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Donati CM, Medici F, Zamfir AA, Galietta E, Cammelli S, Buwenge M, Masetti R, Prete A, Strigari L, Forlani L, D’Angelo E, Morganti AG. CyberKnife in Pediatric Oncology: A Narrative Review of Treatment Approaches and Outcomes. Curr Oncol 2025; 32:76. [PMID: 39996876 PMCID: PMC11854067 DOI: 10.3390/curroncol32020076] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/23/2024] [Revised: 01/20/2025] [Accepted: 01/27/2025] [Indexed: 02/26/2025] Open
Abstract
Pediatric cancers, while rare, pose unique challenges due to the heightened sensitivity of developing tissues and the increased risk of long-term radiation-induced effects. Radiotherapy (RT) is a cornerstone in pediatric oncology, but its application is limited by concerns about toxicity, particularly secondary malignancies, growth abnormalities, and cognitive deficits. CyberKnife (CK), an advanced robotic radiosurgery system, has emerged as a promising alternative due to its precision, non-invasiveness, and ability to deliver hypofractionated, high-dose RT while sparing healthy tissues. This narrative review explores the existing evidence on CK application in pediatric patients, synthesizing data from case reports, small series, and larger cohort studies. All the studies analyzed reported cases of tumors located in the skull or in the head and neck region. Findings suggest CK's potential for effective tumor control with favorable toxicity profiles, especially for complex or inoperable tumors. However, the evidence remains limited, with the majority of studies involving small sample sizes and short follow-up periods. Moreover, concerns about the "dose-bath" effect and limited long-term data on stochastic risks warrant cautious adoption. Compared to Linac-based RT and proton therapy, CK offers unique advantages in reducing session numbers and enhancing patient comfort, while its real-time tracking provides superior accuracy. Despite these advantages, CK is associated with significant limitations, including a higher potential for low-dose scatter (often referred to as the "dose-bath" effect), extended treatment times in some protocols, and high costs requiring specialized expertise for operation. Emerging modalities like π radiotherapy further underscore the need for comparative studies to identify the optimal technique for specific pediatric cases. Notably, proton therapy remains the benchmark for minimizing long-term toxicity, but its cost and availability limit its accessibility. This review emphasizes the need for balanced evaluations of CK and highlights the importance of planning prospective studies and long-term follow-ups to refine its role in pediatric oncology. A recent German initiative to establish a CK registry for pediatric CNS lesions holds significant promise for advancing evidence-based applications and optimizing treatment strategies in this vulnerable population.
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Affiliation(s)
- Costanza M. Donati
- Radiation Oncology, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Via Albertoni 15, 40128 Bologna, Italy; (C.M.D.); (E.G.); (S.C.); (A.G.M.)
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, 40100 Bologna, Italy; (M.B.); (R.M.); (A.P.); (L.F.)
| | - Federica Medici
- Département de Radiothérapie, Gustave Roussy, 94805 Villejuif, France;
| | - Arina A. Zamfir
- Radiation Oncology, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Via Albertoni 15, 40128 Bologna, Italy; (C.M.D.); (E.G.); (S.C.); (A.G.M.)
| | - Erika Galietta
- Radiation Oncology, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Via Albertoni 15, 40128 Bologna, Italy; (C.M.D.); (E.G.); (S.C.); (A.G.M.)
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, 40100 Bologna, Italy; (M.B.); (R.M.); (A.P.); (L.F.)
| | - Silvia Cammelli
- Radiation Oncology, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Via Albertoni 15, 40128 Bologna, Italy; (C.M.D.); (E.G.); (S.C.); (A.G.M.)
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, 40100 Bologna, Italy; (M.B.); (R.M.); (A.P.); (L.F.)
| | - Milly Buwenge
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, 40100 Bologna, Italy; (M.B.); (R.M.); (A.P.); (L.F.)
| | - Riccardo Masetti
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, 40100 Bologna, Italy; (M.B.); (R.M.); (A.P.); (L.F.)
- Pediatric Oncology and Hematology “Lalla Seràgnoli”, IRCCS Azienda Ospedaliero Universitaria di Bologna, 40128 Bologna, Italy
| | - Arcangelo Prete
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, 40100 Bologna, Italy; (M.B.); (R.M.); (A.P.); (L.F.)
- Pediatric Oncology and Hematology “Lalla Seràgnoli”, IRCCS Azienda Ospedaliero Universitaria di Bologna, 40128 Bologna, Italy
| | - Lidia Strigari
- Department of Medical Physics, IRCCS Azienda Ospedaliero-Universitaria di Bologna, 40128 Bologna, Italy;
| | - Ludovica Forlani
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, 40100 Bologna, Italy; (M.B.); (R.M.); (A.P.); (L.F.)
| | - Elisa D’Angelo
- Department of Radiation Oncology, Bellaria Hospital-AUSL Bologna, 40139 Bologna, Italy;
| | - Alessio G. Morganti
- Radiation Oncology, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Via Albertoni 15, 40128 Bologna, Italy; (C.M.D.); (E.G.); (S.C.); (A.G.M.)
- Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna, 40100 Bologna, Italy; (M.B.); (R.M.); (A.P.); (L.F.)
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Liu S, Li H, Dong Y, Zhang D. Ameloblastic fibrosarcoma of the maxilla arising in an old woman, a rare case report and literature review. BMC Oral Health 2024; 24:743. [PMID: 38937725 PMCID: PMC11212157 DOI: 10.1186/s12903-024-04509-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/21/2024] [Accepted: 06/20/2024] [Indexed: 06/29/2024] Open
Abstract
BACKGROUND Ameloblastic fibrosarcoma (AFS) is a rare malignant odontogenic tumor, commonly occurring in young adults and typically affecting the mandibular region. We report an exceptionally rare and highly atypical case of AFS in an elderly female patient originating from the maxillary bone. CASE PRESENTATION A 66-year-old woman was admitted with a two-week history of a lump in her left upper molar. CT scans suggested a cyst in the maxillary bone. An incisional biopsy revealed a spindle cell neoplasm. MRI showed abnormalities in the left maxilla, indicating a possible tumorous lesion. The patient underwent a subtotal maxillectomy, wide tumor excision, intraoral epithelial flap transplantation, and dental extraction. Histology identified atypical tumor cells with visible mitotic figures. Immunohistochemistry showed negative for PCK and CD34 expression, but positive for Vimentin and SMA expression. The Ki-67 proliferation index ranged from 30 to 50%. These findings suggested a potentially malignant soft tissue tumor in the left maxilla, leaning towards a diagnosis of AFS. The patient received postoperative radiotherapy. There was no recurrence during the six-month follow-up. CONCLUSION Based on repeated pathological evidence, we report a rare case of an elderly female with AFS originating from the maxillary bone. Surgery and postoperative radiotherapy resulted in a favorable outcome.
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Affiliation(s)
- Shiyue Liu
- Department of Oncology, Xiangyang No.1 People's Hospital, Hubei Univeristy of Medicine, Jiefang Road No. 15, XiangYang, Hubei, 441000, China
| | - Hong Li
- Department of Rehabilitation Medicine, Xiangyang No.1 People's Hospital, Hubei Univeristy of Medicine, XiangYang, Hubei, China
| | - Youhong Dong
- Department of Oncology, Xiangyang No.1 People's Hospital, Hubei Univeristy of Medicine, Jiefang Road No. 15, XiangYang, Hubei, 441000, China
| | - Dongdong Zhang
- Department of Oncology, Xiangyang No.1 People's Hospital, Hubei Univeristy of Medicine, Jiefang Road No. 15, XiangYang, Hubei, 441000, China.
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Ward MC, Koyfman SA, Bakst RL, Margalit DN, Beadle BM, Beitler JJ, Chang SSW, Cooper JS, Galloway TJ, Ridge JA, Robbins JR, Sacco AG, Tsai CJ, Yom SS, Siddiqui F. Retreatment of Recurrent or Second Primary Head and Neck Cancer After Prior Radiation: Executive Summary of the American Radium Society® (ARS) Appropriate Use Criteria (AUC): Expert Panel on Radiation Oncology - Head and Neck Cancer. Int J Radiat Oncol Biol Phys 2022; 113:759-786. [PMID: 35398456 DOI: 10.1016/j.ijrobp.2022.03.034] [Citation(s) in RCA: 30] [Impact Index Per Article: 10.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/27/2021] [Revised: 02/16/2022] [Accepted: 03/28/2022] [Indexed: 11/19/2022]
Abstract
BACKGROUND Re-treatment of recurrent or second primary head and neck cancers occurring in a previously irradiated field is complex. Few guidelines exist to support practice. METHODS We performed an updated literature search of peer-reviewed journals in a systematic fashion. Search terms, key questions, and associated clinical case variants were formed by panel consensus. The literature search informed the committee during a blinded vote on the appropriateness of treatment options via the modified Delphi method. RESULTS The final number of citations retained for review was 274. These informed five key questions, which focused on patient selection, adjuvant re-irradiation, definitive re-irradiation, stereotactic body radiation (SBRT), and re-irradiation to treat non-squamous cancer. Results of the consensus voting are presented along with discussion of the most current evidence. CONCLUSIONS This provides updated evidence-based recommendations and guidelines for the re-treatment of recurrent or second primary cancer of the head and neck.
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Affiliation(s)
- Matthew C Ward
- Levine Cancer Institute, Atrium Health, Charlotte, North Carolina; Southeast Radiation Oncology Group, Charlotte, North Carolina.
| | | | | | - Danielle N Margalit
- Dana-Farber/Brigham and Women's Cancer Center, Harvard Medical School, Boston, Massachusetts
| | - Beth M Beadle
- Stanford University School of Medicine, Palo Alto, California
| | | | | | | | | | - John A Ridge
- Fox Chase Cancer Center, Philadelphia, Pennsylvania
| | - Jared R Robbins
- University of Arizona College of Medicine Tucson, Tucson, Arizona
| | - Assuntina G Sacco
- University of California San Diego Moores Cancer Center, La Jolla, California
| | - C Jillian Tsai
- Memorial Sloan Kettering Cancer Center, New York, New York
| | - Sue S Yom
- University of California, San Francisco, California
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Zhu Q, Wang C, Wang Y, Guo S, Ye J. Clinical management of primary odontogenic sarcoma in the mandible: a case report after WHO nomination. J Biomed Res 2022; 36:58-62. [PMID: 35403611 PMCID: PMC8894286 DOI: 10.7555/jbr.35.20210133] [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] [Indexed: 11/08/2022] Open
Abstract
Ameloblastic fibro-odontosarcoma (AFOS) now designated as odontogenic sarcoma is an extremely rare odontogenic tumor, which histologically presents as a biphasic neoplasm with a malignant mesenchymal component plus ameloblastic epithelium. Here we report a 27-year-old Chinese female with the complaint of a painful swelling for half a month in the right mandible. A segmental mandibulectomy, with an immediate mandibular reconstruction using a free vascularized osteocutaneous fibular flap was performed using surgical guide models. Histological analysis revealed a primary odontogenic sarcoma. The postoperative period was uneventful, and no clinical indication of recurrence or metastasis was observed during the 3-year follow-up. No adjuvant therapy was proposed. This is the first odontogenic sarcoma case reported in China after the new World Health Organization classification of odontogenic lesions.
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Affiliation(s)
- Qinghai Zhu
- Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, Jiangsu 210029, China
- Depatment of Oral and Maxillofacial Surgery, the Affiliated Stomatological Hospital of Nanjing Medical University, Nanjing, Jiangsu 210029, China
| | - Chenxing Wang
- Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, Jiangsu 210029, China
- Depatment of Oral and Maxillofacial Surgery, the Affiliated Stomatological Hospital of Nanjing Medical University, Nanjing, Jiangsu 210029, China
| | - Yizhou Wang
- Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, Jiangsu 210029, China
- Depatment of Oral and Maxillofacial Surgery, the Affiliated Stomatological Hospital of Nanjing Medical University, Nanjing, Jiangsu 210029, China
| | - Songsong Guo
- Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, Jiangsu 210029, China
- Depatment of Oral and Maxillofacial Surgery, the Affiliated Stomatological Hospital of Nanjing Medical University, Nanjing, Jiangsu 210029, China
| | - Jinhai Ye
- Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, Jiangsu 210029, China
- Depatment of Oral and Maxillofacial Surgery, the Affiliated Stomatological Hospital of Nanjing Medical University, Nanjing, Jiangsu 210029, China
- Jinhai Ye, Department of Oral and Maxillofacial Surgery, the Affiliated Stomatological Hospital of Nanjing Medical University, 136 Hanzhong Road, Gulou District, Nanjing, Jiangsu 210029, China. Tel: +86-25-69593380, E-mail:
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Sánchez-Romero C, Paes de Almeida O, Bologna-Molina R. Mixed odontogenic tumors: A review of the clinicopathological and molecular features and changes in the WHO classification. World J Clin Oncol 2021; 12:1227-1243. [PMID: 35070741 PMCID: PMC8716991 DOI: 10.5306/wjco.v12.i12.1227] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/19/2021] [Revised: 05/25/2021] [Accepted: 11/25/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Ameloblastic fibromas and ameloblastic fibrosarcomas are rare odontogenic tumors, and controversy exists in the classification of cases presenting hard-tissue production: Ameloblastic fibrodentinoma (AFD) and ameloblastic fibro-odontoma (AFO). These cases are currently considered “developing odontomas” (hamartomatous lesions).
AIM To analyze the clinicopathologic features of these lesions and discuss the changes in the 2017 World Health Organization classification.
METHODS An electronic literature search was performed in the PubMed/MEDLINE database. An electronic search of the English language literature was performed and last updated in September 2020 in the PubMed/MEDLINE database using the following terms: “ameloblastic fibroma”, “ameloblastic fibrodentinoma”, “ameloblastic fibro-odontoma”, “ameloblastic sarcoma”, “ameloblastic fibrosarcoma”, “ameloblastic fibrodentinosarcoma”, “ameloblastic fibroodontosarcoma” and “odontogenic carcinosarcoma”. The inclusion criteria were odontogenic tumor series, case reports and systematic reviews that provided sufficient clinical, radiological and microscopic documentation to confirm the diagnosis.
RESULTS The database search strategy resulted in 947 papers. Articles focusing on other topics, articles that were not in English, duplicate articles, and articles without fulfilling the inclusion criteria were excluded. Finally, 96 publications were included in this review to describe and discuss the main features of the searched entities. Several aspects of AFO and AFD, such as biological behavior, age of occurrence, amount of hard tissue, and potential for malignant transformation into odontogenic sarcomas, support the neoplastic nature in most of the reported cases. Considering the clinical, radiographic, histopathological and molecular characteristics of odontogenic lesions with hard tissue production, we suggest that these types of lesions should continue to be recognized as odontogenic tumors by maintaining the classically used terms.
CONCLUSION This recommendation will be relevant for future clinical, microscopic, and molecular studies to better understand the biology of these interesting odontogenic tumors.
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Affiliation(s)
- Celeste Sánchez-Romero
- Department of Oral Pathology, Faculty of Dentistry, University Juarez of the Durango State, Durango 33106, Mexico
| | - Oslei Paes de Almeida
- Department of Diagnosis, Faculty of Piracicaba, State University of Campinas, São Paulo Brazil, Piracicaba 13414-903, Sao Paulo, Brazil
| | - Ronell Bologna-Molina
- Department of Oral Pathology, Faculty of Dentistry, University Juarez of the Durango State, Durango 33106, Mexico
- Department of Oral and Maxillofacial Pathology, Universidad de la República UDELAR (URUGUAY), Montevideo 16400, Uruguay
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George SL, Izquierdo E, Campbell J, Koutroumanidou E, Proszek P, Jamal S, Hughes D, Yuan L, Marshall LV, Carceller F, Chisholm JC, Vaidya S, Mandeville H, Angelini P, Wasti A, Bexelius T, Thway K, Gatz SA, Clarke M, Al-Lazikani B, Barone G, Anderson J, Tweddle DA, Gonzalez D, Walker BA, Barton J, Depani S, Eze J, Ahmed SW, Moreno L, Pearson A, Shipley J, Jones C, Hargrave D, Jacques TS, Hubank M, Chesler L. A tailored molecular profiling programme for children with cancer to identify clinically actionable genetic alterations. Eur J Cancer 2019; 121:224-235. [PMID: 31543384 PMCID: PMC6839402 DOI: 10.1016/j.ejca.2019.07.027] [Citation(s) in RCA: 48] [Impact Index Per Article: 8.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/18/2019] [Revised: 06/27/2019] [Accepted: 07/23/2019] [Indexed: 11/10/2022]
Abstract
BACKGROUND For children with cancer, the clinical integration of precision medicine to enable predictive biomarker-based therapeutic stratification is urgently needed. METHODS We have developed a hybrid-capture next-generation sequencing (NGS) panel, specifically designed to detect genetic alterations in paediatric solid tumours, which gives reliable results from as little as 50 ng of DNA extracted from formalin-fixed paraffin-embedded (FFPE) tissue. In this study, we offered an NGS panel, with clinical reporting via a molecular tumour board for children with solid tumours. Furthermore, for a cohort of 12 patients, we used a circulating tumour DNA (ctDNA)-specific panel to sequence ctDNA from matched plasma samples and compared plasma and tumour findings. RESULTS A total of 255 samples were submitted from 223 patients for the NGS panel. Using FFPE tissue, 82% of all submitted samples passed quality control for clinical reporting. At least one genetic alteration was detected in 70% of sequenced samples. The overall detection rate of clinically actionable alterations, defined by modified OncoKB criteria, for all sequenced samples was 51%. A total of 8 patients were sequenced at different stages of treatment. In 6 of these, there were differences in the genetic alterations detected between time points. Sequencing of matched ctDNA in a cohort of extracranial paediatric solid tumours also identified a high detection rate of somatic alterations in plasma. CONCLUSION We demonstrate that tailored clinical molecular profiling of both tumour DNA and plasma-derived ctDNA is feasible for children with solid tumours. Furthermore, we show that a targeted NGS panel-based approach can identify actionable genetic alterations in a high proportion of patients.
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Affiliation(s)
- Sally L George
- Paediatric Tumour Biology, Division of Clinical Studies, The Institute of Cancer Research, London, UK; Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK.
| | - Elisa Izquierdo
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK; Glioma Team, Division of Molecular Pathology and Cancer Therapeutics, The Institute of Cancer Research, London, UK
| | - James Campbell
- Bioinformatics Core Facility, The Institute of Cancer Research, London, UK
| | - Eleni Koutroumanidou
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK
| | - Paula Proszek
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK
| | - Sabri Jamal
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK
| | - Deborah Hughes
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK
| | - Lina Yuan
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK
| | - Lynley V Marshall
- Paediatric Tumour Biology, Division of Clinical Studies, The Institute of Cancer Research, London, UK; Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Fernando Carceller
- Paediatric Tumour Biology, Division of Clinical Studies, The Institute of Cancer Research, London, UK; Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Julia C Chisholm
- Paediatric Tumour Biology, Division of Clinical Studies, The Institute of Cancer Research, London, UK; Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Sucheta Vaidya
- Paediatric Tumour Biology, Division of Clinical Studies, The Institute of Cancer Research, London, UK; Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Henry Mandeville
- Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Paola Angelini
- Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Ajla Wasti
- Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Tomas Bexelius
- Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Khin Thway
- Pathology Department, Royal Marsden NHS Foundation Trust, London, UK
| | - Susanne A Gatz
- Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK; Sarcoma Molecular Pathology Team, Divisions of Molecular Pathology and Cancer Therapeutics, The Institute of Cancer Research, London, UK; Cancer Research UK Clinical Trials Unit, Institute of Cancer and Genomic Sciences, University of Birmingham, Birmingham, UK
| | - Matthew Clarke
- Glioma Team, Division of Molecular Pathology and Cancer Therapeutics, The Institute of Cancer Research, London, UK
| | - Bissan Al-Lazikani
- Bioinformatics Core Facility, The Institute of Cancer Research, London, UK
| | - Giuseppe Barone
- Department of Haematology and Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK
| | - John Anderson
- Department of Haematology and Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK; Developmental Biology and Cancer Programme, UCL GOS Institute of Child Health, London, UK
| | - Deborah A Tweddle
- Northern Institute for Cancer Research, Newcastle University, Newcastle, UK
| | - David Gonzalez
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK; Centre for Cancer Research and Cell Biology, Queens University Belfast, Belfast, UK
| | - Brian A Walker
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK; Myeloma Center, University of Arkansas for Medical Sciences, Little Rock, AR, USA
| | - Jack Barton
- Developmental Biology and Cancer Programme, UCL GOS Institute of Child Health, London, UK
| | - Sarita Depani
- Department of Haematology and Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK
| | - Jessica Eze
- Department of Haematology and Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK; Department of Histology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK
| | - Saira W Ahmed
- Department of Haematology and Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK; Department of Histology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK
| | - Lucas Moreno
- Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK; HNJ-CNIO Clinical Research Unit, Hospital Universitario Nino Jesus, Madrid, Spain; Paediatric Oncology & Haematology, Vall d'Hebron University Hospital, Barcelona, Spain
| | - Andrew Pearson
- Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
| | - Janet Shipley
- Sarcoma Molecular Pathology Team, Divisions of Molecular Pathology and Cancer Therapeutics, The Institute of Cancer Research, London, UK
| | - Chris Jones
- Glioma Team, Division of Molecular Pathology and Cancer Therapeutics, The Institute of Cancer Research, London, UK
| | - Darren Hargrave
- Cancer Research UK Clinical Trials Unit, Institute of Cancer and Genomic Sciences, University of Birmingham, Birmingham, UK; Department of Haematology and Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK
| | - Thomas S Jacques
- Department of Haematology and Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK; Department of Histology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK
| | - Michael Hubank
- Molecular Diagnostics Department, The Institute of Cancer Research and Clinical Genomics, The Royal Marsden NHS Foundation, London, UK
| | - Louis Chesler
- Paediatric Tumour Biology, Division of Clinical Studies, The Institute of Cancer Research, London, UK; Children and Young People's Unit, Royal Marsden NHS Foundation Trust, London, UK
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Chrcanovic B, Gomez R. Ameloblastic fibrodentinosarcoma and ameloblastic fibro-odontosarcoma: A systematic review. JOURNAL OF STOMATOLOGY, ORAL AND MAXILLOFACIAL SURGERY 2018; 119:401-406. [PMID: 29574112 DOI: 10.1016/j.jormas.2018.03.007] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Received: 10/22/2017] [Revised: 01/15/2018] [Accepted: 03/18/2018] [Indexed: 11/25/2022]
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Atarbashi-Moghadam S, Lotfi A, Mokhtari S. A mixed odontogenic sarcoma: A challenging histopathologic case and brief review of the literature. J Oral Maxillofac Pathol 2018; 22:S29-S34. [PMID: 29491601 PMCID: PMC5824513 DOI: 10.4103/jomfp.jomfp_74_17] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/09/2017] [Accepted: 12/21/2017] [Indexed: 01/27/2023] Open
Abstract
Ameloblastic fibro-odontosarcoma (AFOS) is an extremely rare malignant mixed odontogenic tumor. The ectomesenchymal part of the neoplasm shows malignancy, whereas the epithelial component is rather benign. In addition, small areas with deposition of enamel matrix and dentine material are seen. The rarity of this neoplasm and microscopic similarities with other malignant and benign tumors can lead to diagnostic problems. Here, we describe the histopathologic features of a new case of AFOS of the mandible in a 34-year-old female patient. It is essential for oral pathologists to be familiar with the microscopic features of this rare neoplasm to have a proper diagnosis. This is also the first reported case of AFOS that closely resembles osteosarcoma in some areas.
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Affiliation(s)
- Saede Atarbashi-Moghadam
- Department of Oral and Maxillofacial Pathology, Dental School of Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Ali Lotfi
- Department of Oral and Maxillofacial Pathology, Dental School of Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Sepideh Mokhtari
- Education Development Office, School of Dentistry, Tehran University of Medical Sciences, Tehran, Iran
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Niu H, Liu J, Chen Y, Geng N. Ameloblastic fibro-odontosarcoma of the mandible with active epithelial proliferation: A rare case report. Mol Clin Oncol 2017; 7:971-975. [PMID: 29285358 PMCID: PMC5740910 DOI: 10.3892/mco.2017.1448] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/03/2016] [Accepted: 05/03/2017] [Indexed: 02/05/2023] Open
Abstract
Ameloblastic fibro-odontosarcoma (AFOS) is an extremely rare subtype of odontogenic sarcoma, with no more than 19 cases reported in the English literature to date. AFOS is a biphasic neoplasm, with deposits of dentin and enamel matrix. We herein present a case of AFOS with active epithelial proliferation in a 31-year-old female patient. The patient was referred to the West China Hospital of Stomatology (Chengdu, China) due to a 6-month history of a swelling in the left mandible. Following clinical and radiological examination, the initial preoperative diagnosis was ameloblastoma, with local invasion and the possibility of malignant transformation. Left hemimandibular resection was subsequently performed. The postoperative histopathological diagnosis was AFOS, accompanied by active epithelial proliferation. Immunohistochemically, cytokeratin (CK)14 and CK19 were intensely positive in the epithelium, whereas the mesenchymal cells were strongly positive for vimentin. The Ki-67 labeling index was considerably higher in the mesenchymal component (mean, 40%) compared with that in the epithelial element (mean, 5–8%). Three months after the surgical procedure, the patient remained clinically and radiologically disease-free.
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Affiliation(s)
- Haoman Niu
- State Key Laboratory of Oral Disease, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan 610041, P.R. China
| | - Junyu Liu
- State Key Laboratory of Oral Disease, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan 610041, P.R. China
| | - Yu Chen
- Department of Oral Pathology, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan 610041, P.R. China
| | - Ning Geng
- Department of Oral Pathology, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan 610041, P.R. China
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Augello M, Rabufetti A, Ghazal G, Yurtsever H, Leiggener C. Ameloblastic fibro-odontoma in children. Clinical aspects and review of the literature. ORAL AND MAXILLOFACIAL SURGERY CASES 2017. [DOI: 10.1016/j.omsc.2017.01.001] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/14/2022] Open
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Chen SJ, Zheng XW, Lin X, Liu H. Ameloblastic fibro-odontosarcoma of the mandible in a pediatric patient. Eur Ann Otorhinolaryngol Head Neck Dis 2016; 133:419-421. [PMID: 27130809 DOI: 10.1016/j.anorl.2015.11.010] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/07/2015] [Revised: 11/26/2015] [Accepted: 11/30/2015] [Indexed: 12/27/2022]
Abstract
INTRODUCTION Ameloblastic fibro-odontosarcoma is an extremely rare subtype of odontogenic sarcoma, with only 13 cases reported in the literature. CASE REPORT A 4-year-old male presented with a painless mandibular swelling, which appeared 4months previously. Cone beam computed tomography revealed an extensive, ill-circumscribed, multilocular radiolucency of the right mandible extending from the first deciduous molar to the ramus with slightly dense opacities. Histological examination of the incisional biopsy specimen revealed a biphasic tumor with sarcomatous mesenchyme and benign ameloblastic epithelial component compatible with a diagnosis of ameloblastic fibrosarcoma. A right hemimandibular resection was performed. Areas of deposition of dentinoid and enamel material closely adjacent to ameloblastic epithelium were noted in the excised specimen. A final diagnosis of ameloblastic fibro-odontosarcoma was made. After four years of close follow-up, there is no sign of recurrence or metastasis. CONCLUSION Although rare, ameloblastic fibro-odontosarcoma should be considered in the differential diagnosis of jaw lesions with radiographic radiolucencies exhibiting poorly circumscribed outlines and containing radiopaque material. Definite diagnosis depends on histopathological examination. Complete surgical excision is the treatment of choice.
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Affiliation(s)
- S-J Chen
- Department of Head and Neck Surgery, Tumor Hospital of Fujian Province, Fujian Medical University, Fuzhou, Fujian, PR China
| | - X-W Zheng
- Department of Pathology, Tumor Hospital of Fujian Province, Fujian Medical University, Fuzhou, Fujian, PR China
| | - X Lin
- Department of Head and Neck Surgery, Tumor Hospital of Fujian Province, Fujian Medical University, Fuzhou, Fujian, PR China
| | - H Liu
- Department of Head and Neck Surgery, Tumor Hospital of Fujian Province, Fujian Medical University, Fuzhou, Fujian, PR China.
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Kirjavainen A, Tuovinen V, Sándor GK. Large ameloblastic fibro-odontoma in a 7-year-old girl with analysis of 108 cases. Ann Maxillofac Surg 2016; 6:15-20. [PMID: 27563600 PMCID: PMC4979334 DOI: 10.4103/2231-0746.186132] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022] Open
Abstract
BACKGROUND Odontogenic tumors such as ameloblastic fibro-odontoma (AFO) are rare conditions in children and are often asymptomatic. AFOs are found by routine clinical and radiological examination or when they cause obvious intra- or extra-oral swelling. MATERIALS AND METHODS A case of an AFO in a 7-year-old girl is described, and 107 cases from the literature and this report are analyzed. RESULTS The total of 108 cases revealed the average age at presentation of AFO to be 6.3 years in boys and 9.6 years in girls. There was a slight male predilection and AFO lesions most often occurred in the posterior mandible. AFO was almost always associated with an unerupted tooth or teeth. CONCLUSIONS While the recurrence rate of AFO was found to be 5.5%, long-term postoperative clinical and radiological follow-up is advised to ensure no future signs of aggressive recurrence.
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Affiliation(s)
- Antti Kirjavainen
- Department of Oral and Maxillofacial Surgery, Jyväskylä Central Hospital, Jyväskylä, Finland
| | - Veikko Tuovinen
- Department of Oral and Maxillofacial Surgery, Jyväskylä Central Hospital, Jyväskylä, Finland
| | - George K. Sándor
- Department of Oral and Maxillofacial Surgery, Medical Research Center, Oulu University Hospital, Institute of Dentistry, University of Oulu, Oulu
- BioMediTech, Institute of Bioscience and Technology, University of Tampere, Tampere, Finland
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