Open Access
Numéro
J Oral Med Oral Surg
Volume 24, Numéro 1, Janvier 2018
Page(s) 6 - 10
Section Article original / Original article
DOI https://doi.org/10.1051/mbcb/2017027
Publié en ligne 25 mai 2018

© The authors, 2018

Licence Creative Commons
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 cited.

Introduction

Ameloblastoma is the most frequent benign but locally aggressive odontogenic tumor of epithelial origin, representing about 14% of the jaw tumors and cysts [13]. In Africa, it afflicts predominantly patients between the third and the fourth decades of life [4,5]. Histologically, the tumor is classified in a diversity of subtypes of which solid/multicystic/conventional and unicystic ameloblastomas are the most frequent [6,7]. Surgical resection with wide margins of healthy tissue is the mainstay of the current treatment with proven efficacy [8]. Such an approach may result in functional and esthetical impairments as usually mutilating whereas the midface reconstruction is challenging. Additionally, postoperative recurrences of ameloblastoma are common with some of the recurrent tumors lethal [9]. Likely due to the rarity of maxilla ameloblastoma whose highest reported frequency is 25.7% of all ameloblastomas [10], papers dealing with the treatment of this tumor are scarce in the literature. This article aimed to report the oral functions, visual function, facial morphology and tumor recurrence in 11 patients after maxillary ameloblastoma surgery.

Patients and methods

The medical records of 11 patients who underwent surgical treatment for maxilla histologically confirmed ameloblastoma from 1994 to 2012 in two referral hospitals in Burkina Faso (CHU Souro Sanou and CHU Yalgado Ouedraogo) are retrospectively reviewed. On clinical and radiological grounds, the tumor excision was performed under general anesthesia via bucco-gingival sulcus incision with or without paralateronasal cutaneous incision, according to the lesion extent. Bone reconstruction consisted in immediate non-vascularized bone grafting. The graft was harvested from the iliac crest or the calvaria and fixed with screws and plates. In teeth loss after partial maxillectomy, patients were proposed a conventional dental prosthesis. Four patients had this rehabilitation. Histological diagnosis of ameloblastoma was established post operatively, on the surgical specimen, in accordance with the practice in management of clinically benign tumors or cystic lesions in Burkina Faso. Patients' postsurgical follow-up was done as long as possible and consisted in clinical examination and computed tomography scan. It was possible in 8 patients, for a period ranging from 2 to 12 years.

The data collected included the tumor localization and extent, the surgical approach (radical resection versus conservative surgery), the surgical defect extent, the post-operative oral functions, visual function and facial morphology, the tumor eventual recurrence and the time of its onset. Anterior maxilla referred to localization between the two maxillary canines. Posterior maxilla referred to localization behind the canine. Radical resection consisted in the tumor removal with healthy margins confirmed at the histological examination. Conservative surgery referred to the tumor enucleation with or without the margins curettage.

The surgical defect extent was graded according to the Cordeiro and Santamaria maxillary defects classification system [11]: type 1 maxillectomy defect was defined as that consecutive to loss of one or two maxilla walls, excluding the palate; type 2A maxillectomy defect was that which resulted from resection of the lower five walls and less than 50% of the palate with preservation of the orbital floor; type 2B maxillectomy defect referred to that resulting from loss of the lower five walls and more than 50% of the palate with preservation of the orbital floor; type 3 maxillectomy defect referred to that which resulted from loss of all the six maxillary walls; type 4 maxillectomy defect was defined as that consecutive to resection of the orbital contents and the maxilla walls, excluding the palate.

Oral functions referred to mastication after dental rehabilitation and elocution. They were broadly scored as satisfactory (ability of mastication, normal elocution) or unsatisfactory (difficulty of mastication, disturbance of elocution). Visual function was evaluated by presence of diplopia after type 3 maxillectomy. Facial morphology was evaluated by presence of enopthtalmos after type 3 maxillectomy and facial asymmetry irrespective of the maxillectomy type.

Results

Facial morphology was satisfactory in 7 patients who had all the tumor removal by a type 1 or 2A maxillectomy and surgical wound closure (Tab. 1). Out of these patients, 4 who had dental rehabilitation by a conventional prosthesis presented satisfactory mastication. Four patients subjected to the tumor removal by a type 3 maxillectomy had all facial asymmetry (Fig. 1). Out of these, 1 patient who did not have the orbital floor defect repair presented diplopia and enophtalmos, 2 patients subjected to palate defect repair by a prosthetic obturator or oral mucosa had elocution impairment. The tumor recurrence was noted in 2 patients after tumor enucleation and in 1 patient after radical surgery out of 8 patients who had a postoperative follow-up.

Table 1

Tumor extent, surgery modalities, results and patient outcomes.

thumbnail Fig. 1

Facial asymmetry in a patient with orbital floor bone grafting and prosthetic obturator after a type 3 maxillectomy.

Discussion

As in the treatment of any benign tumor, one of the goals of maxillary ameloblastoma surgery is the cure by performing the tumor complete removal. To achieve this, authors mostly recommend the tumor excision 1–2 cm beyond its radiological limits as presence of the tumor cells is reported 0.8–12 mm beyond these limits [12,13]. Such an approach implies sacrifice of a variable extent of osseous tissue involving the alveolar ridge, the hard palate, the maxillary sinus walls and even sometimes the orbital floor [10,12,14] but sparing usually the tumor overlying skin and mucosa. In type 1 and even type 2A maxillectomy defects, closure of the surgical wound using the tumor overlying soft tissue may enable to achieve satisfactory oral functions and facial morphology. Type 3 maxillectomy defects commend tissue transfer or prosthesis for imperative reconstruction of the orbital floor, the hard palate, the maxillary arch and the cheek as key functional or morphological structures of the midface. The best results in face substance loss repair are achieved currently thanks to vascularized composite bone-containing free flaps of which fibula, scapula, iliac crest, and radial forearm flaps [15,16]. Alternatives of these means whose technology is hardly available in underserved setting are non-vascularized bone grafts and prostheses. Despite the myriad of maxillary reconstruction options, results of extensive maxillectomy repair may be fairly good or even vexing. This is particularly true where non-vascularized bone grafting and prostheses use are the only available options of facial reconstruction. Propensity of ameloblastoma to recur even after a proper surgery is another frustration in this neoplasm treatment. Rates of recurrence higher after conservative surgery and reaching up to 90–100% support the limited role of this approach in the current management of ameloblastoma [9,17,18]. Carlson et al. declare not rational to treat conservatively a so aggressive lesion with intension to cure [8]. For these authors, the so-called tumor recurrence after such approach is in reality a progression of the lesion [8]. Some authors however advocate conservative surgery in pediatric patients arguing for the risk of facial growth compromise in radical surgery [1921]. Patient's wish is another indication of ameloblastoma conservative surgery. In this way, Sachs et al. report that some patients would consent for conservative and iterative surgery rather than a radical mutilating excision and defect repair [22]. In some patients of this study, decision of ameloblastoma enucleation comes from misdiagnosis given lack of histological examination of the neoplasm prior to its removal. That treatment should be followed by a complementary excision of the tumor operative site margins as Chapelle et al. recommend after enucleation of unicystic ameloblastoma with mural invasion [17]. Choice may be challenging between radical surgery resulting potentially in alteration of quality of life and conservative surgical approach with unavoidable recurrence of ameloblastoma. In this dilemma, our opinion is that in a setting of poorly compliance of patients for post-operative follow-up such as Africa, recurrence should be regarded as the major consideration. With respect to that consideration, Effiom et al. recommend aggressive radical surgery whenever possible [7]. Whatever the surgical modality, ameloblastoma has a potentiality for recurrence commending a strict post-surgical follow up of the ameloblastoma patient. As a recurrent tumor course may be for a long time clinically asymptomatic, there is an evidence that modern imaging including computed tomography and magnetic resonance plays currently a key-role in earlier diagnostic of the recurrences. Feinberg and Steinberg recommend tomography 2–3 weeks, 6 months then every year, postoperatively [14] while Chapelle et al. recommend it every year during the 5 first years postoperatively then every two-year during 25 years [17] supporting lack of standardization in the timing of the follow-ups.

Conclusion

In setting of patient's poor post-surgical follow-up, wide excision with negative margins combined to a suited facial reconstruction should be the treatment of maxillary ameloblastoma whenever possible.

Conflicts of interest

The authors declare that they have no conflicts of interest in relation to this article.

References

  1. Lasisi TJ, Adisa AO, Olusanya AA. Appraisal of jaw swellings in a Nigerian tertiary healthcare facility. J Clin Exp Dent 2013;5:42–47. [CrossRef] [Google Scholar]
  2. Bassey GO, Osunde OD, Anyanechi CE. Maxillofacial tumors and tumor-like lesions in a Nigerian teaching hospital: an eleven year retrospective analysis. Afr Health Sci 2014;14:56–63. [CrossRef] [PubMed] [Google Scholar]
  3. Oginni FO, Stoelinga PJ, Ajike SA, Obuekwe ON, Olokun BA, Adebola RA, et al. A prospective epidemiological study on odontogenic tumours in a black African population, with emphasis on the relative frequency of ameloblastoma. Int J Oral Maxillofac Surg 2015;44:1099–1105. [CrossRef] [PubMed] [Google Scholar]
  4. Nitassi S, Boulaadass M, Tobi I, Essakali L, Kzadri M. Améloblastome : diagnostic et traitement. A propos de 26 cas. Med Buccale Chir Buccale 2009;15:93–100. [CrossRef] [Google Scholar]
  5. Ba B, Singaré DK, Diallo M, Coulibaly AD, Théra TD, Keital K, et al. L’améloblastome mandibulaire : à propos de 51 cas. Med Buccale Chir Buccale 2016;22:7–11. [CrossRef] [Google Scholar]
  6. Dhanuthai K, Chantarangsu S, Rojanawatsirivej S, Phattarataratip E, Darling M, Jackson-Boeters L, et al. Ameloblastoma: a multicentric study. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:782–788. [CrossRef] [PubMed] [Google Scholar]
  7. Effiom OA, Ogundana OM, Akinshipo AO, Akintoye SO. Ameloblastoma: current etiopathological concepts and management. Oral Dis 2017 2018;24:3:307–316. [Google Scholar]
  8. Carlson ER, Marx RE. The Ameloblastoma: primary, curative surgical management. J Oral Maxillofac Surg 2006;64:484–494. [CrossRef] [PubMed] [Google Scholar]
  9. Nastri AL, Wiesenfeld D, Radden BG, Eveson J, Scully C. Maxillary ameloblastoma: a retrospective study of 13 cases. Br J Oral Maxillofac Surg 1995;33:28–32. [CrossRef] [PubMed] [Google Scholar]
  10. Milman T, Lee V, LiVolsi V. Maxillary Ameloblastoma with orbital involvement: an institutional experience and literature review. Ophthal Plast Reconstr Surg 2016;32:441–446. [CrossRef] [PubMed] [Google Scholar]
  11. Cordeiro PG, Santamaria E. A classification system and algorithm for reconstruction on maxillectomy and midfacial defects. Plast Reconstr Surg 2000;105:2331–2346. [CrossRef] [PubMed] [Google Scholar]
  12. Zwahlen RA, Gratz KW. Maxillary ameloblastomas: a review of the literature and of a 15-year database. Craniomaxillofac Surg 2002;30:273–279. [CrossRef] [Google Scholar]
  13. Tortorici S, Difalco P, Buzzanca ML, Burruano F. Management of primary ameloblastoma of the jaw: a 15 years' experience. Minerva Stomatol 2012;61:175–182. [PubMed] [Google Scholar]
  14. Feinberg SE, Steinberg B. Surgical management of ameloblastoma: current status of literature. Oral Surg Oral Med Oral Pathol 1996;81:383–388. [CrossRef] [Google Scholar]
  15. Dalgorf D, Higgins K. Reconstruction of the midface and maxilla. Curr Opin Otolaryngol Head Neck Surg 2008;16:303–311. [CrossRef] [PubMed] [Google Scholar]
  16. Cordeiro PG, Chen CM. A 15-year review of midface reconstruction after total and subtotal maxillectomy: part I. Algorithm and outcomes. Plast Reconstr Surg 2012;129:124–136 [CrossRef] [Google Scholar]
  17. Chapelle KA, Stoelinga PJ, de Wilde PC, Brouns JJ, Voorsmit RA. Rational approach to diagnosis and treatment of ameloblastomas andodontogenic keratocysts. Br J Oral Maxillofac Surg 2004;42:381–390. [CrossRef] [PubMed] [Google Scholar]
  18. Pogrel MA, Montes DM. Is there a role for enucleation in the management of ameloblastoma? Int J Oral Maxillofac Surg 2009;38:807–812. [CrossRef] [PubMed] [Google Scholar]
  19. Al-Khateeb T, Ababneh KT. Ameloblastoma in young Jordanians: a review of the clinicopathologic features and treatment of 10 cases. J Oral Maxillofac Surg 2003;61:13–18. [CrossRef] [PubMed] [Google Scholar]
  20. Huang IY, Lai ST, Chen CH, Chen CM, Wu CW, Shen YH. Surgical management of ameloblastoma in children. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:478–485. [CrossRef] [PubMed] [Google Scholar]
  21. Hong J, Yun PY, Chung IH, Myoung H, Suh JD, Seo BM, et al. Long-term follow up on recurrence of 305 ameloblastoma cases. Int J Oral Maxillofac Surg 2007;36:283–288. [CrossRef] [PubMed] [Google Scholar]
  22. Sachs SA. Surgical excision with peripheral ostectomy. A definitive, yet conservative approach to the surgical management of ameloblastoma. J Oral Maxillofac Surg 2006;64:476–483. [CrossRef] [PubMed] [Google Scholar]

All Tables

Table 1

Tumor extent, surgery modalities, results and patient outcomes.

All Figures

thumbnail Fig. 1

Facial asymmetry in a patient with orbital floor bone grafting and prosthetic obturator after a type 3 maxillectomy.

In the text

Les statistiques affichées correspondent au cumul d'une part des vues des résumés de l'article et d'autre part des vues et téléchargements de l'article plein-texte (PDF, Full-HTML, ePub... selon les formats disponibles) sur la platefome Vision4Press.

Les statistiques sont disponibles avec un délai de 48 à 96 heures et sont mises à jour quotidiennement en semaine.

Le chargement des statistiques peut être long.