Prominent response with helical tomotherapy in recurrent ameloblastic carcinoma of maxillary sinus: a case report
© Koca et al.; licensee BioMed Central Ltd. 2014
Received: 28 March 2014
Accepted: 11 July 2014
Published: 15 July 2014
Ameloblastoma is a benign but locally aggressive tumor of odontogenic epithelial tissue. Reports of radiotherapy treatment modalities are limited in the literature.
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.
In cases of ameloblastic carcinoma with, post-surgical recurrence or patients not suitable for surgical treatment, helical tomotherapy can be an effective treatment option.
Ameloblastoma is a locally aggressive benign tumor derived from the odontogenic epithelial tissues. The tumor has a slight increased preponderance in females and is mainly diagnosed in the third or fourth decade of life[2, 3]. It accounts for about 1% of all jaw tumors. Ameloblastoma prevalently occurs in the ramus and the angulus of the mandible, and rarely in the maxilla. While often clinically asymptomatic, the tumor is usually spotted with bone expansion or detected in routine radiological studies. Numerous histological types have been reported based on the histological findings. The most recent WHO classification has categorized ameloblastoma, to malignant ameloblastoma and ameloblastic carcinoma. Malignant ameloblastoma is different from ameloblastoma since metastases may occur in the former. Both have benign histology. Ameloblastic carcinoma has malignant cytological features regardless of the metastasis occurrence. In ameloblastoma, metastasis rarely occurs.
The basic form of treatment for localized ameloblastoma is radical surgery. The only treatment option in metastatic disease appears to be chemotherapy, although the outcome is not favorable. Radiotherapy modalities are limited in the literature.
Here we report a case of ameloblastic carcinoma with basal cell histology, where helical tomotherapy achieved a prominent response.
Dosimetric Parameters of PTV 60 (SIB) and PTV 54
Although the term ‘ameloblastoma’ was coined by Churchill in 1933, the first detailed description of this lesion was given by Falkson in 1879. While known as benign odontogenic tumors, ameloblastomas are slow-growing tumors with a high recurrence rate and tendency for local invasion, expansion and destruction in the bone[10–12]. Dental caries, trauma, infection, inflammation, dental disorders, malnutrition and viral pathogens have been suggested to play a role in the etiology. The most common symptom is a slow-growing painless swelling, and less frequently, dental malocclusion, pain, paresthesia or anesthesia might occur. In rare cases, pain may be experienced especially when the tumor is infected, but it causes no symptoms unless there is nerve involvement[6, 14].
In 80% of the cases, ameloblastoma originates in the mandible. It mainly involves the angulus and ramus regions of the mandible (70% of cases), whereas 20% of the all cases it involves the premolar region, and 10% the anterior region[2, 5, 15].
According to the histological findings, the tumors are classified as follicular, plexiform, acanthomatous, granular, basal cell and desmoplastic type. The main clinicoradiographic types of ameloblastoma have been defined as conventional solid or multicystic intraosseous, well-defined unicystic (intraosseous) and peripheral (extraosseous)[10, 11, 16].
In the diagnosis of ameloblastoma, the radiological tools such as panoramic radiography, CT, MRI and PET-CT can be used. Panoramic radiography is often used in daily practice, while the other methods are better at detecting the presence of metastases, contours, content and soft tissue extension of the lesion[4, 5, 17, 18].
Although considered a benign tumor, ameloblastoma may develop recurrence after resection and become clinically more aggressive, while leading to massive local destruction and metastasis. 15-25% of the cases develop recurrence after radical surgery, while conservative surgery recurrence rate is 75-90%. In patients allowing radical surgery, despite controversies, 1–2 cm margin is sufficient as it significantly decreases the recurrence rate. Since there is always risk of recurrence, even 25–30 years after the primary treatment, patients should be monitored for a long time. Its metastatic spread incidence has been reported as 1 to 4.5% of all cases. Even though rare, cases with metastases to lungs, pleura, spleen, kidney, heart, skull, spine, brain, and lymph nodes have been reported. Surgery may be an option in the presence of metastases. A review of the current literature reveals that various chemotherapeutic agents have been used, including cisplatin, cyclophosphamide, carboplatin, paclitaxel, doxorubicin, methotrexate, prednisone, bleomycin, 5-fluorouracil and dacarbazine, and varying degrees of responses have been reported for each agent[8, 20].
As ameloblastoma is a rare and slow-growing tumor, the use of radiotherapy in the treatment should be discussed. Also there are limited data for detailed radiotherapy field design and dose prescriptions. The information regarding radiosensitivity in the current literature is ambiguous. In incomplete resection cases, adjuvant radiotherapy may be considered a treatment option[8, 21]. The current studies found in the literature fail to provide sufficient information on the use of radiotherapy/chemoradiotherapy in metastatic disease. Besides, the use of radiotherapy might increase the incidence of conventional bone complications, osteonecrosis and bone carcinoma. With the help of advanced radiotherapy techniques, such complications are tried to be minimized. However, there has been no research examining the use of helical tomotherapy in the treatment of ameloblastoma.
Ameloblastoma is a slow-growing tumor with no standard chemotherapy treatment options, and primarily treated with curative surgical procedure. In conclusion, we suggest that helical tomotherapy can provide an effective treatment option in ameloblastoma cases where complete resection is not feasible or in patients developing local recurrence.
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