Medina A, Velasco Martinez I, McIntyre B, Chandran R. Ameloblastoma: clinical presentation, multidisciplinary management and outcome. CASE REPORTS IN PLASTIC SURGERY AND HAND SURGERY 2021; 8:27-36. [PMID: 33681413 PMCID: PMC7901703 DOI: 10.1080/23320885.2021.1886854] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Indexed: 12/03/2022]
Abstract
We studied 21 patients who underwent radical ameloblastoma excision followed by immediate reconstruction. Comorbidities, consumption of alcohol and/or tobacco and BMI status did not contribute to an unfavorable outcome. Giant ameloblastoma (≥5 cm) and/or tumor involving bony curvatures increased surgical complexity, the incidence of complications and hospital stay.
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Affiliation(s)
- Abelardo Medina
- Division of Plastic Surgery, Department of Surgery, School of Medicine, University of Mississippi Medical Center, Jackson, MS, USA
| | - Ignacio Velasco Martinez
- Department of Oral Maxillofacial Surgery and Pathology, School of Dentistry, University of Mississippi Medical Center, Jackson, MS, USA
| | - Benjamin McIntyre
- Division of Plastic Surgery, Department of Surgery, School of Medicine, University of Mississippi Medical Center, Jackson, MS, USA
| | - Ravi Chandran
- Department of Oral Maxillofacial Surgery and Pathology, School of Dentistry, University of Mississippi Medical Center, Jackson, MS, USA
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Zhang G, Zhao L, Wang X, Wang B, Tang W, Xue Q. Pulmonary resection for multiple lung metastasis from ameloblastoma: a rare case report and literature review. Postgrad Med 2020; 133:117-122. [PMID: 32990496 DOI: 10.1080/00325481.2020.1829841] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/27/2023]
Abstract
INTRODUCTION Ameloblastoma is a benign odontogenic epithelial neoplasm. Lung metastasis of ameloblastoma is extremely rare, and its biological behavior is still largely unknown. There is no consensus regarding the best method to treat metastasizing ameloblastoma. CASE PRESENTATION This report documents a 37-year-old female patient with multiple incidental bilateral pulmonary nodules on computed tomography (CT) and a medical history of ameloblastoma of the left mandible. On admission, her physical examinations and laboratory examinations were unremarkable. The patient underwent partial lobectomy of the middle right and lower right lung nodules via video-assisted thoracoscopic surgery, and the pathological diagnosis was confirmed as 'metastasizing ameloblastoma.' No adjuvant therapy was administered, and no evidence of progression was observed at the 12-month follow-up. CONCLUSION This is a rare case with multiple lung metastasis from ameloblastoma who was successfully treated with multiple pulmonary resections. The present study indicated that surgery may be considered an appropriate choice for lung metastasis of ameloblastoma.
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Affiliation(s)
- Guochao Zhang
- Department of Thoracic Surgery, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College , Beijing, China
| | - Liang Zhao
- Department of Thoracic Surgery, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College , Beijing, China
| | - Xuefei Wang
- Department of Breast Surgery, Chinese Academy of Medical Sciences & Peking Union Medical College, Peking Union Medical College and Hospital , Beijing, China
| | - Bingzhi Wang
- Department of Pathology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College , Beijing, China
| | - Wei Tang
- Department of Diagnostic Radiology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College , Beijing, China
| | - Qi Xue
- Department of Thoracic Surgery, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College , Beijing, China
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Weaver AN, Francisque F, Bowles DW. Tumor Regression After Treatment With Lenvatinib in FGFR2-Mutated Ameloblastoma. JCO Precis Oncol 2020; 4:1403-1406. [PMID: 35050789 DOI: 10.1200/po.20.00175] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/16/2023] Open
Affiliation(s)
- Alice N Weaver
- Department of Medicine, Internal Medicine Residency Training Program, University of Alabama at Birmingham, Birmingham, AL
| | - Frantz Francisque
- Department of Medicine, Division of Medical Oncology, University of Colorado, Aurora, CO
| | - Daniel W Bowles
- Department of Medicine, Division of Medical Oncology, University of Colorado, Aurora, CO.,Rocky Mountain Regional VA Medical Center, Aurora, CO
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Blackford Winders C, Bell CM, Goldschmidt S. Case Report: Amyloid-Producing Odontogenic Tumor With Pulmonary Metastasis in a Spinone Italiano-Proof of Malignant Potential. Front Vet Sci 2020; 7:576376. [PMID: 33134357 PMCID: PMC7552887 DOI: 10.3389/fvets.2020.576376] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/26/2020] [Accepted: 08/21/2020] [Indexed: 11/13/2022] Open
Abstract
A 1-year-old male Spinone Italiano dog was treated for an amyloid-producing odontogenic tumor on the right maxilla with a cytoreductive surgery followed by a definitive radiation protocol. Six years later, the dog presented for a new mass on the rostral mandible as well as a lung nodule without recurrence of the original maxillary tumor. Both the mandibular mass and the lung nodule were histologically confirmed to be amyloid-producing odontogenic tumor based on the appearance of sheets and cords of the odontogenic epithelium disrupted by amorphous extracellular amyloid. This case illustrates the metastatic potential for amyloid-producing odontogenic tumor in dogs and asynchronous occurrence of multiple APOTs in the oral cavity.
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Affiliation(s)
| | - Cynthia M Bell
- Specialty Oral Pathology for Animals, Geneseo, IL, United States
| | - Stephanie Goldschmidt
- College of Veterinary Medicine, University of Minnesota, St. Paul, MN, United States
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16
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Shi HA, Ng CWB, Kwa CT, Sim QXC. Ameloblastoma: A succinct review of the classification, genetic understanding and novel molecular targeted therapies. Surgeon 2020; 19:238-243. [PMID: 32712102 DOI: 10.1016/j.surge.2020.06.009] [Citation(s) in RCA: 32] [Impact Index Per Article: 6.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/15/2020] [Accepted: 06/15/2020] [Indexed: 12/30/2022]
Abstract
Ameloblastomas are benign but locally invasive neoplasms which may grow to massive proportions and cause significant morbidity. Although some types of ameloblastoma can be treated predictably with aggressive surgical treatment, recurrent ameloblastoma and metastasising ameloblastoma are still difficult to treat. Recent studies have identified recurrent somatic and activating mutations in the mitogen-activated protein kinase (MAPK) and sonic hedgehog (SHH) signalling pathways in ameloblastoma. This development provided a possibility that molecular targeted therapies can be used as neoadjuvant treatment. In this review, we provide a summary of the latest WHO classification of ameloblastoma, the current understanding of genetic mutations and novel molecular targeted therapies arising from the recent developments.
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Affiliation(s)
- Hongyi Adrian Shi
- Department of Oral & Maxillofacial Surgery, National Dental Centre Singapore, Singapore.
| | - Chee Wee Benjamin Ng
- Department of Oral & Maxillofacial Surgery, National Dental Centre Singapore, Singapore
| | - Chong Teck Kwa
- Department of Oral & Maxillofacial Surgery, National Dental Centre Singapore, Singapore
| | - Qiu Xia Chelsia Sim
- Department of Oral & Maxillofacial Surgery, National Dental Centre Singapore, Singapore
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17
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de Arruda JAA, Arantes DAC, Schuch LF, Mosconi C, Abreu LG, de Andrade BAB, Romañach MJ, Mesquita RA, Silva TA, Batista AC, Mendonça EF. A Rare Case of an Aggressive Clear Cell Variant of Calcifying Epithelial Odontogenic Tumor in the Posterior Maxilla. Int J Surg Pathol 2020; 28:526-535. [PMID: 31986944 DOI: 10.1177/1066896920901755] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/18/2023]
Abstract
A clear cell variant of calcifying epithelial odontogenic tumor is a rare benign odontogenic neoplasm, accounting for 33 cases described in the literature. In this article, we report a challenging example of clear cell variant of calcifying epithelial odontogenic tumor of the posterior maxilla in a 45-year-old female patient showing locally aggressive growth and recurrence. Microscopically, islands of polyhedral cells containing abundant cytoplasm, well-developed intercellular bridges blended with clear cells were observed. The nuclei were frequently pleomorphic and permeated by hyaline calcified material. Immunohistochemistry revealed positivity for pan-cytokeratin (AE1/AE3), cytokeratins (CK-14 and CK-19), Bcl-2, p53, and p63. The Ki-67 proliferative index was ~10%. As odontogenic tumors are rare, when a significant clear cell component is observed, the differential diagnosis with other lesions of the jaws with similar morphology, including other odontogenic tumors with prominent clear cell component, clear cell odontogenic carcinomas, and metastatic tumors, is difficult.
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Affiliation(s)
| | | | | | - Carla Mosconi
- Universidade Federal de Goiás, Goiânia, Goiás, Brazil
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18
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González-González R, López-Verdín S, Lavalle-Carrasco J, Molina-Frechero N, Isiordia-Espinoza M, Carreón-Burciaga RG, Bologna-Molina R. Current concepts in ameloblastoma-targeted therapies in B-raf proto-oncogene serine/threonine kinase V600E mutation: Systematic review. World J Clin Oncol 2020; 11:31-42. [PMID: 31976308 PMCID: PMC6935689 DOI: 10.5306/wjco.v11.i1.31] [Citation(s) in RCA: 28] [Impact Index Per Article: 5.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/25/2019] [Revised: 10/23/2019] [Accepted: 11/06/2019] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Ameloblastomas are common benign epithelial odontogenic neoplasms that present an aggressive and unpredictable behavior that may modify treatment strategies. Different signaling pathways that participate in the progression of these tumors have been identified. B-raf proto-oncogene serine/threonine kinase (BRAF) is a protein involved in the behavior of ameloblastomas, and it is related to many cell mechanisms. BRAF gene mutations have been identified in ameloblastomas, of which the BRAF V600E (valine substituted by glutamic acid at amino acid 600) mutation has been the most common and can be present concomitantly with other mutations that may be involved in its behavior. Targeted therapies have been used as an alternative in the case of resistance or contraindications to conventional treatments. AIM To document the presence of BRAF V600E and additional mutations, their behavior, and targeted therapies in these tumors. METHODS An electronic literature search was conducted according to PRISMA guidelines in PubMed/MEDLINE, Cochrane, EMBASE, and SpringerLink using the terms "ameloblastomas", "BRAF V600E", "additional mutations", and "targeted therapies". Ameloblastomas were classified according to WHO guidelines. Inclusion criteria were articles in English, published not more than 10 years ago, and studies with laboratory works related to BRAF V600E. Articles were evaluated by two independent reviewers and retrieved for full-text evaluation. The EBLIP Critical Appraisal Checklist was used to evaluate the quality of the eligible studies. Descriptive statistical analysis was performed. RESULTS Two independent reviewers, with a substantial concordance indicated by a kappa coefficient of k = 0.76, evaluated a total of 19 articles that were included in this study. The analysis registered 521 conventional ameloblastomas (AM), 81 unicystic ameloblastomas (UA), 13 ameloblastic carcinomas (AC), three metastatic ameloblastomas (MA), and six peripheral ameloblastomas (PA), of which the histopathological type, anatomic location, laboratory tests, expression of BRAF mutation, and additional mutations were registered. The BRAF V600E mutation was found in 297 AM (57%), 63 UA (77.7%), 3 AC (23%), 1 MA (50%), and 5 PA (83.3%). Follicular type predominated with a total of 116 cases (40%), followed by plexiform type with 63 cases (22.1%). Furthermore, both types presented additional mutations, in which alterations in JAK3 P132T, SMARCB1, PIK3CA, CTNNB1, SMO, and BRAF G606E genes were found. Four case reports were found with targeted therapy to BRAF V600E. CONCLUSION The identification of BRAF V600E and additional mutations as an aid in targeted therapies has been a breakthrough in alternative treatments of ameloblastomas where surgical treatments are contraindicated.
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Affiliation(s)
- Rogelio González-González
- Department of Research, School of Dentistry, Universidad Juárez del Estado de Durango, Durango 34000, Mexico
| | - Sandra López-Verdín
- Research Institute of Dentistry, Health Science Center, Universidad de Guadalajara, Guadalajara 4430, Mexico
| | - Jesús Lavalle-Carrasco
- Department of Research, School of Dentistry, Universidad Juárez del Estado de Durango, Durango 34000, Mexico
| | - Nelly Molina-Frechero
- Department of Health Care, Xochimilco Unit, Universidad Autónoma Metropolitana Xochimilco, México 04960, Mexico
| | - Mario Isiordia-Espinoza
- Department of Clinics, Biomedical Sciences Division, Centro Universitario de los Altos, Universidad de Guadalajara, Tepetitlán de Morelos 47620, Mexico
| | - Ramón G Carreón-Burciaga
- Department of Research, School of Dentistry, Universidad Juárez del Estado de Durango, Durango 34000, Mexico
| | - Ronell Bologna-Molina
- Molecular Pathology Area, School of Dentistry, Universidad de la República, Montevideo 11600, Uruguay
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19
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Li D, Xu S, Sun M, Qiao L, Wang L, Liu Y. MAID chemotherapy regimen as a treatment strategy for metastatic malignant ameloblastoma: A case report. Medicine (Baltimore) 2019; 98:e15873. [PMID: 31232921 PMCID: PMC6636961 DOI: 10.1097/md.0000000000015873] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/14/2023] Open
Abstract
RATIONALE Ameloblastoma is generally characterized as a benign tumor originating in odontogenic epithelium. However, few cases of metastatic malignant ameloblastoma have also been reported. Due to the low incidence of malignant ameloblastoma, there is no established treatment regimen. To explore effective treatment for malignant ameloblastoma, we reported this case study. PATIENTS CONCERNS This report described a case of a 28-year-old malignant ameloblastoma female patient with multiple metastasis (brain and lung). DIAGNOSES The patient presented ameloblastoma of the left mandible in 2012. Three years later, local recurrence and brain metastasis was observed during a follow-up examination. Five years later, malignant ameloblastoma was detected by imaging and immunohistochemistry in the bilateral multiple pulmonary nodules and mediastinal lymph nodes. INTERVENTIONS The patient was initially treated with tumor resection. Three years later after local recurrence and brain metastasis, she was accepted the extensive mandibulectomy supplemented with brain stereotactic body radiotherapy (SBRT). When diagnosed with pulmonary metastasis, the patient received combined chemotherapy regimen of MAID (mesna, adriamycin, ifosfamide and dacarbazine) for 6 cycles. OUTCOMES The efficacy evaluation was partial remission (PR) after the 6 cycles of MAID. The last patient follow-up was July 24th 2018, and no evidence of progression was observed. The progression-free survival (PFS) of the patient was more than 9 months. LESSONS Surgical resection is the optimal treatment for locally recurrent ameloblastoma. SBRT may be an effective treatment for unresectable oligometastasis of malignant ameloblastoma. Finally, combined chemotherapy of MAID showed encouraging effects in the management of metastatic malignant ameloblastoma.
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Affiliation(s)
- Danyang Li
- Department of Medical Oncology of Zhengzhou University Affiliated Cancer Hospital
| | - Shuning Xu
- Department of Medical Oncology of Zhengzhou University Affiliated Cancer Hospital
| | - Miaomiao Sun
- Department of Pathology of Zhengzhou University Affiliated Cancer Hospital
| | - Lei Qiao
- Department of Medical Oncology of Zhengzhou University Affiliated Cancer Hospital
| | - Lifeng Wang
- Department of Imaging of Zhengzhou University Affiliated Cancer Hospital, Henan Cancer Hosptial, Zhengzhou, Henan, China
| | - Ying Liu
- Department of Medical Oncology of Zhengzhou University Affiliated Cancer Hospital
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20
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You Z, Liu SP, Du J, Wu YH, Zhang SZ. Advancements in MAPK signaling pathways and MAPK-targeted therapies for ameloblastoma: A review. J Oral Pathol Med 2019; 48:201-205. [PMID: 30489659 DOI: 10.1111/jop.12807] [Citation(s) in RCA: 43] [Impact Index Per Article: 7.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/07/2018] [Revised: 11/14/2018] [Accepted: 11/22/2018] [Indexed: 12/12/2022]
Abstract
Numerous signal transduction pathways are closely associated with the occurrence, development, and prognosis of ameloblastoma (AM). Mitogen-activated protein kinase (MAPK) is a serine/threonine-specific protein kinase that transduces intracellular signals in critical cellular phenomena. A number of recent analyses have reported that the MAPK signaling pathway contributes significantly to AM. High-throughput DNA sequencing methods, such as next-generation sequencing using Illumina have yielded advancements in studies on MAPK signaling pathways and their association with AM; in particular, BRAF V600E is mediated by the activation of the Ras/Raf/MAPK pathway. This review discusses advancements in studies on MAPK signaling pathways and MAPK-targeted inhibitors or antibodies, along with the merits and demerits of MAPK-targeted therapies, finally followed by a discussion regarding more efficient potential MAPK-targeted therapies to treat AM with few side effects, thereby providing novel insights into targeted therapy for AM.
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Affiliation(s)
- Zhu You
- Shandong Provincial Key Laboratory of Oral Tissue Regeneration, School of Stomatology, Shandong University, Jinan, Shandong Province, China.,Department of Oral and Maxillofacial Surgery, School of Stomatology, Shandong University, Jinan, Shandong Province, China.,Department of Oral and Maxillofacial Surgery, Shandong Provincial Hospital, Affiliated to Shandong University, Jinan, Shandong, China
| | - Shao-Peng Liu
- Shandong Provincial Key Laboratory of Oral Tissue Regeneration, School of Stomatology, Shandong University, Jinan, Shandong Province, China.,Department of Oral and Maxillofacial Surgery, School of Stomatology, Shandong University, Jinan, Shandong Province, China
| | - Jing Du
- Shandong Provincial Key Laboratory of Oral Tissue Regeneration, School of Stomatology, Shandong University, Jinan, Shandong Province, China.,Department of Endodontics, School of Stomatology, Shandong University, Jinan, Shandong Province, China
| | - Yi-Hua Wu
- Department of Periodontology, Shandong Provincial Hospital, Affiliated to Shandong University, Jinan, Shandong, China
| | - Shi-Zhou Zhang
- Department of Oral and Maxillofacial Surgery, Shandong Provincial Hospital, Affiliated to Shandong University, Jinan, Shandong, China
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21
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Aoki T, Akiba T, Kondo Y, Sasaki M, Kajiwara H, Ota Y. The use of radiation therapy in the definitive management of ameloblastic carcinoma: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol 2018; 127:e56-e60. [PMID: 30393089 DOI: 10.1016/j.oooo.2018.09.009] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/19/2018] [Revised: 09/19/2018] [Accepted: 09/24/2018] [Indexed: 11/27/2022]
Abstract
Although rare, ameloblastic carcinoma (AC) is one of the most common malignant tumors occurring in the odontogenic epithelium. Evidenced-based medical guidelines for AC are not established because AC is a rare type of cancer. However, surgery for AC is the primary initial treatment modality. A few reports suggest that radiotherapy (RTx) is effective for AC; however, the tumor is generally considered to be radioresistant. Furthermore, if RTx is performed for AC, osteonecrosis may occur because most of the ACs arise from the jaw. Here, we report a case of an older patient with maxillary AC who had underlying medical problems and who received conventional RTx with good outcomes. Ten years after the treatment, no major adverse event accompanying irradiation, local recurrence, or metastasis was observed. We discuss the therapeutic effect and usefulness of RTx on AC. As indicated by our findings, RTx for AC may represent a definitive treatment strategy for patients with poor general conditions or those with tumors that cannot be resected completely.
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Affiliation(s)
- Takayuki Aoki
- Department of Oral and Maxillofacial Surgery, Tokai University School of Medicine, Isehara, Kanagawa, Japan.
| | - Takeshi Akiba
- Department of Radiation Oncology, Tokai University School of Medicine, Isehara, Kanagawa, Japan
| | - Yusuke Kondo
- Department of Pathology, Tokai University School of Medicine, Isehara, Kanagawa, Japan
| | - Masashi Sasaki
- Department of Oral and Maxillofacial Surgery, Tokai University School of Medicine, Isehara, Kanagawa, Japan
| | - Hiroshi Kajiwara
- Department of Pathology, Tokai University School of Medicine, Isehara, Kanagawa, Japan
| | - Yoshihide Ota
- Department of Oral and Maxillofacial Surgery, Tokai University School of Medicine, Isehara, Kanagawa, Japan
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22
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Al Sineedi F, Aruveetil YA, Kavarodi AM, Harbi SO. Bilocular unicystic ameloblastoma of the mandible in a 9 yr old child - A diagnostic and management dilemma. Saudi Dent J 2018; 30:250-255. [PMID: 29942110 PMCID: PMC6011211 DOI: 10.1016/j.sdentj.2018.03.001] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/15/2015] [Revised: 02/20/2018] [Accepted: 03/02/2018] [Indexed: 11/27/2022] Open
Abstract
Unicystic ameloblastoma is a less encountered variant of the ameloblastoma that usually presented as unicystic lesions of jaw occurring in 3rd and 4th decades of life. It shows a typical ameloblastomatous epithelium lining the cyst cavity, with or without variable tumor proliferations. The case presented here is of a 9 yr old boy who was referred to our center for the management of a large diffuse swelling on the right side of the face. Clinical and radiologic evaluation showed two interconnected cystic lesions in the right body and the symphyseal regions of the mandible associated with impacted canines bilaterally. The initial histopathology of both cystic spaces showed the lesion to be dentigerous cysts and the results were reconfirmed in two other centers. A complete surgical enucleation of this bilocular cyst was done sparing the impacted teeth. The histopathologic examination of the post-operative specimen showed features of Unicystic Ameloblastoma. The patient was followed up on a regular basis for more than 3 years. There is no signs of recurrence and his latest radiographic examinations shows good bone formation. The impacted teeth are erupting into position. This case reports the difficulty in clinical diagnosis and the peculiar bilocular presentation of unicystic ameloblastoma which was conservatively managed by surgical enucleation of the complete lesion, sparing the dentition.
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23
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Abtahi MA, Zandi A, Razmjoo H, Ghaffari S, Abtahi SM, Jahanbani-Ardakani H, Kasaei Z, Kasaei-Koupaei S, Sajjadi S, Sonbolestan SA, Abtahi SH. Orbital invasion of ameloblastoma: A systematic review apropos of a rare entity. J Curr Ophthalmol 2017; 30:23-34. [PMID: 29564405 PMCID: PMC5859465 DOI: 10.1016/j.joco.2017.09.001] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/01/2017] [Revised: 08/20/2017] [Accepted: 09/01/2017] [Indexed: 12/22/2022] Open
Abstract
Purpose Ameloblastoma is a non-encapsulated and slow-growing tumor with high recurrence rate. Orbital involvement by this neoplasm is an extremely rare entity. In this study, we present a systematic review on this situation along with clinical and paraclinical features of a case. Methods An electronic search was conducted on major medical sources. Data of the cases in the literature in addition to our own case were extracted, summarized, and statistically analyzed. Results A total of 36 other cases from 20 relevant studies were also reviewed. Review topics included epidemiology, clinical presentation, pathologic features, differential diagnosis, imaging, treatment, and prognosis. We provided a five-year history of a 50-year-old man with orbital/skull base invasion of plexiform maxillary ameloblastoma. Conclusions Maxillary ameloblastoma is a locally aggressive neoplasm, and physicians must be alert to the biologic behavior of this tumor to detect any invasion to critical structures such as orbit and cranium. Orbital ameloblastoma causes significant morbidity and mortality. We advocate meticulous patient follow-up with regular clinical examinations and paraclinical work-up for timely detection of any invasion or recurrence. The best must be done to avoid extensions by aggressive removal of maxillary ameloblastoma.
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Affiliation(s)
- Mohammad-Ali Abtahi
- Isfahan Eye Research Center, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran.,Department of Ophthalmology, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Alireza Zandi
- Isfahan Eye Research Center, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran.,Department of Ophthalmology, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Hassan Razmjoo
- Isfahan Eye Research Center, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran.,Department of Ophthalmology, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Sara Ghaffari
- School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran.,Isfahan Medical Students Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Seyed-Mojtaba Abtahi
- Department of Otolaryngology, Head and Neck Surgery, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Hamidreza Jahanbani-Ardakani
- School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran.,Isfahan Medical Students Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Zahra Kasaei
- Department of Pathology, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Samira Kasaei-Koupaei
- Department of Pediatric Dentistry, School of Dentistry, Khorasgan University of Medical Sciences, Isfahan, Iran
| | - Sepideh Sajjadi
- School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran.,Isfahan Medical Students Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Seyed-Ali Sonbolestan
- Isfahan Eye Research Center, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran.,Department of Ophthalmology, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Seyed-Hossein Abtahi
- Isfahan Eye Research Center, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran.,Department of Ophthalmology, Feiz Hospital, Isfahan University of Medical Sciences, Isfahan, Iran.,Isfahan Medical Students Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
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24
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Valkadinov I, Conev N, Dzhenkov D, Donev I. Rare case of ameloblastoma with pulmonary metastases. Intractable Rare Dis Res 2017; 6:211-214. [PMID: 28944145 PMCID: PMC5608933 DOI: 10.5582/irdr.2017.01032] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/05/2022] Open
Abstract
Ameloblastoma is a rare low-grade odontogenic tumor of epithelial origin. The World Health Organization (WHO) has defined malignant ameloblastoma (MA) as a histologically benign-appearing ameloblastoma that has metastasized. Treatment of the primary ameloblastoma usually consists of radical excision of the tumor and adjuvant radiotherapy. Chemotherapy should be used to treat metastases due to its indolent clinical course. Presented here is the case of a 43-year-old woman who was admitted to a hospital in 2006 with a large mass involving the neck and left mandible. The mass had formed over years and had been neglected. The woman was diagnosed with a primary ameloblastoma of the mandible. Surgical resection was performed, followed by adjuvant radiotherapy. In September 2016, she was admitted again, and the findings were consistent with metastases of the previously identified ameloblastoma to the lungs. The patient was evaluated for further chemotherapy with 6 cycles of cisplatin at a dose of 100 mg/m2 on day 1, 5-FU at a dose of 1000 mg/m2/day on day 1-4 (3 wk), and pegylated filgrastim. The current case represents the classical course of a rare disease, which in this instance involved the common presentation of MA. This case is a valid incidence of MA based on the typical histology, findings from a lung biopsy, the immunohistochemical profile of the tumor, the typical clinical features, and a history of a previous primary disease.
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Affiliation(s)
- Ivan Valkadinov
- Clinic of Medical Oncology, UMHAT “St. Marina”, Varna, Bulgaria
| | - Nikolay Conev
- Clinic of Medical Oncology, UMHAT “St. Marina”, Varna, Bulgaria
| | - Dian Dzhenkov
- Centre of Clinical Pathology, UMHAT “St. Marina”, Varna, Bulgaria
| | - Ivan Donev
- Clinic of Medical Oncology, UMHAT “St. Marina”, Varna, Bulgaria
- Address correspondence to: Dr. Ivan Donev, Clinic of Medical Oncology, UMHAT “St. Marina”, 1 “Hristo Smirnenski” Blvd., Varna 9000, Bulgaria. E-mail:
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25
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Effiom OA, Ogundana OM, Akinshipo AO, Akintoye SO. Ameloblastoma: current etiopathological concepts and management. Oral Dis 2017; 24:307-316. [PMID: 28142213 DOI: 10.1111/odi.12646] [Citation(s) in RCA: 149] [Impact Index Per Article: 18.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/19/2017] [Accepted: 01/20/2017] [Indexed: 02/06/2023]
Abstract
Ameloblastoma is a benign odontogenic tumor of epithelial origin. It is locally aggressive with unlimited growth capacity and has a high potential for malignant transformation as well as metastasis. Ameloblastoma has no established preventive measures although majority of patients are between ages 30 and 60 years. Molecular and genetic factors that promote oncogenic transformation of odontogenic epithelium to ameloblastoma are strongly linked to dysregulation of multiple genes associated with mitogen-activated protein kinase, sonic hedgehog, and WNT/β-catenin signaling pathways. Treatment of ameloblastoma is focused on surgical resection with a wide margin of normal tissue because of its high propensity for locoregional invasion; but this is often associated with significant patient morbidity. The relatively high recurrence rate of ameloblastoma is influenced by the type of molecular etiological factors, the management approach, and how early the patient presents for treatment. It is expected that further elucidation of molecular factors that orchestrate pathogenesis and recurrence of ameloblastoma will lead to new diagnostic markers and targeted drug therapies for ameloblastoma.
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Affiliation(s)
- O A Effiom
- Department of Oral and Maxillofacial Pathology/Biology, Faculty of Dental Sciences, University of Lagos, Lagos, Nigeria
| | - O M Ogundana
- Department of Oral and Maxillofacial Pathology/Biology, Faculty of Dental Sciences, University of Lagos, Lagos, Nigeria
| | - A O Akinshipo
- Department of Oral and Maxillofacial Pathology/Biology, Faculty of Dental Sciences, University of Lagos, Lagos, Nigeria
| | - S O Akintoye
- Department of Oral Medicine, School of Dental Medicine, University of Pennsylvania, Philadelphia, PA, USA
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26
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Abstract
Oral squamous cell carcinoma (OSCC) has been estimated to be the sixth most common cancer worldwide. The distant metastasis plays a critical role in the management and prognosis in oral cancer patients. Regarding the distant metastasis from the oral cancer, the hypopharynx is the most common primary site, followed by the base of tongue and anterior tongue. The present review article analyzes the characteristics of the distant metastases from the oral cavity from 1937 to 2015.
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Affiliation(s)
- Soussan Irani
- Department of Oral Pathology, Dental Faculty, Hamadan University of Medical Sciences, Hamadan, Iran
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27
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Kennedy WR, Werning JW, Kaye FJ, Mendenhall WM. Treatment of ameloblastoma and ameloblastic carcinoma with radiotherapy. Eur Arch Otorhinolaryngol 2016; 273:3293-7. [DOI: 10.1007/s00405-016-3899-3] [Citation(s) in RCA: 28] [Impact Index Per Article: 3.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/12/2015] [Accepted: 01/07/2016] [Indexed: 11/28/2022]
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28
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Kishore M, Panat SR, Aggarwal A, Upadhyay N, Agarwal N. Ameloblastic Carcinoma: A Case Report. J Clin Diagn Res 2015; 9:ZD27-8. [PMID: 26393226 DOI: 10.7860/jcdr/2014/8246.6248] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/17/2013] [Accepted: 09/03/2014] [Indexed: 11/24/2022]
Abstract
Ameloblastic carcinoma is a rare malignant lesion with characteristic histologic features and behavior that dictates more aggressive surgical approach than that of a simple ameloblastoma. Here we present a case of ameloblastic carcinoma of the mandible in a 30-year-old male patient with a clinical course of typical aggressiveness and extensive local destruction.
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Affiliation(s)
- Mallika Kishore
- Post Graduate Student, Department of Oral Medicine and Radiology, Institute of Dental Sciences , Bareilly, U.P, India
| | - Sunil R Panat
- Professor & Head, Department of Oral Medicine and Radiology, Institute of Dental Sciences , Bareilly, U.P, India
| | - Ashish Aggarwal
- Senior Lecturer, Department of Oral Medicine and Radiology, Institute of Dental Sciences , Bareilly, U.P, India
| | - Nitin Upadhyay
- Senior Lecturer, Department of Oral Medicine and Radiology, Institute of Dental Sciences , Bareilly, U.P, India
| | - Nupur Agarwal
- Senior Lecturer, Department of Oral Medicine and Radiology, Institute of Dental Sciences , Bareilly, U.P, India
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Larrañaga JJ, Sahovaler A, Picco PI, Mazzaro EL, Figari MF. Management Issues in the Treatment of an Ameloblastoma with an Atypical Presentation. Craniomaxillofac Trauma Reconstr 2015; 8:257-61. [PMID: 26269737 DOI: 10.1055/s-0035-1549012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/21/2014] [Accepted: 11/01/2014] [Indexed: 10/23/2022] Open
Abstract
Benign ameloblastoma (BA) is the most common tumor arising from the odontogenic epithelium. Surgical resection with adequate margins is the mainstay of treatment due to the high index of tumor recurrence when not completely excised. Although locoregional spread has been described in the literature, it is very uncommon. We describe the treatment and follow-up of a 22-year-old woman with multiple recurrences and locoregional spread of a mandibular ameloblastoma who was referred to our center after several tumor resections with subsequent reconstructions. After a complete macroscopical removal of a new recurrence, the mandible was primarily reconstructed. A local homolateral recurrence and a second lesion in the contralateral maxilla were detected after 1-year follow-up and accordingly treated. After 4 years the patient showed a new tumor in the temporal fossa and was reoperated. The histopathology was consistent with a BA in all cases. Even though it is rare, locoregional spread of BA has been reported previously. Recurrences discovered during follow-up may require further resections. A close follow-up is mandatory, and treatment of these cases may result demanding requiring a multiple team approach, including oncologists and radiotherapists.
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Affiliation(s)
- Juan José Larrañaga
- Division of Head and Neck, Department of General Surgery, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
| | - Axel Sahovaler
- Division of Head and Neck, Department of General Surgery, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
| | - Pedro Ignacio Picco
- Division of Head and Neck, Department of General Surgery, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
| | - Eduardo Luis Mazzaro
- Division of Head and Neck, Department of General Surgery, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
| | - Marcelo Fernando Figari
- Division of Head and Neck, Department of General Surgery, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
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Ameloblastoma: a clinical review and trends in management. Eur Arch Otorhinolaryngol 2015; 273:1649-61. [DOI: 10.1007/s00405-015-3631-8] [Citation(s) in RCA: 91] [Impact Index Per Article: 9.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/03/2015] [Accepted: 04/15/2015] [Indexed: 12/22/2022]
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Basat SO, Öreroğlu AR, Orman C, Aksan T, Üsçetin İ, Akan M. Recurrent Ameloblastoma in the Free Fibula Flap: Review of Literature and an Unusual Case Report. J Maxillofac Oral Surg 2015. [PMID: 26225082 DOI: 10.1007/s12663-014-0704-y] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022] Open
Abstract
Ameloblastoma is the second most common odontogenic tumor of the oral cavity with the primary site being the mandible. The ratio of maxillomandibular involvement however is 5:1 in favor of the mandible. The most common complaint is a painless swelling over the mandibular area. Despite its benign nature, ameloblastoma has a high local recurrence rate, with the most recurrences seen within 5 years after operation. Biopsy and radiological evaluation may be helpful in differentiating the subtypes of ameloblastoma. Differentiation is important because some subtypes are more aggressive than the others. Preoperative planning may be done according to this classification, which can help decrease the recurrence rate. In our case, a 26-year-old female patient with recurrent ameloblastoma which developed on the fibular flap is presented. The free fibular flap and the left parasymphyseal part of the mandible were totally excised. Ameloblastoma was confirmed on pathological examination. We reconstructed the left mandibular site with a reconstruction plate and recurrence was not seen during follow up period.
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Affiliation(s)
- Salih Onur Basat
- Plastic, Reconstructive and Aesthetic Surgery Clinic, Okmeydani Training and Research Hospital, Darulaceze cad. No: 25 Şişli, Istanbul, Turkey
| | | | - Cagdas Orman
- Plastic, Reconstructive and Aesthetic Surgery Clinic, Okmeydani Training and Research Hospital, Darulaceze cad. No: 25 Şişli, Istanbul, Turkey
| | - Tolga Aksan
- Plastic, Reconstructive and Aesthetic Surgery Clinic, Okmeydani Training and Research Hospital, Darulaceze cad. No: 25 Şişli, Istanbul, Turkey
| | - İlker Üsçetin
- Plastic, Reconstructive and Aesthetic Surgery Clinic, Okmeydani Training and Research Hospital, Darulaceze cad. No: 25 Şişli, Istanbul, Turkey
| | - Mithat Akan
- Department of Plastic, Reconstructive and Aesthetic Surgery, Medipol University, Istanbul, Turkey
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Kim Y, Choi SW, Lee JH, Ahn KM. A single cervical lymph node metastasis of malignant ameloblastoma. J Craniomaxillofac Surg 2014; 42:2035-40. [PMID: 25458349 DOI: 10.1016/j.jcms.2014.09.010] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/04/2014] [Revised: 09/05/2014] [Accepted: 09/25/2014] [Indexed: 11/25/2022] Open
Abstract
INTRODUCTION Cervical node metastasis of malignant ameloblastoma is extremely rare. Because of its rarity, there is no standard treatment modality in a single lymph node metastasis in malignant ameloblastoma. MATERIALS AND METHODS Eleven patients of malignant ameloblastoma involving a single cervical lymph node metastasis and one new case were reviewed. Neck treatment was classified into neck dissection and simple excision. Local nodal recurrence, distant metastasis and follow-up periods were investigated. RESULTS Eight patients were treated with neck dissection (group A) and four patients underwent a simple node excision (group B). Two patients in group A experienced multiple organ metastases such as liver and lung seven months and 13 years after neck dissection respectively. The other patients showed no recurrence and metastasis. In group B, there was no report of a regional neck recurrence and distant metastasis during follow-up of 1-7 years. CONCLUSION Multiple nodes metastasis requires a radical neck dissection; however, simple excision with close follow-up may be used in a single node metastasis in malignant ameloblastoma.
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Affiliation(s)
- Yoori Kim
- Department of Oral and Maxillofacial Surgery, College of Medicine, University of Ulsan, Asan Medical Center, Seoul, South Korea
| | - Sung-Weon Choi
- Oral Oncology Clinic, National Cancer Center, Gyeonggi-do, South Korea
| | - Jong-Ho Lee
- Department of Oral and Maxillofacial Surgery, School of Dentistry, Seoul National University, Seoul, South Korea
| | - Kang-Min Ahn
- Department of Oral and Maxillofacial Surgery, College of Medicine, University of Ulsan, Asan Medical Center, Seoul, South Korea.
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Jayaraj G, Sherlin HJ, Ramani P, Premkumar P, Natesan A, Ramasubramanian A, Jagannathan N. Metastasizing Ameloblastoma – A perennial pathological enigma? Report of a case and review of literature. J Craniomaxillofac Surg 2014; 42:772-9. [DOI: 10.1016/j.jcms.2013.11.009] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/04/2013] [Revised: 08/23/2013] [Accepted: 11/04/2013] [Indexed: 01/14/2023] Open
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Koca T, Başaran H, Arslan D, Sezen D, Çerkeşli ZA, Kılınç Ö, Karaca S, Başsorgun Cİ, Okay HÖ, Demirci M. Prominent response with helical tomotherapy in recurrent ameloblastic carcinoma of maxillary sinus: a case report. Radiat Oncol 2014; 9:157. [PMID: 25027948 PMCID: PMC4108973 DOI: 10.1186/1748-717x-9-157] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/28/2014] [Accepted: 07/11/2014] [Indexed: 11/30/2022] Open
Abstract
INTRODUCTION Ameloblastoma is a benign but locally aggressive tumor of odontogenic epithelial tissue. Reports of radiotherapy treatment modalities are limited in the literature. CASE PRESENTATION A thirty-five year old male presented with complaints of headache radiating to his face for about six months and impaired vision. The patient's Positron Emission Tomography (PET) showed a mass in the left maxillary sinus extending to the nasal cavity and invading the adjacent tissues. An R2 (macroscopic residual tumor) surgical resection performed to debulk the tumor. Due to the recurrence and residual mass, the patient was treated with helical tomotherapy. At 2 months post-radiotherapy, patient's vision returned to normal. PET scan showed a significant reduction in lesion size 12 months post-radiation. CONCLUSION In cases of ameloblastic carcinoma with, post-surgical recurrence or patients not suitable for surgical treatment, helical tomotherapy can be an effective treatment option.
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Affiliation(s)
- Timur Koca
- Department of Radiation Oncology, Regional Training and Research Hospital, Erzurum, Turkey
| | - Hamit Başaran
- Department of Radiation Oncology, Regional Training and Research Hospital, Erzurum, Turkey
| | - Deniz Arslan
- Department of Medical Oncology, Regional Training and Research Hospital, Erzurum, Turkey
| | - Duygu Sezen
- Department of Radiation Oncology, Regional Training and Research Hospital, Erzurum, Turkey
| | - Zümrüt Arda Çerkeşli
- Department of Radiation Oncology, Regional Training and Research Hospital, Erzurum, Turkey
| | - Özlem Kılınç
- Department of Radiation Oncology, Regional Training and Research Hospital, Erzurum, Turkey
| | - Sibel Karaca
- Department of Radiation Oncology, Regional Training and Research Hospital, Erzurum, Turkey
| | | | - Hilmi Önder Okay
- Regional Training and Research Hospital, Clinic of Neurosurgery, Erzurum, Turkey
| | - Münir Demirci
- Regional Training and Research Hospital, Clinic of Nuclear Medicine, Erzurum, Turkey
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Abstract
A 74-year-old patient was admitted to our hospital with recurrent ameloblastoma of the right mandible. Multiple lung nodules were noted during the presurgical evaluation. FDG PET/CT was subsequently performed to assess the nature of the nodules and search the possible primary tumors. The images showed abnormal FDG activity not only in the lung nodules but also in the lumbar vertebral body and the liver. Pathologic examination after hepatic biopsy demonstrated metastases from ameloblastoma.
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Lin Y, He JF, Li ZY, Liu JH. Ameloblastoma with varied sites of metastasis: report of two cases and literature review. J Craniomaxillofac Surg 2013; 42:e301-4. [PMID: 24280106 DOI: 10.1016/j.jcms.2013.10.010] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/23/2013] [Revised: 07/29/2013] [Accepted: 10/08/2013] [Indexed: 10/26/2022] Open
Abstract
OBJECTIVE We report two rare cases of lung metastasis from maxillary ameloblastoma, in order to review its risk and analyse the types of metastases that can present with this disease. METHODS A 40-year-old male with multiple recurrences and a 46-year-old female, who had undergone successful surgical treatment of a maxillary ameloblastoma, presented with metastatic lesions. The primary tumour and metastases were benign in both patients. We reviewed and analysed 20 cases of the same condition reported in recent years. RESULTS Our initial treatment for the primary maxillary lesion was performed more than 10 years before the pulmonary lesions presented. Due to the aggressive nature of this tumour, metastases in the lungs and cervical lymph nodes (male patient) were confirmed. CONCLUSION These cases presented a diagnostic challenge due to the multiple and varied sites of recurrence, which indicate the natural behaviour of this tumour. Different routes of metastasis can occur, including implanting, haematogenous, and lymphatic spread. CT-guided percutaneous transthoracic lung biopsy is an important method to confirm metastatic ameloblastoma.
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Affiliation(s)
- Yi Lin
- Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital, College of Medicine, Zhejiang University, #79 Qingchun Road, Hangzhou 310003, Zhejiang, PR China
| | - Jian-feng He
- Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital, College of Medicine, Zhejiang University, #79 Qingchun Road, Hangzhou 310003, Zhejiang, PR China.
| | - Zhi-yong Li
- Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital, College of Medicine, Zhejiang University, #79 Qingchun Road, Hangzhou 310003, Zhejiang, PR China
| | - Jian-hua Liu
- Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital, College of Medicine, Zhejiang University, #79 Qingchun Road, Hangzhou 310003, Zhejiang, PR China
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Ghiam A, Al Zahrani A, Feld R. A case of recurrent metastatic ameloblastoma and hypercalcaemia successfully treated with carboplatin and paclitaxel: long survival and prolonged stable disease. Ecancermedicalscience 2013; 7:323. [PMID: 23781278 PMCID: PMC3680230 DOI: 10.3332/ecancer.2013.323] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/18/2013] [Indexed: 11/20/2022] Open
Abstract
We describe a case of recurrent metastatic malignant ameloblastoma to the lungs with hypercalcaemia in a 47-year-old man. The first lung metastasis was resected nine years after the initial primary, and the tumour recurred with extensive pulmonary metastases 21 years after the primary tumour was resected. This case presented with malignancy-associated hypercalcaemia, likely due to paraneoplastic syndrome, which is exceedingly unusual in association with malignant ameloblastoma. He was successfully treated with carboplatin/paclitaxel and showed the longest survival and stable disease, from the diagnosis of recurrent metastasis, recorded as a case report. This regimen is reasonably well tolerated and can be repeated safely.
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Affiliation(s)
- Af Ghiam
- Princess Margaret Hospital, University Health Network, Toronto, Canada ; Department of Radiation Oncology, University of Toronto, Canada
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