|CASE REPORT - TUMOR
|Year : 2019 | Volume
| Issue : 1 | Page : 197-200
Successful management of a gigantic ameloblastic fibroma: A 12-year follow-up
Wladimir Gushiken De Campos1, Camilla Vieira Esteves1, Gustavo Alkmin Paiva2, Camila Eduarda Zambon2, André Caroli Rocha2, Celso Augusto Lemos1
1 Department of Oral Medicine, School of Dentistry, University of São Paulo, São Paulo, Brazil
2 Department of Oral and Maxillofacial Surgery, Clinics Hospital, University of São Paulo, São Paulo, Brazil
|Date of Web Publication||13-Jun-2019|
Dr. Wladimir Gushiken De Campos
Department of Stomatology, School of Dentistry, University of São Paulo, Avenida Professor Lineu Prestes, 2227, São Paulo
An ameloblastic fibroma (AF) is a benign mixed odontogenic tumor that mainly affects patients in the first and second decades of life. It is usually associated with an impacted tooth, commonly the first or second permanent molar. We present a case of an 11-year-old male patient diagnosed with AF, showing well-defined borders almost completely affecting the body and inferior alveolar nerve (IAN) branch on the right side of the mandible and displacing teeth 46 and 47 associated with the lesion. The treatment included conservative surgery, with oral rehabilitation, including IAN lateralization, implant placement without sensorial alteration, and posterior rehabilitation, performed after 10 years of follow-up. Rehabilitation with implants is a safe and effective procedure for the prosthetic rehabilitation of the posterior atrophic mandible. However, sensorial alteration of the IAN occurs in 100% of cases and tends to regress with time or may be permanent in few cases.
Keywords: Ameloblastic fibroma, mandibular tumor, odontogenic tumor
|How to cite this article:|
De Campos WG, Esteves CV, Paiva GA, Zambon CE, Rocha AC, Lemos CA. Successful management of a gigantic ameloblastic fibroma: A 12-year follow-up. Ann Maxillofac Surg 2019;9:197-200
|How to cite this URL:|
De Campos WG, Esteves CV, Paiva GA, Zambon CE, Rocha AC, Lemos CA. Successful management of a gigantic ameloblastic fibroma: A 12-year follow-up. Ann Maxillofac Surg [serial online] 2019 [cited 2019 Jul 20];9:197-200. Available from: http://www.amsjournal.com/text.asp?2019/9/1/197/260345
| Introduction|| |
An ameloblastic fibroma (AF) is composed of odontogenic epithelium proliferation in cellular ectomesenchyme, resembling the dental papilla. Clinical and radiographic features are essential for differential diagnosis.
The AF is a rare mesenchymal odontogenic tumor (OT) consisting of a solid mass of soft tissue with a smooth outer surface.
In several cases, treatment of the lesion leads to large defects in the jaws. The reconstruction of the mandibular defect should aim for the complete recovery of esthetics and function.,
In cases of rehabilitation with implants in the posterior region of the atrophic mandible, the inferior alveolar nerve lateralization (IANL) and transposition can be used for dental implant placement. In this technique, the IAN is exposed through an osteotomy of the vestibular cortex and displaced laterally, while the implants are installed. The IAN is then placed back into position against the implant.
The advantages of IANL include the possibility of placing long implants and its anchorage in two cortical bones, which favor initial stability. Some inherent disadvantages include the dysfunction of the IAN, leading to alteration in the sensation of the lower lip and skin of the chin.,
| Case Report|| |
An 11-year-old male patient was referred for evaluation of a swelling evolving for approximately 3 months without any painful symptoms. Clinical examination showed facial asymmetry in the middle and lower third of the face without inflammatory signs [Figure 1]a.
|Figure 1: (a) Front view: Initial photo. (b) Front view: 12-year follow-up|
Click here to view
The panoramic radiograph examination revealed a radiolucent image of an expansive, multiloculated lesion with well-defined borders that affected the body and ramus of the right mandible and displaced teeth 46 and 47 toward the basal cortex [Figure 2]a.
|Figure 2: (a) Panoramic radiograph: A huge osteolytic lesion involving the right mandibular body and ascending ramus. (b) 12-year follow-up radiography: Resolution of the lesion, with bone neoformation and dental implant osseointegration|
Click here to view
On computed tomography examination, a solid lesion with external and internal cortical expansion and cortical thinning of the right mandibular ramus associated with 47 as well as regions with cortical discontinuity was observed. The lesion had attenuation similar to the muscles and small foci of hypoattenuation in its interior region [Figure 3].
|Figure 3: Computed tomography: Coronal view of an osteolytic lesion with bulging of the buccal and lingual cortex|
Click here to view
The differential diagnoses included solid ameloblastoma, myxoma, and AF since all were benign tumors of odontogenic origin. After incisional biopsy, the diagnosis of AF was confirmed. The patient's mother signed the consent declaration for the minor.
The lesion was enucleated along with teeth 46 and 47 under general anesthesia through an intraoral approach. Tooth 45 was preserved [Figure 4].
|Figure 4: Transoperative photo: Surgical excision of the lesion using conservative technique|
Click here to view
At the 7-year follow-up, there were no signs of relapse in the clinical and radiographic examinations. Tooth 45 had been extracted by orthodontic indication. After 10 years of AF excision [Figure 1]b, three implants were placed in the right posterior mandible region with the IANL technique.
In the 7-day postoperative period, the patient presented with hypoesthesia of the IAN, who was treated with low-level laser therapy sessions. Four laser therapy sessions improved 97% of the IAN hypoesthesia.
Twelve years after the tumor excision, there were no clinical or imaging signs of recurrence of the lesion, and the implants showed osseointegration for future fixed prosthetic rehabilitation [Figure 2]b.
| Discussion|| |
A rare benign OT called AF was reported in this case. Only 21.6% OTs are classified as mesenchymal OT, where AF represents 2.16% of all tumors and is the seventh most frequent OT. On the other hand, AF represents 1.4% of all OTs and is the eighth most common OT.
According to the last WHO Classification of Head and Neck Tumors, AF produces dental hard tissues due to the maturation of the lesion into odontomas, which does not characterize different pathological entities, but different stages of maturation.
The case reported here presents an 11-year-old male patient. AF is found to be highly prevalent between the first and second decades of life. Usually, 77.7% of individuals are affected within the first and second decades of life, with a mean age of 14.8 years.
A recent study analyzed 172 cases, showing an age range of 1 month–57 years with an average of 14.9 years and median of 11 years. Approximately 80% of cases were diagnosed before 22 years of age. The male:female sex ratio of affected individuals was 1.4:1.,
The OTs affect the mandible 2.5 times more than the maxilla. The posterior mandible is most frequently affected by OTs with a maxilla:mandible ratio of 1:2.1. In the reported case, the AF was located in the posterior region of the mandible, which is the site most commonly affected by OT and AF.
Radiographically, AF may present as a well-delimited, unilocular, or multilocular image with sclerotic borders in most cases. The multilocular characteristic is usually observed in large lesions, while the unilocular characteristic is related to minor lesions.
AF which is a radiolucent lesion shows well-defined borders in 94% of cases. The image presents unilocular characteristics in 56% of cases and multilocular characteristics in 44% of the cases. The lesions <5 cm in size were usually unilocular, while those >5 cm were mostly multilocular (P < 0.001).
Approximately 75% of cases showed the involvement of an unerupted tooth with AF. Most AFs included one or more teeth (80.5%) that usually belonged to the permanent dentition., The reported case presented with a large lesion of approximately 5-cm size with painless increase in volume, associated with a multilocular image.
In about 65% of the cases, the painless expansion of the cortical region was related to the lesion, while the painless expansion of the cortical region caused asymmetry in 17.7% of cases. Expansion of cortical bone with painful symptoms was reported in 8.5% of AFs.
In contrast, the lesion was completely asymptomatic and discovered on radiographs used to assess delayed tooth eruption in 15.6% of cases and identified on routine radiographs in 17% of cases. An increase in volume was the most frequent complaint before the diagnosis (71.8%); however, 23.3% lesions were detected radiographically. The increase in volume was a more frequent complaint than radiographic findings. The chief complaint of our patient was painless asymmetry.
Due to the benign clinical behavior of AF, an initial aggressive treatment was not indicated. Meticulous surgical enucleation with careful clinical and radiographic monitoring was sufficient for the treatment of these lesions.
A study analyzed 123 cases of AF reported in the literature. More than 90% of the cases were primarily treated conservatively, and only 10 patients were treated in a radical manner with marginal resection or segmental resection due to the extensive tumor size. With an average follow-up of 64.1 years in the study, AF showed a recurrence rate of 33.3% and a recurrence-free period of 33.2 months. The recurrence-free period was significantly higher in patients treated with radical procedures than conservative procedures (P = 0.0348). Malignant transformation occurred in 11.4% of AF recurrence cases. Patients younger than 22 years during the primary manifestation of the tumor were less susceptible to malignant transformation as compared to patients above this age (P = 0.0081).
Most patients were treated conservatively with enucleation and curettage (86%), and only few were treated with radical surgery using marginal resection or segmental resection. Radical surgery was related to extensive lesions, with the majority cases of this procedure (71%) used in people over 22 years of age. AF has a recurrence rate of 16.3%, where the recurrence occurs in 61% of patients below 22 years of age and 39% of patients over 22 years of age. In the case reported, the patient was 11 years old and conservative surgery was performed. No recurrence was reported.
The prosthetic rehabilitation of the atrophic posterior mandibular alveolar ridge after the treatment of jaw injuries is complex and often requires surgical procedures before rehabilitation. In the present case, the patient was treated conservatively with low morbidity and showed 100% improvement in IAN sensitivity and excellent esthetic and functional results.
| Conclusion|| |
The treatment for AF by curettage in young patients is effective in the management of these lesions and has the advantages of reduction of surgical morbidity, preservation of the IAN, and subsequent rehabilitation of the patient in an esthetic and functional way.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
This study was financed in part by the Coordenação de Aperfeiçoamento de Pessoal de NívelSuperior - Brasil (CAPES) - Finance Code 001.
Conflicts of interest
There are no conflicts of interest.
| References|| |
Buchner A, Vered M. Ameloblastic fibroma: A stage in the development of a hamartomatous odontoma or a true neoplasm? Critical analysis of 162 previously reported cases plus 10 new cases. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:598-606.
Muller S, Parker DC, Kapadia SB, Budnick SD, Barnes EL. Ameloblastic fibrosarcoma of the jaws. A clinicopathologic and DNA analysis of five cases and review of the literature with discussion of its relationship to ameloblastic fibroma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995;79:469-77.
Torroni A, Marianetti TM, Romandini M, Gasparini G, Cervelli D, Pelo S. Mandibular reconstruction with different techniques. J Craniofac Surg 2015;26:885-90.
Abayev B, Juodzbalys G. Inferior alveolar nerve lateralization and transposition for dental implant placement. Part I: A systematic review of surgical techniques. J Oral Maxillofac Res 2015;6:e2.
Osterne RL, Brito RG, Alves AP, Cavalcante RB, Sousa FB. Odontogenic tumors: A 5-year retrospective study in a Brazilian population and analysis of 3406 cases reported in the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;111:474-81.
da Silva LP, Serpa MS, Tenório JR, do Nascimento GJ, de Souza-Andrade ES, Veras-Sobral AP. Retrospective study of 289 odontogenic tumors in a Brazilian population. Med Oral Patol Oral Cir Bucal 2016;21:e271-5.
Wright JM, Vered M. Update from the 4th
edition of the World Health Organization classification of head and neck tumours: Odontogenic and maxillofacial bone tumors. Head Neck Pathol 2017;11:68-77.
Philipsen HP, Reichart PA, Praetorius F. Mixed odontogenic tumours and odontomas. Considerations on interrelationship. Review of the literature and presentation of 134 new cases of odontomas. Oral Oncol 1997;33:86-99.
Chen Y, Wang JM, Li TJ. Ameloblastic fibroma: A review of published studies with special reference to its nature and biological behavior. Oral Oncol 2007;43:960-9.
Costa DO, Alves AT, Calasans-Maia MD, Cruz RL, Lourenço Sde Q. Maxillary ameloblastic fibroma: A case report. Braz Dent J 2011;22:171-4.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]