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Prominent response with helical tomotherapy in recurrent ameloblastic carcinoma of maxillary sinus: a case report

Prominent response with helical tomotherapy in recurrent ameloblastic carcinoma of maxillary... 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. Keywords: Ameloblastic Carcinoma, Helical Tomotherapy Introduction carcinoma has malignant cytological features regard- Ameloblastoma is a locally aggressive benign tumor de- less of the metastasis occurrence. In ameloblastoma, rived from the odontogenic epithelial tissues [1]. The metastasis rarely occurs [8]. tumor has a slight increased preponderance in females The basic form of treatment for localized ameloblas- and is mainly diagnosed in the third or fourth decade of toma is radical surgery. The only treatment option in life [2,3]. It accounts for about 1% of all jaw tumors [4]. metastatic disease appears to be chemotherapy, although Ameloblastoma prevalently occurs in the ramus and the the outcome is not favorable. Radiotherapy modalities angulus of the mandible, and rarely in the maxilla [5]. are limited in the literature [9]. While often clinically asymptomatic, the tumor is usually Here we report a case of ameloblastic carcinoma with spotted with bone expansion or detected in routine basal cell histology, where helical tomotherapy achieved radiological studies [6]. Numerous histological types a prominent response. have been reported based on the histological findings [7]. The most recent WHO classification has categorized Case ameloblastoma, to malignant ameloblastoma and amelo- A thirty-five year old male presented with complaints of blastic carcinoma. Malignant ameloblastoma is differ- headache radiating to his face for about six months and ent from ameloblastoma since metastases may occur in impaired vision. He presented to an outpatient of Ear, the former. Both have benign histology. Ameloblastic Nose and Throat clinic in April 2012. The Magnetic Resonance Imaging (MRI) and Positron Emission * Correspondence: timurkoca3@gmail.com Tomography (PET) images (Figure 1) shows a tumor Department of Radiation Oncology, Regional Training and Research extending to the sphenoidal sinus from the anterior Hospital, Erzurum, Turkey Full list of author information is available at the end of the article segment of the sphenoid bone. The tumor invades the © 2014 Koca 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/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Koca et al. Radiation Oncology 2014, 9:157 Page 2 of 5 http://www.ro-journal.com/content/9/1/157 Figure 1 Pre-treatment Positron Emission Tomography (PET) images of the case patient. ethmoid bone, sellar and supra-sellar regions, bilateral patient was operated again in June 2012. After the recovery retro-orbital areas, almost completely invading the left from infection, due to the recurrent and residual mass, the maxillary sinus and extending to the nasal cavity. The multi-disciplinary oncology council decided to consult with mass was observed to displace optic nerves bilaterally. the Radiation Oncology Clinic for post-operative external The tumor size was measured at 57 × 56 × 63 mm. radiotherapy. The visual field examination The visual as- Post-contrast series demonstrated intense and hetero- sessment conducted by the Eye Diseases Clinic before the geneous contrast enhancement. Based on the biopsy initiation of external radiotherapy revealed bilateral visual findings, the patient was diagnosed with ameloblastic impairment and diplopia. Serial tomographic sections were carcinoma with basaloid appearance (Figure 2). The taken for the purpose of contouring in radiation therapy patient underwent surgery in the Neurosurgery Clinic plan. The sections were transmitted to the contouring work in May 2012. Due to the size of the mass and proximity station through Digital Imaging and Communications in to critical organs, only R2 (macroscopic residual tumor) Medicine (DICOM), and Reconstructive Digital Radiog- resection could be performed. The patient developed post- raphy (DRR) was obtained. The DRR contouring of the pa- TM operative infection, and was treated with appropriate anti- tient was performed using a Tomocon (Tetramed , biotic therapy, which failed to treat the infection. The Slovak republic) contouring workstation. The patient’seyes, Koca et al. Radiation Oncology 2014, 9:157 Page 3 of 5 http://www.ro-journal.com/content/9/1/157 The MR images taken at 2 months after radiotherapy showed partial regression in lesion size. Complete clin- ical remission was achieved in patient’s bilateral visual impairment and diplopia during follow-up. The PET scan taken at 12 months after radiotherapy demon- strated regression of the tumor in the left maxillary sinus, which reduced to 3 × 3.5 × 3 cm, and 18F-fluoro- 2-deoxy-D-glucose (FDG) uptake decreased (Previous SUD: 25.36; Current SUD 5.33), while a complete re- sponse was achieved in the lesion intracranial extensions (Figure 3). The brain MRI detected no pathological contrast involvement in this area. The patient is under ongoing follow-up. He has not reported any complaints in the check-up conducted in January 2014. Discussion Although the term ‘ameloblastoma’ was coined by Churchill in 1933, the first detailed description of this Figure 2 Ameloblastic carcinoma with basaloid appearance. lesion was given by Falkson in 1879 [3]. While known as benign odontogenic tumors, ameloblastomas are slow-growing tumors with a high recurrence rate and optic nerves, lenses, brain stem, optic chiasm, parotid tendency for local invasion, expansion and destruction glands, spinal cord, cochleae, vestibules and skin were con- in the bone [10-12]. Dental caries, trauma, infection, toured as critical organs. Prior to the radiotherapy for ame- inflammation, dental disorders, malnutrition and viral loblastic carcinoma, two separate clinical target volumes pathogens have been suggested to play a role in the eti- (CTV) were defined. CTV1 was defined by adding a 5 mm ology [13]. The most common symptom is a slow- margin to the gross tumor volume, while planning target growing painless swelling, and less frequently, dental volume (PTV) 1 was obtained by adding a 3 mm margin to malocclusion, pain, paresthesia or anesthesia might the CTV1 volume, and then administration of 60 Gy total occur. In rare cases, pain may be experienced espe- dose was planned using simultaneous integrated boost cially when the tumor is infected, but it causes no (SIB) technique. PTV was defined by adding a 15 mm symptoms unless there is nerve involvement [6,14]. margin to CTV volume, and the prescription dose was 50 In 80% of the cases, ameloblastoma originates in the Gy to PTV (Table 1). The patient’s treatment plan was de- mandible. It mainly involves the angulus and ramus re- signed with the Tomotherapy planning system (Accuray gions of the mandible (70% of cases), whereas 20% of Inc., Madison, USA). With this planning system, the appro- the all cases it involves the premolar region, and 10% priate prescription doses for organs at risk (OAR) were de- the anterior region [2,5,15]. fined, and routine quality assurance for the prescribed According to the histological findings, the tumors doses was conducted to prepare the patient for the treat- are classified as follicular, plexiform, acanthomatous, ment. Related to metastases and close proximity of OAR’s granular, basal cell and desmoplastic type [7]. The to PTV’s, some of the OAR dose objectives were slightly main clinicoradiographic types of ameloblastoma have exceeded Quantec recommendations. The patient was in- been defined as conventional solid or multicystic formed about the treatment related adverse effects and intraosseous, well-defined unicystic (intraosseous) and informed consent was signed before the start of the treat- peripheral (extraosseous) [10,11,16]. ment session. Prior to each session of treatment, daily Mega In the diagnosis of ameloblastoma, the radiological Voltage Computerized Tomography (MVCT) scans were tools such as panoramic radiography, CT, MRI and PET- performed, and these were compared with the images of CT can be used. Panoramic radiography is often used in treatment planning to achieve set-up accuracy. daily practice, while the other methods are better at de- tecting the presence of metastases, contours, content Table 1 Dosimetric Parameters of PTV (SIB) and PTV 60 54 and soft tissue extension of the lesion [4,5,17,18]. Variable D (Gy) D (Gy) D (Gy) HI CI max mean min Although considered a benign tumor, ameloblastoma PTV 64.72 61.38 47.44 0.08 0.78 may develop recurrence after resection and become clin- ically more aggressive, while leading to massive local PTV 64.72 60.45 24.61 0.28 0.82 destruction and metastasis [7]. 15-25% of the cases de- Abreviations: D maximum dose, D mean dose, D Minimum dose, max mean min PTV planned target volume, HI Homogenity index, CI Conformity index. velop recurrence after radical surgery, while conservative Koca et al. Radiation Oncology 2014, 9:157 Page 4 of 5 http://www.ro-journal.com/content/9/1/157 Figure 3 Positron Emission Tomography (PET) images of the patient after external radiotherapy. surgery recurrence rate is 75-90% [19]. In patients allowing varying degrees of responses have been reported for each radical surgery, despite controversies, 1–2cmmarginis agent [8,20]. sufficient as it significantly decreases the recurrence rate As ameloblastoma is a rare and slow-growing tumor, [2]. Since there is always risk of recurrence, even 25–30 the use of radiotherapy in the treatment should be years after the primary treatment, patients should be moni- discussed. Also there are limited data for detailed tored for a long time [3]. Its metastatic spread incidence radiotherapy field design and dose prescriptions. The has been reported as 1 to 4.5% of all cases [2]. Even information regarding radiosensitivity in the current though rare, cases with metastases to lungs, pleura, literature is ambiguous [9]. In incomplete resection spleen, kidney, heart, skull, spine, brain, and lymph cases, adjuvant radiotherapy may be considered a nodeshavebeen reported [11].Surgery maybean treatment option [8,21]. The current studies found in option in the presence of metastases. A review of the the literature fail to provide sufficient information on current literature reveals that various chemotherapeutic the use of radiotherapy/chemoradiotherapy in meta- agents have been used, including cisplatin, cyclophospha- static disease [8]. Besides, the use of radiotherapy mide, carboplatin, paclitaxel, doxorubicin, methotrexate, might increase the incidence of conventional bone prednisone, bleomycin, 5-fluorouracil and dacarbazine, and complications, osteonecrosis and bone carcinoma [9]. Koca et al. Radiation Oncology 2014, 9:157 Page 5 of 5 http://www.ro-journal.com/content/9/1/157 With the help of advanced radiotherapy techniques, 9. Angiero F, Borloni R, Macchi M, Stefani M: Ameloblastic Carcinoma of the Maxillary Sinus. Anticancer Res 2008, 28:3847–3854. such complications are tried to be minimized. However, 10. Cihangiroğlu M, Akfırat M, YıldırımH: CT and MRI findings of there has been no research examining the use of helical ameloblastoma in two cases. Neuroradiology 2002, 44:434–437. tomotherapy in the treatment of ameloblastoma. 11. Miyamato CT, Brady LW, Markoe A, Salinger D: Ameloblastoma of the Jaw. Am J Clin Oncol 1991, 14:225–230. 12. Sehdev MK, Huvos AG, Strong EW, Gerold FP, Willis GW: Ameloblastoma of Conclusion maxilla and mandible. Cancer 1974, 33:324–333. 13. Nakamura N, Mitsuyasu T, Higuchi Y, Sandra F, Ohishi M: Growth Ameloblastoma is a slow-growing tumor with no standard characteristics of ameloblastoma involving the inferior alveolar nerve: chemotherapy treatment options, and primarily treated a clinical and histopathologic study. Oral Surg Oral Med Oral Pathol Oral with curative surgical procedure. In conclusion, we suggest Radiol Endod 2001, 91:557.562. 14. Becelli R, Carboni A, Cerulli G, Perugini M, Iannetti G: Mandibular that helical tomotherapy can provide an effective treatment Ameloblastoma: Analysis of Surgical Treatment Carried Out in 60 option in ameloblastoma cases where complete resection is Patients Between 1977 and 1998. J Craniofac Surg 2002, 13:395–400. not feasible or in patients developing local recurrence. 15. Hollows P, Fasanmade A, Hayter JP: Ameloblastoma - a diagnostic problem. Br Dent J 2000, 188:243.244. 16. Han MH, Chang KH, Lee CH, Na DG, Yeon KM, Han MC: Cystic Expansile Competing interests Masses of the Maxilla: Differential Diagnosis with CT and MR. AJNR Am J The authors declare that they have no competing interests. Neuroradiol 1995, 16:333–338. 17. Kawai T, Murakami S, Kishino M, Matsuya T, Sakuda M, Fuchihata H: Authors’ contributions Diagnostic imaging in two cases of recurrent maxillary ameloblastoma: TK (Corresponding author): Carried out contouring the patient, drafted, comparative evaluation of plain radiographs, CT and MR images. Br J generally evaluated and write the manuscript, HB: Made the tables and Oral Maxillofac Surg 1998, 36:304.310. carried out coordination between clinics, DA: Carried out the patient 18. Ziegler CM, Woertche R, Brief J, Hassfeld S: Clinical indications for digital interrogation and follow-up, write the manuscript, DS: Evaluated the patients volume tomography in oral and maxillofacial surgery. Dentomaxillofac radiographs and made calculations, ZAÇ: Carried out the patient set-up Radiol 2002, 31:126.130. preperation, ÖK: Carried out set-up verifications, SK: Prepared the treatment 19. Nakamura N, Higuchi Y, Mitsuyasu T, Sandra F, Ohishi M: Comparison of plans of the case, CİB: Carried out histo-pathological examination, HÖO: long-term results between different approaches to ameloblastoma. Carried out surgery, MD: Carried out PET scans. All authors read and Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002, 93:13–20. approved the final manuscript. 20. Ghiam A, Al Zahrani A, Feld R: Acase of recurrent metastatic ameloblastoma and hypercalcaemia successfully treated with carboplatin and paclitaxel: Author details 1 long survival and prolonged stable disease. Ecancer 2013, 7:323. Department of Radiation Oncology, Regional Training and Research 2 21. Veena M, Manoranjan V, Kiran A, Anshu J, Mohammed AS, Kavita G, Aastha Hospital, Erzurum, Turkey. Department of Medical Oncology, Regional 3 G: Ameloblastic carcinoma: a rare entity. BMJ Case Reports 2011, 10:1136. Training and Research Hospital, Erzurum, Turkey. Department of Pathology, Akdeniz University Medical Faculty Hospital, Antalya, Turkey. Regional doi:10.1186/1748-717X-9-157 Training and Research Hospital, Clinic of Neurosurgery, Erzurum, Turkey. 5 Cite this article as: Koca et al.: Prominent response with helical Regional Training and Research Hospital, Clinic of Nuclear Medicine, tomotherapy in recurrent ameloblastic carcinoma of maxillary sinus: Erzurum, Turkey. a case report. Radiation Oncology 2014 9:157. Received: 28 March 2014 Accepted: 11 July 2014 Published: 15 July 2014 References 1. Gardner DG, Heikinheimo K, Shear M, Philpsen HP, Coleman H: “Ameloblastomas,” in World Health Organization Classification of Tumors.In Pathology and Genetics.Head and Neck Tumours. Edited by Barnes L, Eveson JW, Reichart P, Sidransky D. Lyon, France: IARC Press; 2005:296–300. 2. Torres-Lagares D, Infante-Cossío P, Hernández-Guisado JM, Gutiérrez-Pérez JL: Mandibular ameloblastoma. A review of the literature and presentation of six cases. Med Oral Patol Oral Cir Bucal 2005, 10:231.238. 3. Iordanidis S, Makos C, Dimitrakopoulos J, Kariki H: Ameloblastoma of the maxilla. Case report Aust Dent J 1999, 44:51–55. 4. Hanna T, Tomasz Z, Anna R, Tomasz K: Ameloblastoma of the Nasal Septum Origin: A Case Report. Case Rep Otolaryngol 2013, 2013:3. Article ID 280509. Submit your next manuscript to BioMed Central 5. Tozaki M, Hayashi K, Fukuda K: Dynamic multislice helical CT of maxillomandibular lesions: distinction of ameloblastomas from other and take full advantage of: cystic lesions. Radiat Med 2001, 19:225.230. 6. Escande C, Chaine A, Menard P, Ernenwein D, Ghoul S, Bouattour A, Berdal A, • Convenient online submission Bertrand JC, Ruhin-Poncet B: A treatment algorithm for adult ameloblastomas • Thorough peer review according to the Pitié-Salpêtrière Hospital experience. J Craniomaxillofac Surg 2009, 37:363–369. • No space constraints or color figure charges 7. Ogunsalu C, Daisley H, Henry K, Bedayse S, White K, Jagdeo B, Baldeo S: • Immediate publication on acceptance A new radiological classification for ameloblastoma based on analysis of • Inclusion in PubMed, CAS, Scopus and Google Scholar 19 cases. West Indian Med J 2006, 55:434–439. 8. Amzerin M, Fadoukhair Z, Belbaraka R, Iraqui M, Boutayeb S, M'rabti H, • Research which is freely available for redistribution Kebdani T, Hassouni K, Benjaafar N, El Gueddari BK, Errihani H: Metastatic ameloblastoma responding to combination chemotherapy: case report Submit your manuscript at and review of the literature. J Med Case Rep 2011, 5:491. www.biomedcentral.com/submit http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Radiation Oncology Springer Journals

Prominent response with helical tomotherapy in recurrent ameloblastic carcinoma of maxillary sinus: a case report

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Copyright © 2014 by Koca et al.; licensee BioMed Central Ltd.
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Medicine & Public Health; Oncology; Radiotherapy
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25027948
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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. Keywords: Ameloblastic Carcinoma, Helical Tomotherapy Introduction carcinoma has malignant cytological features regard- Ameloblastoma is a locally aggressive benign tumor de- less of the metastasis occurrence. In ameloblastoma, rived from the odontogenic epithelial tissues [1]. The metastasis rarely occurs [8]. tumor has a slight increased preponderance in females The basic form of treatment for localized ameloblas- and is mainly diagnosed in the third or fourth decade of toma is radical surgery. The only treatment option in life [2,3]. It accounts for about 1% of all jaw tumors [4]. metastatic disease appears to be chemotherapy, although Ameloblastoma prevalently occurs in the ramus and the the outcome is not favorable. Radiotherapy modalities angulus of the mandible, and rarely in the maxilla [5]. are limited in the literature [9]. While often clinically asymptomatic, the tumor is usually Here we report a case of ameloblastic carcinoma with spotted with bone expansion or detected in routine basal cell histology, where helical tomotherapy achieved radiological studies [6]. Numerous histological types a prominent response. have been reported based on the histological findings [7]. The most recent WHO classification has categorized Case ameloblastoma, to malignant ameloblastoma and amelo- A thirty-five year old male presented with complaints of blastic carcinoma. Malignant ameloblastoma is differ- headache radiating to his face for about six months and ent from ameloblastoma since metastases may occur in impaired vision. He presented to an outpatient of Ear, the former. Both have benign histology. Ameloblastic Nose and Throat clinic in April 2012. The Magnetic Resonance Imaging (MRI) and Positron Emission * Correspondence: timurkoca3@gmail.com Tomography (PET) images (Figure 1) shows a tumor Department of Radiation Oncology, Regional Training and Research extending to the sphenoidal sinus from the anterior Hospital, Erzurum, Turkey Full list of author information is available at the end of the article segment of the sphenoid bone. The tumor invades the © 2014 Koca 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/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Koca et al. Radiation Oncology 2014, 9:157 Page 2 of 5 http://www.ro-journal.com/content/9/1/157 Figure 1 Pre-treatment Positron Emission Tomography (PET) images of the case patient. ethmoid bone, sellar and supra-sellar regions, bilateral patient was operated again in June 2012. After the recovery retro-orbital areas, almost completely invading the left from infection, due to the recurrent and residual mass, the maxillary sinus and extending to the nasal cavity. The multi-disciplinary oncology council decided to consult with mass was observed to displace optic nerves bilaterally. the Radiation Oncology Clinic for post-operative external The tumor size was measured at 57 × 56 × 63 mm. radiotherapy. The visual field examination The visual as- Post-contrast series demonstrated intense and hetero- sessment conducted by the Eye Diseases Clinic before the geneous contrast enhancement. Based on the biopsy initiation of external radiotherapy revealed bilateral visual findings, the patient was diagnosed with ameloblastic impairment and diplopia. Serial tomographic sections were carcinoma with basaloid appearance (Figure 2). The taken for the purpose of contouring in radiation therapy patient underwent surgery in the Neurosurgery Clinic plan. The sections were transmitted to the contouring work in May 2012. Due to the size of the mass and proximity station through Digital Imaging and Communications in to critical organs, only R2 (macroscopic residual tumor) Medicine (DICOM), and Reconstructive Digital Radiog- resection could be performed. The patient developed post- raphy (DRR) was obtained. The DRR contouring of the pa- TM operative infection, and was treated with appropriate anti- tient was performed using a Tomocon (Tetramed , biotic therapy, which failed to treat the infection. The Slovak republic) contouring workstation. The patient’seyes, Koca et al. Radiation Oncology 2014, 9:157 Page 3 of 5 http://www.ro-journal.com/content/9/1/157 The MR images taken at 2 months after radiotherapy showed partial regression in lesion size. Complete clin- ical remission was achieved in patient’s bilateral visual impairment and diplopia during follow-up. The PET scan taken at 12 months after radiotherapy demon- strated regression of the tumor in the left maxillary sinus, which reduced to 3 × 3.5 × 3 cm, and 18F-fluoro- 2-deoxy-D-glucose (FDG) uptake decreased (Previous SUD: 25.36; Current SUD 5.33), while a complete re- sponse was achieved in the lesion intracranial extensions (Figure 3). The brain MRI detected no pathological contrast involvement in this area. The patient is under ongoing follow-up. He has not reported any complaints in the check-up conducted in January 2014. Discussion Although the term ‘ameloblastoma’ was coined by Churchill in 1933, the first detailed description of this Figure 2 Ameloblastic carcinoma with basaloid appearance. lesion was given by Falkson in 1879 [3]. While known as benign odontogenic tumors, ameloblastomas are slow-growing tumors with a high recurrence rate and optic nerves, lenses, brain stem, optic chiasm, parotid tendency for local invasion, expansion and destruction glands, spinal cord, cochleae, vestibules and skin were con- in the bone [10-12]. Dental caries, trauma, infection, toured as critical organs. Prior to the radiotherapy for ame- inflammation, dental disorders, malnutrition and viral loblastic carcinoma, two separate clinical target volumes pathogens have been suggested to play a role in the eti- (CTV) were defined. CTV1 was defined by adding a 5 mm ology [13]. The most common symptom is a slow- margin to the gross tumor volume, while planning target growing painless swelling, and less frequently, dental volume (PTV) 1 was obtained by adding a 3 mm margin to malocclusion, pain, paresthesia or anesthesia might the CTV1 volume, and then administration of 60 Gy total occur. In rare cases, pain may be experienced espe- dose was planned using simultaneous integrated boost cially when the tumor is infected, but it causes no (SIB) technique. PTV was defined by adding a 15 mm symptoms unless there is nerve involvement [6,14]. margin to CTV volume, and the prescription dose was 50 In 80% of the cases, ameloblastoma originates in the Gy to PTV (Table 1). The patient’s treatment plan was de- mandible. It mainly involves the angulus and ramus re- signed with the Tomotherapy planning system (Accuray gions of the mandible (70% of cases), whereas 20% of Inc., Madison, USA). With this planning system, the appro- the all cases it involves the premolar region, and 10% priate prescription doses for organs at risk (OAR) were de- the anterior region [2,5,15]. fined, and routine quality assurance for the prescribed According to the histological findings, the tumors doses was conducted to prepare the patient for the treat- are classified as follicular, plexiform, acanthomatous, ment. Related to metastases and close proximity of OAR’s granular, basal cell and desmoplastic type [7]. The to PTV’s, some of the OAR dose objectives were slightly main clinicoradiographic types of ameloblastoma have exceeded Quantec recommendations. The patient was in- been defined as conventional solid or multicystic formed about the treatment related adverse effects and intraosseous, well-defined unicystic (intraosseous) and informed consent was signed before the start of the treat- peripheral (extraosseous) [10,11,16]. ment session. Prior to each session of treatment, daily Mega In the diagnosis of ameloblastoma, the radiological Voltage Computerized Tomography (MVCT) scans were tools such as panoramic radiography, CT, MRI and PET- performed, and these were compared with the images of CT can be used. Panoramic radiography is often used in treatment planning to achieve set-up accuracy. daily practice, while the other methods are better at de- tecting the presence of metastases, contours, content Table 1 Dosimetric Parameters of PTV (SIB) and PTV 60 54 and soft tissue extension of the lesion [4,5,17,18]. Variable D (Gy) D (Gy) D (Gy) HI CI max mean min Although considered a benign tumor, ameloblastoma PTV 64.72 61.38 47.44 0.08 0.78 may develop recurrence after resection and become clin- ically more aggressive, while leading to massive local PTV 64.72 60.45 24.61 0.28 0.82 destruction and metastasis [7]. 15-25% of the cases de- Abreviations: D maximum dose, D mean dose, D Minimum dose, max mean min PTV planned target volume, HI Homogenity index, CI Conformity index. velop recurrence after radical surgery, while conservative Koca et al. Radiation Oncology 2014, 9:157 Page 4 of 5 http://www.ro-journal.com/content/9/1/157 Figure 3 Positron Emission Tomography (PET) images of the patient after external radiotherapy. surgery recurrence rate is 75-90% [19]. In patients allowing varying degrees of responses have been reported for each radical surgery, despite controversies, 1–2cmmarginis agent [8,20]. sufficient as it significantly decreases the recurrence rate As ameloblastoma is a rare and slow-growing tumor, [2]. Since there is always risk of recurrence, even 25–30 the use of radiotherapy in the treatment should be years after the primary treatment, patients should be moni- discussed. Also there are limited data for detailed tored for a long time [3]. Its metastatic spread incidence radiotherapy field design and dose prescriptions. The has been reported as 1 to 4.5% of all cases [2]. Even information regarding radiosensitivity in the current though rare, cases with metastases to lungs, pleura, literature is ambiguous [9]. In incomplete resection spleen, kidney, heart, skull, spine, brain, and lymph cases, adjuvant radiotherapy may be considered a nodeshavebeen reported [11].Surgery maybean treatment option [8,21]. The current studies found in option in the presence of metastases. A review of the the literature fail to provide sufficient information on current literature reveals that various chemotherapeutic the use of radiotherapy/chemoradiotherapy in meta- agents have been used, including cisplatin, cyclophospha- static disease [8]. Besides, the use of radiotherapy mide, carboplatin, paclitaxel, doxorubicin, methotrexate, might increase the incidence of conventional bone prednisone, bleomycin, 5-fluorouracil and dacarbazine, and complications, osteonecrosis and bone carcinoma [9]. Koca et al. Radiation Oncology 2014, 9:157 Page 5 of 5 http://www.ro-journal.com/content/9/1/157 With the help of advanced radiotherapy techniques, 9. Angiero F, Borloni R, Macchi M, Stefani M: Ameloblastic Carcinoma of the Maxillary Sinus. Anticancer Res 2008, 28:3847–3854. such complications are tried to be minimized. However, 10. Cihangiroğlu M, Akfırat M, YıldırımH: CT and MRI findings of there has been no research examining the use of helical ameloblastoma in two cases. Neuroradiology 2002, 44:434–437. tomotherapy in the treatment of ameloblastoma. 11. Miyamato CT, Brady LW, Markoe A, Salinger D: Ameloblastoma of the Jaw. Am J Clin Oncol 1991, 14:225–230. 12. Sehdev MK, Huvos AG, Strong EW, Gerold FP, Willis GW: Ameloblastoma of Conclusion maxilla and mandible. Cancer 1974, 33:324–333. 13. Nakamura N, Mitsuyasu T, Higuchi Y, Sandra F, Ohishi M: Growth Ameloblastoma is a slow-growing tumor with no standard characteristics of ameloblastoma involving the inferior alveolar nerve: chemotherapy treatment options, and primarily treated a clinical and histopathologic study. Oral Surg Oral Med Oral Pathol Oral with curative surgical procedure. In conclusion, we suggest Radiol Endod 2001, 91:557.562. 14. Becelli R, Carboni A, Cerulli G, Perugini M, Iannetti G: Mandibular that helical tomotherapy can provide an effective treatment Ameloblastoma: Analysis of Surgical Treatment Carried Out in 60 option in ameloblastoma cases where complete resection is Patients Between 1977 and 1998. J Craniofac Surg 2002, 13:395–400. not feasible or in patients developing local recurrence. 15. Hollows P, Fasanmade A, Hayter JP: Ameloblastoma - a diagnostic problem. Br Dent J 2000, 188:243.244. 16. Han MH, Chang KH, Lee CH, Na DG, Yeon KM, Han MC: Cystic Expansile Competing interests Masses of the Maxilla: Differential Diagnosis with CT and MR. AJNR Am J The authors declare that they have no competing interests. Neuroradiol 1995, 16:333–338. 17. Kawai T, Murakami S, Kishino M, Matsuya T, Sakuda M, Fuchihata H: Authors’ contributions Diagnostic imaging in two cases of recurrent maxillary ameloblastoma: TK (Corresponding author): Carried out contouring the patient, drafted, comparative evaluation of plain radiographs, CT and MR images. Br J generally evaluated and write the manuscript, HB: Made the tables and Oral Maxillofac Surg 1998, 36:304.310. carried out coordination between clinics, DA: Carried out the patient 18. Ziegler CM, Woertche R, Brief J, Hassfeld S: Clinical indications for digital interrogation and follow-up, write the manuscript, DS: Evaluated the patients volume tomography in oral and maxillofacial surgery. Dentomaxillofac radiographs and made calculations, ZAÇ: Carried out the patient set-up Radiol 2002, 31:126.130. preperation, ÖK: Carried out set-up verifications, SK: Prepared the treatment 19. Nakamura N, Higuchi Y, Mitsuyasu T, Sandra F, Ohishi M: Comparison of plans of the case, CİB: Carried out histo-pathological examination, HÖO: long-term results between different approaches to ameloblastoma. Carried out surgery, MD: Carried out PET scans. All authors read and Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002, 93:13–20. approved the final manuscript. 20. Ghiam A, Al Zahrani A, Feld R: Acase of recurrent metastatic ameloblastoma and hypercalcaemia successfully treated with carboplatin and paclitaxel: Author details 1 long survival and prolonged stable disease. Ecancer 2013, 7:323. Department of Radiation Oncology, Regional Training and Research 2 21. Veena M, Manoranjan V, Kiran A, Anshu J, Mohammed AS, Kavita G, Aastha Hospital, Erzurum, Turkey. Department of Medical Oncology, Regional 3 G: Ameloblastic carcinoma: a rare entity. BMJ Case Reports 2011, 10:1136. Training and Research Hospital, Erzurum, Turkey. Department of Pathology, Akdeniz University Medical Faculty Hospital, Antalya, Turkey. Regional doi:10.1186/1748-717X-9-157 Training and Research Hospital, Clinic of Neurosurgery, Erzurum, Turkey. 5 Cite this article as: Koca et al.: Prominent response with helical Regional Training and Research Hospital, Clinic of Nuclear Medicine, tomotherapy in recurrent ameloblastic carcinoma of maxillary sinus: Erzurum, Turkey. a case report. Radiation Oncology 2014 9:157. Received: 28 March 2014 Accepted: 11 July 2014 Published: 15 July 2014 References 1. Gardner DG, Heikinheimo K, Shear M, Philpsen HP, Coleman H: “Ameloblastomas,” in World Health Organization Classification of Tumors.In Pathology and Genetics.Head and Neck Tumours. Edited by Barnes L, Eveson JW, Reichart P, Sidransky D. Lyon, France: IARC Press; 2005:296–300. 2. Torres-Lagares D, Infante-Cossío P, Hernández-Guisado JM, Gutiérrez-Pérez JL: Mandibular ameloblastoma. A review of the literature and presentation of six cases. 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Ogunsalu C, Daisley H, Henry K, Bedayse S, White K, Jagdeo B, Baldeo S: • Immediate publication on acceptance A new radiological classification for ameloblastoma based on analysis of • Inclusion in PubMed, CAS, Scopus and Google Scholar 19 cases. West Indian Med J 2006, 55:434–439. 8. Amzerin M, Fadoukhair Z, Belbaraka R, Iraqui M, Boutayeb S, M'rabti H, • Research which is freely available for redistribution Kebdani T, Hassouni K, Benjaafar N, El Gueddari BK, Errihani H: Metastatic ameloblastoma responding to combination chemotherapy: case report Submit your manuscript at and review of the literature. J Med Case Rep 2011, 5:491. www.biomedcentral.com/submit

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Published: Jul 15, 2014

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