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Carbon ion therapy for ameloblastic carcinoma

Carbon ion therapy for ameloblastic carcinoma Ameloblastic carcinomas are rare odontogenic tumors. Treatment usually consists of surgical resection and sometimes adjuvant radiation. We report the case of a 71 year-old male patient undergoing carbon ion therapy for extensive local relapse of ameloblastic carcinoma. Treatment outcome was favourable with a complete remission at 6 weeks post completion of radiotherapy while RT-treatment itself was tolerated well with only mild side effects. High dose radiation hence is a potential alternative for patients unfit or unwilling to undergo extensive surgery or in cases when only a subtotal resection is planned or the resection is mutilating. Introduction presentation in our department with the patient com- Ameloblastic carcinomas are very rare odontogenic plaining of increasing pain in the left maxilla and probe tumors sometimes referred to as malignant ameloblas- excision confirming the diagnosis of locally recurrent toma [1]. To date, 31 cases have been reported world- ameloblastic carcinoma of the maxilla (Figures 1 and 2). wide, with the largest series describing 14 cases [2] As the referring surgeon doubted successful removal of Definitions and classifications of ameloblastic carcino- the extensive tumor mass, faced with the inherent mor- mas have changed over the years, there have been var- bidity, salvage surgery did not seem a feasible option. ious classifications, the latest by Slootweg and Müller Pre-therapeutic staging did not show signs of distant [3] emphasizing histogenesis of the tumor leading to the spread and the patient was referred to radiotherapy. new WHO classification in 2005 [4]. Slootweg and Mül- Interdisciplinary discussion recommended high-dose ler defined ameloblastic carcinoma as a tumor combin- carbon ion therapy for this patient, hence he received a ing morphologic features of both ameloblastoma and dose of 60 GyE carbon ions. carcinoma, which can arise de novo, ex ameloblastoma, or ex odontogenic cyst [3]. Radiotherapy Most of the reported cases were discovered in the Immobilization/planning examinations mandible, only one fifth occur in the maxilla [5,6]. Immobilization consisted of an individual thermoplastic Though they claimed to exhibit a tendency towards head mask with thermoplastic shoulder fixation. Plan- aggressive local growth and local relapse [1], distant ning examinations consisted of a planning CT scan metastases are uncommon. Being a rare disease, there (3 mm slice thickness) with the patient positioned in the are no treatment guidelines. However, standard treat- individual fixation device and contrast-enhanced MRI ment has been complete surgical resection in reported for 3 D image correlation. cases [5,7]. Target volumes/dose prescription Report of a case CTV1 included the macroscopic tumor. PTV1 consisted A 71-year-old patient with extensive relapse of an ame- of a 3 mm margin around the CTV1 but did not extend loblastic carcinoma was referred to our institution for into critical organs at risk (i.e. brain stem, spinal cord). carbon ion therapy by his maxillo-facial surgeon. He CTV2 included CTV1 and all involved paranasal had been initially diagnosed 10 years ago when the sinuses. As there were no suspect lymph nodes, elective tumor was completely resected. Local recurrence was nodal irradiation was not performed given the rarity of found on a follow-up MRI scan 6 months prior to nodal metastases. We prescribed a dose of 44,8 GyE carbon ions in 2,99 * Correspondence: alexandra.jensen@med.uni-heidelberg.de GyE/fraction (5 fractions per week) to the CTV2, Dept of Radiation Oncology, INF 400, 69120 Heidelberg, Germany followed by a boost to CTV1 with 14,9 GyE at 2,98 Full list of author information is available at the end of the article © 2011 Jensen et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Jensen et al. Radiation Oncology 2011, 6:13 Page 2 of 5 http://www.ro-journal.com/content/6/1/13 Figure 1 Extensive ameloblastic carcinoma originating from Figure 3 Carbon ion dose distribution: (summation: primary the left maxilla: axial, contrast-enhanced T1 weighted MRI. plan to 45 GyE C12, boost plan to 15 GyE) 100% corresponding to 60 GyE, axial view. Orange outline: CTV2; red outline: CTV 1; Green star: local maximum dose within the slice. Red GyE/fraction. aiming at covering the CTV with the 95% star: global maximum. prescription isodose. Figures 3, 4, and 5 show axial, cor- onal, and sagittal views of the summation dose distribu- tion (voxel-by-voxel addition of basic and boost plan) carbon ion beam with a full-width/half-maximum with 100% corresponding to 60.0 GyE. Figure 6 depicts (FWHM) of 5 mm is extracted from the accelerator sys- the dose-volume-histogram (DVH). Treatment was tem (synchrotron) and magnetically deflected to subse- given at the HIT (Heidelberg Ion-Beam Therapy Centre) quently scan all planned iso-energetic slices roughly after inverse treatment planning in active beam applica- corresponding to the tumour’s radiological depth. Using tion (raster-scanning method) [8]. A monoenergetic this method almost any desired dose distribution can be Figure 4 Carbon ion dose distribution: (summation: primary plan to 45 GyE C12, boost plan to 15 GyE) 100% corresponding to 60 GyE, coronal view. Orange outline: CTV2; red Figure 2 Extensive ameloblastic carcinoma originating from outline: CTV 1; Green star: local maximum dose within the slice. Red the left maxilla: coronal, contrast-enhanced T1 weighted MRI. star: global maximum. Jensen et al. Radiation Oncology 2011, 6:13 Page 3 of 5 http://www.ro-journal.com/content/6/1/13 and shifts were always corrected using a robotic table allowing position correction in six degrees of freedom. Treatment schedule/follow-up Treatment was carried out in 5 fractions per week hence over approximately 4 weeks. The first follow-up examination including clinical examination and diagnostic, contrast-enhanced MRI was carried out 6 weeks post completion of radiation treat- ment, the second follow-up 3 months thereafter. Further follow-up radiooncological follow-up appointments are scheduled in 6-monthly intervals. The patient was also encouraged to undergo regular check-ups including full ENT clinical examinations in regular intervals (usually every 6 weeks). Treatment outcome Figure 5 Carbon ion dose distribution: (summation: primary Treatment was tolerated well. Only very mild skin plan to 45 GyE C12, boost plan to 15 GyE) 100% changes (hyperpigmentation CTC°I and erythema CTC° corresponding to 60 GyE, sagittal view. Orange outline: CTV2; red I, no desquamation) over the left cheek and mild outline: CTV 1; Green star: local maximum dose within the slice. Red mucous membrane reactions (mucositic CTC °I-II) at star: global maximum. the soft/hard left palate could be observed. No change of taste, dysphagia, or weight loss was observed. There created and dose to surrounding critical structures can wasonlyaverymildxerostomia(CTC°I).Whenthe be minimized. patient presented to our department again 6 weeks post Daily image guidance consisted of orthogonal x-ray completion of radiotherapy, he was in a very good clini- controls in treatment position with the x-ray tube/ cal state, the pain in the right maxilla was already resol- receptor mounted on a robot to allow imaging in almost ving steadily. While the patient was on oral morphin any treatment table position. After acquisition of ortho- sulfates during treatment, only needed non-steroidal gonal x-rays, an automatic 2D-3 D pre-match was car- anti-inflammatories occasionally on follow-up. No resi- ried out (Siemens syngo PT treatment) and verified by dual acute radiogenic reactions could be detected other the radiotherapist/radiation oncologist with regard to than residual xerostomia (CTC°I). Furthermore, no resi- bony anatomy. Manual adjustment of the match was dual tumor could be detected on his follow-up MRI- carried out on-line and the resulting correction vector, scans 6 weeks post completion of therapy (Figures 7 and including rotations, subsequently applied to the patient 8) and 3 months thereafter. position. Patient position was controlled in each session Discussion With the disease being extremely rare, clinicians have to rely on various reported cases for guidance. Hence, the establishment of treatment standards is not possible. Radiotherapy has been controversially discussed in the past. Most reported cases underwent surgical removal. Reports of ameloblastic carcinomas receiving radiation therapy are scarce and mostly from the pre-3 D and cobalt era [9-11]. To our knowledge, radiotherapy has only been given as adjuvant therapy in only a few cases [11-15] within the past 20 years. Radiation doses between 41,4 Gy and 54 Gy have been comparatively conservative [12,13,16] or not been reported [2,14,17] leading to local relapse in half of the cases. Higher radiation doses between 66 and 72 Gy in close margin/ positive-margin resections as reported by Philip et al [15] lead to local control for the duration of available Figure 6 Carbon ion summation plan: DVH. follow-up (0.8 - 3.3 years) in the reported 3 cases. Jensen et al. Radiation Oncology 2011, 6:13 Page 4 of 5 http://www.ro-journal.com/content/6/1/13 margins [1]) is warranted to counterbalance high ten- dency of local relapse, RT was given in a high-precision technique as carbon ion therapy. Carbon ion therapy in active beam application with raster-scanned particle beams is able to produce extremely steep gradients hence delivering high doses to the tumor while sparing normal surrounding tissues. In contrast to intensity- modulated radiation therapy, integral dose to the irra- diated volume is substantially lower. Also, increased bio- logical effectiveness of carbon ion beams has been shown to be beneficial in other radioresistant tumors [18-20]. While it is beyond the scope of this case report to establish a clinical standard, our case shows that fast complete remissions of extensive ameloblastic carcino- mas are possible using carbon ion therapy at substantial doses. Moreover, this treatment is accompanied by very mild treatment-related side effects (erythema, xerosto- mia CTC°I and mucositis CTC °I-II) and no major radiation-related toxicity; hence the patient could be spared extensive, mutilating and potentially incomplete surgical procedures. Figure 7 1st folow-up 6 weeks post completion of RT: complete remission with only posttherapeutic changes: axial, To our knowledge, this is the first case of ameloblastic contrast enhanced T1 weighted MRI. carcinoma being treated with carbon ion therapy and resulted so far in an excellent posttherapeutic outcome. Therefore radiotherapy with carbon ions should be con- While it has been discussed in cases with incomplete sidered in the definitive treatment of these rare tumors. resections or nodal metastases, there is no evidence for radiotherapy as a potentially definitive treatment modal- Conflicts of interest ity yet. Faced with the opinion that aggressive treatment The authors declare that they have no competing interests. (recommending surgical wide excision with 2-3 cm Author details 1 2 Dept of Radiation Oncology, INF 400, 69120 Heidelberg, Germany. Dept. of Medical Physics, Heidelberg Ion Therapy Centre (HIT), INF 450, 69120 Heidelberg, Germany. Authors’ contributions All authors read and approved the final manuscript Received: 22 October 2010 Accepted: 6 February 2011 Published: 6 February 2011 References 1. Avon SL, McComb J, Clokie C: Ameloblastic carcinoma: case report and literature review. J Can Dent Assoc 2003, 69:573-576. 2. Hall JM, Weathers DR, Unni KK: Ameloblastic carcinoma: an analysis of 14 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007, 103:799-807. 3. Slootweg PJ, Müller H: Malignant ameloblstoma or ameloblastic carcinoma. Oral Surg Oral Med Oral Pathol 1984, 57:168-176. 4. Barnes L, Everson JW, Reichart P, editors: Pathology and Genetics: Head and neck tumors. Lyon, World Health Organization 2005, 287-289. 5. Kruse ALD, Zwahlen RA, Grätz KW: New classification of maxillary ameloblastic carcinoma based on an evidence-based literature reviwe over the last 60 years. Head Neck Oncol 2009, 1:31. 6. Lolachi CM, Mada SK, Jacobs JR: Ameloblastic carcinoma of the maxilla. J Laryngol Otol 1995, 109:1019-1022. 7. Lucca M, D’Innocenzo R, Kraus JA, Gagari E, Hall J, Shastri K: Ameloblastic Figure 8 1st folow-up 6 weeks post completion of RT: carcinoma of the maxilla: a report of 2 cases. J Oral Maxillofac Surg 2010. complete remission with only posttherapeutic changes: 8. Haberer T, Becher W, Schardt D, Kraft G: Magnetic scanning system for coronal, contrast enhanced T1. heavy ion therapy. Nucl Instr Meth Phys Res 1993, 330:296-305. Jensen et al. Radiation Oncology 2011, 6:13 Page 5 of 5 http://www.ro-journal.com/content/6/1/13 9. Daramola JO, Abioye AA, Ajagbe HA, Aghadiuno PU: Maxillary malignant ameloblastoma with intraorbital extension: report of a case. J Oral Surg 1980, 38:203-206. 10. Lee L, Maxymiw WG, Wood RE: Ameloblastic carcinoma of the maxilla metastatic to the mandible. Case report. J Craniomaxillofac Surg 1990, 18:247-250. 11. Miyamoto CT, Brady LW, Markoe A, Salinger D: Ameloblastoma of the jaw. Treatment with radiation therapy and a case report. Am J Clin Oncol 1991, 14:225-230. 12. Infante-Cossio P, Hernandez-Guisado JM, Fernandez-Machin P, Garcia- Perla A, Guitierrez-Perez JL: Ameloblastic carcinoma of the maxilla: report of 3 cases. J Cranio Maxillofac Surg 1998, 26:159-162. 13. Zwahlen RA, Vogt P, Fischer FS, Grätz KW: Case report: myocardial metastasis of a maxillary malignant ameloblastoma. J Oral Maxillofac Surg 2003, 61:731-734. 14. Goldenberg D, Sciubba J, Koch W, Tufano RP: Malignant odontogenic tumors: a 22-year experience. Laryngoscope 2004, 114:1770-1774. 15. Philip M, Morris CG, Werning JW, Mendenhall WM: Radiotherapy in the treatment of ameloblastic carcinoma. J HK Coll Radiol 2005, 8:157-161. 16. Simko EJ, Brannon RB, Eibling DE: Ameloblastic carcinoma of the mandible. Head Neck 1998, 20:654-659. 17. Dhir K, Sciubba J, Tufano RP: Ameloblastic carcinoma of the maxilla. Oral Oncol 2003, 39:736-741. 18. Schulz-Ertner D, Nikoghosyan A, Hof H, Didinger B, Combs SE, Jäkel O, Karger CP, Edler L, Debus J: Carbon ion therapy of skull base chondrosarcomas. Int J Radiat Oncol Biol Phys 2007, 67:171-177. 19. Schulz-Ertner D, Karger CP, Feuerhake A, Nikoghosyan A, Combs SE, Jäkel O, Edler L, Scholz M, Debus J: Effectiveness of carbon ion radiotherapy in the treatment of skull-base chordomas. Int J Radiat Oncol Biol Phys 2007, 68:449-457. 20. Schulz-Ertner D, Nikoghosyan A, Didinger B, Münter M, Jäkel O, Karger CP, Debus J: Therapy strategies for locally advanced adenoid-cystic carcinomas using modern radiation therapy techniques. Cancer 2005, 104:338-344. doi:10.1186/1748-717X-6-13 Cite this article as: Jensen et al.: Carbon ion therapy for ameloblastic carcinoma. Radiation Oncology 2011 6:13. Submit your next manuscript to BioMed Central and take full advantage of: • Convenient online submission • Thorough peer review • No space constraints or color figure charges • Immediate publication on acceptance • Inclusion in PubMed, CAS, Scopus and Google Scholar • Research which is freely available for redistribution Submit your manuscript at www.biomedcentral.com/submit http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Radiation Oncology Springer Journals

Carbon ion therapy for ameloblastic carcinoma

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Springer Journals
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Copyright © 2011 by Jensen et al; licensee BioMed Central Ltd.
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Medicine & Public Health; Oncology; Radiotherapy
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Abstract

Ameloblastic carcinomas are rare odontogenic tumors. Treatment usually consists of surgical resection and sometimes adjuvant radiation. We report the case of a 71 year-old male patient undergoing carbon ion therapy for extensive local relapse of ameloblastic carcinoma. Treatment outcome was favourable with a complete remission at 6 weeks post completion of radiotherapy while RT-treatment itself was tolerated well with only mild side effects. High dose radiation hence is a potential alternative for patients unfit or unwilling to undergo extensive surgery or in cases when only a subtotal resection is planned or the resection is mutilating. Introduction presentation in our department with the patient com- Ameloblastic carcinomas are very rare odontogenic plaining of increasing pain in the left maxilla and probe tumors sometimes referred to as malignant ameloblas- excision confirming the diagnosis of locally recurrent toma [1]. To date, 31 cases have been reported world- ameloblastic carcinoma of the maxilla (Figures 1 and 2). wide, with the largest series describing 14 cases [2] As the referring surgeon doubted successful removal of Definitions and classifications of ameloblastic carcino- the extensive tumor mass, faced with the inherent mor- mas have changed over the years, there have been var- bidity, salvage surgery did not seem a feasible option. ious classifications, the latest by Slootweg and Müller Pre-therapeutic staging did not show signs of distant [3] emphasizing histogenesis of the tumor leading to the spread and the patient was referred to radiotherapy. new WHO classification in 2005 [4]. Slootweg and Mül- Interdisciplinary discussion recommended high-dose ler defined ameloblastic carcinoma as a tumor combin- carbon ion therapy for this patient, hence he received a ing morphologic features of both ameloblastoma and dose of 60 GyE carbon ions. carcinoma, which can arise de novo, ex ameloblastoma, or ex odontogenic cyst [3]. Radiotherapy Most of the reported cases were discovered in the Immobilization/planning examinations mandible, only one fifth occur in the maxilla [5,6]. Immobilization consisted of an individual thermoplastic Though they claimed to exhibit a tendency towards head mask with thermoplastic shoulder fixation. Plan- aggressive local growth and local relapse [1], distant ning examinations consisted of a planning CT scan metastases are uncommon. Being a rare disease, there (3 mm slice thickness) with the patient positioned in the are no treatment guidelines. However, standard treat- individual fixation device and contrast-enhanced MRI ment has been complete surgical resection in reported for 3 D image correlation. cases [5,7]. Target volumes/dose prescription Report of a case CTV1 included the macroscopic tumor. PTV1 consisted A 71-year-old patient with extensive relapse of an ame- of a 3 mm margin around the CTV1 but did not extend loblastic carcinoma was referred to our institution for into critical organs at risk (i.e. brain stem, spinal cord). carbon ion therapy by his maxillo-facial surgeon. He CTV2 included CTV1 and all involved paranasal had been initially diagnosed 10 years ago when the sinuses. As there were no suspect lymph nodes, elective tumor was completely resected. Local recurrence was nodal irradiation was not performed given the rarity of found on a follow-up MRI scan 6 months prior to nodal metastases. We prescribed a dose of 44,8 GyE carbon ions in 2,99 * Correspondence: alexandra.jensen@med.uni-heidelberg.de GyE/fraction (5 fractions per week) to the CTV2, Dept of Radiation Oncology, INF 400, 69120 Heidelberg, Germany followed by a boost to CTV1 with 14,9 GyE at 2,98 Full list of author information is available at the end of the article © 2011 Jensen et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Jensen et al. Radiation Oncology 2011, 6:13 Page 2 of 5 http://www.ro-journal.com/content/6/1/13 Figure 1 Extensive ameloblastic carcinoma originating from Figure 3 Carbon ion dose distribution: (summation: primary the left maxilla: axial, contrast-enhanced T1 weighted MRI. plan to 45 GyE C12, boost plan to 15 GyE) 100% corresponding to 60 GyE, axial view. Orange outline: CTV2; red outline: CTV 1; Green star: local maximum dose within the slice. Red GyE/fraction. aiming at covering the CTV with the 95% star: global maximum. prescription isodose. Figures 3, 4, and 5 show axial, cor- onal, and sagittal views of the summation dose distribu- tion (voxel-by-voxel addition of basic and boost plan) carbon ion beam with a full-width/half-maximum with 100% corresponding to 60.0 GyE. Figure 6 depicts (FWHM) of 5 mm is extracted from the accelerator sys- the dose-volume-histogram (DVH). Treatment was tem (synchrotron) and magnetically deflected to subse- given at the HIT (Heidelberg Ion-Beam Therapy Centre) quently scan all planned iso-energetic slices roughly after inverse treatment planning in active beam applica- corresponding to the tumour’s radiological depth. Using tion (raster-scanning method) [8]. A monoenergetic this method almost any desired dose distribution can be Figure 4 Carbon ion dose distribution: (summation: primary plan to 45 GyE C12, boost plan to 15 GyE) 100% corresponding to 60 GyE, coronal view. Orange outline: CTV2; red Figure 2 Extensive ameloblastic carcinoma originating from outline: CTV 1; Green star: local maximum dose within the slice. Red the left maxilla: coronal, contrast-enhanced T1 weighted MRI. star: global maximum. Jensen et al. Radiation Oncology 2011, 6:13 Page 3 of 5 http://www.ro-journal.com/content/6/1/13 and shifts were always corrected using a robotic table allowing position correction in six degrees of freedom. Treatment schedule/follow-up Treatment was carried out in 5 fractions per week hence over approximately 4 weeks. The first follow-up examination including clinical examination and diagnostic, contrast-enhanced MRI was carried out 6 weeks post completion of radiation treat- ment, the second follow-up 3 months thereafter. Further follow-up radiooncological follow-up appointments are scheduled in 6-monthly intervals. The patient was also encouraged to undergo regular check-ups including full ENT clinical examinations in regular intervals (usually every 6 weeks). Treatment outcome Figure 5 Carbon ion dose distribution: (summation: primary Treatment was tolerated well. Only very mild skin plan to 45 GyE C12, boost plan to 15 GyE) 100% changes (hyperpigmentation CTC°I and erythema CTC° corresponding to 60 GyE, sagittal view. Orange outline: CTV2; red I, no desquamation) over the left cheek and mild outline: CTV 1; Green star: local maximum dose within the slice. Red mucous membrane reactions (mucositic CTC °I-II) at star: global maximum. the soft/hard left palate could be observed. No change of taste, dysphagia, or weight loss was observed. There created and dose to surrounding critical structures can wasonlyaverymildxerostomia(CTC°I).Whenthe be minimized. patient presented to our department again 6 weeks post Daily image guidance consisted of orthogonal x-ray completion of radiotherapy, he was in a very good clini- controls in treatment position with the x-ray tube/ cal state, the pain in the right maxilla was already resol- receptor mounted on a robot to allow imaging in almost ving steadily. While the patient was on oral morphin any treatment table position. After acquisition of ortho- sulfates during treatment, only needed non-steroidal gonal x-rays, an automatic 2D-3 D pre-match was car- anti-inflammatories occasionally on follow-up. No resi- ried out (Siemens syngo PT treatment) and verified by dual acute radiogenic reactions could be detected other the radiotherapist/radiation oncologist with regard to than residual xerostomia (CTC°I). Furthermore, no resi- bony anatomy. Manual adjustment of the match was dual tumor could be detected on his follow-up MRI- carried out on-line and the resulting correction vector, scans 6 weeks post completion of therapy (Figures 7 and including rotations, subsequently applied to the patient 8) and 3 months thereafter. position. Patient position was controlled in each session Discussion With the disease being extremely rare, clinicians have to rely on various reported cases for guidance. Hence, the establishment of treatment standards is not possible. Radiotherapy has been controversially discussed in the past. Most reported cases underwent surgical removal. Reports of ameloblastic carcinomas receiving radiation therapy are scarce and mostly from the pre-3 D and cobalt era [9-11]. To our knowledge, radiotherapy has only been given as adjuvant therapy in only a few cases [11-15] within the past 20 years. Radiation doses between 41,4 Gy and 54 Gy have been comparatively conservative [12,13,16] or not been reported [2,14,17] leading to local relapse in half of the cases. Higher radiation doses between 66 and 72 Gy in close margin/ positive-margin resections as reported by Philip et al [15] lead to local control for the duration of available Figure 6 Carbon ion summation plan: DVH. follow-up (0.8 - 3.3 years) in the reported 3 cases. Jensen et al. Radiation Oncology 2011, 6:13 Page 4 of 5 http://www.ro-journal.com/content/6/1/13 margins [1]) is warranted to counterbalance high ten- dency of local relapse, RT was given in a high-precision technique as carbon ion therapy. Carbon ion therapy in active beam application with raster-scanned particle beams is able to produce extremely steep gradients hence delivering high doses to the tumor while sparing normal surrounding tissues. In contrast to intensity- modulated radiation therapy, integral dose to the irra- diated volume is substantially lower. Also, increased bio- logical effectiveness of carbon ion beams has been shown to be beneficial in other radioresistant tumors [18-20]. While it is beyond the scope of this case report to establish a clinical standard, our case shows that fast complete remissions of extensive ameloblastic carcino- mas are possible using carbon ion therapy at substantial doses. Moreover, this treatment is accompanied by very mild treatment-related side effects (erythema, xerosto- mia CTC°I and mucositis CTC °I-II) and no major radiation-related toxicity; hence the patient could be spared extensive, mutilating and potentially incomplete surgical procedures. Figure 7 1st folow-up 6 weeks post completion of RT: complete remission with only posttherapeutic changes: axial, To our knowledge, this is the first case of ameloblastic contrast enhanced T1 weighted MRI. carcinoma being treated with carbon ion therapy and resulted so far in an excellent posttherapeutic outcome. Therefore radiotherapy with carbon ions should be con- While it has been discussed in cases with incomplete sidered in the definitive treatment of these rare tumors. resections or nodal metastases, there is no evidence for radiotherapy as a potentially definitive treatment modal- Conflicts of interest ity yet. Faced with the opinion that aggressive treatment The authors declare that they have no competing interests. (recommending surgical wide excision with 2-3 cm Author details 1 2 Dept of Radiation Oncology, INF 400, 69120 Heidelberg, Germany. Dept. of Medical Physics, Heidelberg Ion Therapy Centre (HIT), INF 450, 69120 Heidelberg, Germany. Authors’ contributions All authors read and approved the final manuscript Received: 22 October 2010 Accepted: 6 February 2011 Published: 6 February 2011 References 1. Avon SL, McComb J, Clokie C: Ameloblastic carcinoma: case report and literature review. J Can Dent Assoc 2003, 69:573-576. 2. Hall JM, Weathers DR, Unni KK: Ameloblastic carcinoma: an analysis of 14 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007, 103:799-807. 3. Slootweg PJ, Müller H: Malignant ameloblstoma or ameloblastic carcinoma. Oral Surg Oral Med Oral Pathol 1984, 57:168-176. 4. Barnes L, Everson JW, Reichart P, editors: Pathology and Genetics: Head and neck tumors. Lyon, World Health Organization 2005, 287-289. 5. Kruse ALD, Zwahlen RA, Grätz KW: New classification of maxillary ameloblastic carcinoma based on an evidence-based literature reviwe over the last 60 years. Head Neck Oncol 2009, 1:31. 6. Lolachi CM, Mada SK, Jacobs JR: Ameloblastic carcinoma of the maxilla. J Laryngol Otol 1995, 109:1019-1022. 7. Lucca M, D’Innocenzo R, Kraus JA, Gagari E, Hall J, Shastri K: Ameloblastic Figure 8 1st folow-up 6 weeks post completion of RT: carcinoma of the maxilla: a report of 2 cases. J Oral Maxillofac Surg 2010. complete remission with only posttherapeutic changes: 8. Haberer T, Becher W, Schardt D, Kraft G: Magnetic scanning system for coronal, contrast enhanced T1. heavy ion therapy. Nucl Instr Meth Phys Res 1993, 330:296-305. Jensen et al. Radiation Oncology 2011, 6:13 Page 5 of 5 http://www.ro-journal.com/content/6/1/13 9. Daramola JO, Abioye AA, Ajagbe HA, Aghadiuno PU: Maxillary malignant ameloblastoma with intraorbital extension: report of a case. J Oral Surg 1980, 38:203-206. 10. Lee L, Maxymiw WG, Wood RE: Ameloblastic carcinoma of the maxilla metastatic to the mandible. Case report. J Craniomaxillofac Surg 1990, 18:247-250. 11. Miyamoto CT, Brady LW, Markoe A, Salinger D: Ameloblastoma of the jaw. Treatment with radiation therapy and a case report. Am J Clin Oncol 1991, 14:225-230. 12. Infante-Cossio P, Hernandez-Guisado JM, Fernandez-Machin P, Garcia- Perla A, Guitierrez-Perez JL: Ameloblastic carcinoma of the maxilla: report of 3 cases. J Cranio Maxillofac Surg 1998, 26:159-162. 13. Zwahlen RA, Vogt P, Fischer FS, Grätz KW: Case report: myocardial metastasis of a maxillary malignant ameloblastoma. J Oral Maxillofac Surg 2003, 61:731-734. 14. Goldenberg D, Sciubba J, Koch W, Tufano RP: Malignant odontogenic tumors: a 22-year experience. Laryngoscope 2004, 114:1770-1774. 15. Philip M, Morris CG, Werning JW, Mendenhall WM: Radiotherapy in the treatment of ameloblastic carcinoma. J HK Coll Radiol 2005, 8:157-161. 16. Simko EJ, Brannon RB, Eibling DE: Ameloblastic carcinoma of the mandible. Head Neck 1998, 20:654-659. 17. Dhir K, Sciubba J, Tufano RP: Ameloblastic carcinoma of the maxilla. Oral Oncol 2003, 39:736-741. 18. Schulz-Ertner D, Nikoghosyan A, Hof H, Didinger B, Combs SE, Jäkel O, Karger CP, Edler L, Debus J: Carbon ion therapy of skull base chondrosarcomas. Int J Radiat Oncol Biol Phys 2007, 67:171-177. 19. Schulz-Ertner D, Karger CP, Feuerhake A, Nikoghosyan A, Combs SE, Jäkel O, Edler L, Scholz M, Debus J: Effectiveness of carbon ion radiotherapy in the treatment of skull-base chordomas. Int J Radiat Oncol Biol Phys 2007, 68:449-457. 20. Schulz-Ertner D, Nikoghosyan A, Didinger B, Münter M, Jäkel O, Karger CP, Debus J: Therapy strategies for locally advanced adenoid-cystic carcinomas using modern radiation therapy techniques. Cancer 2005, 104:338-344. doi:10.1186/1748-717X-6-13 Cite this article as: Jensen et al.: Carbon ion therapy for ameloblastic carcinoma. Radiation Oncology 2011 6:13. Submit your next manuscript to BioMed Central and take full advantage of: • Convenient online submission • Thorough peer review • No space constraints or color figure charges • Immediate publication on acceptance • Inclusion in PubMed, CAS, Scopus and Google Scholar • Research which is freely available for redistribution Submit your manuscript at www.biomedcentral.com/submit

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Radiation Oncology – Springer Journals

Published: Feb 6, 2011

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