|Year : 2014 | Volume
| Issue : 1 | Page : 17-24
Ameloblastoma: Our clinical experience with 68 cases
Benjamin Fomete1, Ezekiel Taiwo Adebayo2, Joseph Osamudiamen Ogbeifun1
1 Maxillofacial Unit, Ahmadu Bello University Teaching Hospital, Shika-Zaria, Kaduna State, Nigeria
2 Army Dental Centre, Bonny Cantonment, Lagos, Lagos State, Nigeria
|Date of Web Publication||15-May-2014|
P. O. Box 3772, Kaduna
Source of Support: None, Conflict of Interest: None
Introduction: In this environment, previous workers have reported on the challenges of managing large sized ameloblastoma of the jaws with less than adequate facilities. The aim of this review is to present the management of 68 cases of ameloblastoma with emphasis on surgical care. Materials and Methods: Retrospective survey of case notes of patients with histopathologic diagnosis of ameloblastoma (using the criteria of Barnes et al., 2005) seen between January 2006 and August 2010 at the Maxillofacial Unit, Ahmadu Bello University Teaching Hospital, Shika-Zaria, Nigeria was undertaken. Data collected includes histopathological diagnosis, age, gender, clinical information on site of lesion, form of intubation and surgical procedure performed. Results: Out of 94 patients, 68 with histological diagnosis of ameloblastoma (59 mandibular and 9 maxillary) were operated within the study period. The remainder (26) was not treated in hospital. Among 68 patients treated, more were males (38) than females (30), giving a male to female ratio of 1.3:1. The age range was between 14 and 74 years (mean-standard deviation). The duration of the symptoms ranged from 7 months to 24 years, most were follicular ameloblastoma (n = 13) followed by acanthomatous type (n = 7). Endotracheal intubation was the most common (n = 55) followed by fiber optic laryngoscopy (n = 8). The surgical approach most used was extended Risdon with intraoral (n = 24) followed by extended Risdon with lip split and intraoral (n = 17). Segmental resection (en block) formed the bulk of our procedures (n = 22) followed by subtotal mandibulectomy (n = 16). Conclusion: The treatment of ameloblastoma remains controversial. Its destructive nature has left patients with wide defects difficult to reconstruct.
Keywords: Ameloblastoma, reconstruction, surgery
|How to cite this article:|
Fomete B, Adebayo ET, Ogbeifun JO. Ameloblastoma: Our clinical experience with 68 cases. J Orofac Sci 2014;6:17-24
|How to cite this URL:|
Fomete B, Adebayo ET, Ogbeifun JO. Ameloblastoma: Our clinical experience with 68 cases. J Orofac Sci [serial online] 2014 [cited 2017 Mar 25];6:17-24. Available from: http://www.jofs.in/text.asp?2014/6/1/17/132570
| Introduction|| |
Ameloblastoma is a benign, locally aggressive, slow growing neoplasm of the jaws and surrounding tissues. , It accounts for 1% of all oral tumors. In the latest classification of odontogenic tumors and allied cysts published for the World Health Organization. Barnes et al.  described the tumor as histologically benign, of odontogenic epithelial origin and characterized by odontogenic epithelium with mature fibrous stroma without odontogenic ectomesenchyme. Despite its benign histological appearance, ameloblastoma is clinically persistent, disfiguring and can kill from invasion of vital structures, superinfection, recurrence or distant metastases. 
The lesion can occur at any age, but most cases are seen within the 3 rd and 5 th decades. ,, According to Eckardt et al., the tumor does not show a true sexual predilection with a female to male ratio of 1.1:1.  The main histological types of ameloblastoma are follicular and plexiform with subtypes as acanthomatous, spindle cell, granular cell, basal cell and desmoplastic. ,,, Most common site of involvement is the mandible (80%), although the maxilla (20%) may be afflicted. The lesion may present as small incipient lesion detected on radiological examination or as a large tumor producing mass effect. 
Although surgical treatment is the acceptable form of treatment, opinions vary as to how radical it should be. , However, adequate therapy necessitates a compromise between the least destructive treatment possible for a benign tumor and a suitably radical method for preventing a recurrence.  The challenges in the surgical management of this tumor are to provide complete excision as recurrence may occur in incomplete removal and also to reconstruct the bony defect in order to give reasonable cosmetic and functional outcome to the patient. 
In many developing countries, late presentation of patients with ameloblastoma is common resulting in difficulties in intubation. , This review of 68 patients treated for ameloblastoma is aimed at highlighting challenges in the management of this tumor in our environment.
| materials and methods|| |
Data for this study were obtained from retrospective survey of case notes of all patients with jaw tumors seen at the Maxillofacial Unit, Ahmadu Bello University Teaching Hospital, Shika, Zaria between January 2006 and August 2010. As the oldest referral center in Northern Nigeria, it receives patients from the North West and North Central geopolitical zones of Nigeria. The classification of odontogenic tumors according to Barnes et al., was used to select cases from histopathologic reports. Out of 126 cases of odontogenic tumors seen within the period, 94 (74.6%) were classified as ameloblastoma while 68 cases were operated. The rest cases of ameloblastoma were not treated in hospital due to absconding from hospital, inability to afford hospital fees and preference for traditional forms of care. Materials reviewed for each patient included demographics, clinical features of lesions, intraoral and extraoral radiographs, records of routine investigations, surgery notes on the method of intubation, operative procedure and post-operative instructions. Data collected were pooled for analysis.
| Results|| |
A total of 68 patients with ameloblastoma (59 mandibular and 9 maxillary) were operated within the period of January 2006 to August 2010 made up of 38 males and 30 females, male to female ratio was 1.3:1. The age range for male was 14-70 years with an average of 42 years and a mean of 35.3 years, for the female 14-60 years with an average of 37 years and mean of 32.3 years. The majority of patients were in the age range of 21-40 years with more males in the age range of 31-40 years. Of the 68 patients operated, details of their tumor duration were recorded. The range was 7 months to 26 years. Details of the clinical features of 68 operated cases of ameloblastoma are shown in [Table 1] and [Table 2]. Of the 68 patients, the majority had duration between 7 months and 5 years (40 or 60.6%), followed by 6-10 years (32.35%). 33 (48.52%) had a report of specific subtype of ameloblastoma while 35 (51.47%) were unspecified. The histopathologic subtypes of ameloblastoma are shown as [Table 3].
[Figure 1] and [Figure 2] showing the large size of some ameloblastomas seen in this series. Most of the 68 patients had general anesthesia administered using endotracheal intubation (n = 55, 80.9%) followed by fibreoptic laryngoscopy (11.8%). The relationship between the surgical approach of ameloblastoma and type of intubation is shown as [Table 4]. Surgical treatments performed were segmental resection (n = 22, 32.4%), subtotal mandibulectomy (23.5%) and maxillectomy (13.2%). Details of surgical treatments are in [Table 4] and [Table 5]. The various morbidities resulting from treatment of ameloblastoma are listed in [Figure 3] and [Figure 4]. All patients where required were put into intermaxillary fixation using eyelet wires.
|Figure 4: Our own way of securing the airway after anterior, subtotal and total mandibulectomy|
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| Discussion|| |
Ameloblastoma remains a rare tumor constituting 15% of oral neoplasms. In sub-Saharan Africa, many of the workers have presented data indicating that ameloblastoma accounts for the greater proportion of odontogenic tumors. ,, Using the classification of Barnes et al.,  ameloblastoma made up 74.6% of odontogenic tumors seen at this center. It is possible that better index of suspicion and improved facilities for diagnosis will complement the use of this new classification to enhance odontogenic tumor diagnosis in our region.
In general, ameloblastoma can present at any age, though most are seen within the 3 rd and 5 th decades of life.  In this study, the age range was 14-70 years, with the majority in the 3 rd to 5 th decade. Waldron and El-Mofty,  reported that 83% of cases of ameloblastoma occur in the mandible, of these, 61% are seen in the molar/ramus area. Our finding that 86.5% of 68 cases occurred in the mandible while the rest (13.5%) were in the maxilla was similar to that of Waldron and El-Mofty.  Cases of peripheral ameloblastoma are rare in this environment possibly due to low index of suspicion. A rare case of recurrent ameloblastoma in soft tissue was reported from another center in Northern Nigeria following earlier treatment at this study center.  In general, the sex distribution of ameloblastoma is almost equal according to Eckardt et al.  Among 47 patients they reported a female to male ratio of 1.1:1. In our study, we observed a female to male ratio of 1:1.3. Chukwuneke et al.  got 1.6:1 female to male ratio in Southeast Nigeria.
Generally, ameloblastoma presents as painless slow growing mass without significant functional impairment in the early stages of its development. According to Kahairi et al.,  it took about 2 years before their patient developed symptoms such as significant facial asymmetry, malocclusion and difficulty in chewing. This could explain why some patients give a very short history of the tumor meanwhile at surgery the tumor would be very extensive as shown in [Table 5]. In our clinical experience one patient presented late as the presence of jaw swelling [Figure 2] was been used for financial gain through begging. This may reflect the pervasive poverty in this part of Nigeria. The use of health for improper financial gain should be frowned at by the Nigerian society and eliminated through social education. Female patients present late in our environment because some of them use veil to mask the presence of the tumor as in [Figure 3].
The main histological types of ameloblastoma are follicular and plexiform with subtypes as acanthomatous, spindle cell, granular cell, basal cell and desmoplastic. ,,, As shown in [Table 3], follicular ameloblastoma (n = 13) was the most common, the relevance is somewhat diminished by the non-specificity of 51.47% of histopathologic diagnosis of ameloblastoma. Our center relies on general pathologists for histopathology due to paucity of oral pathologists in our environment.
Although the optimal surgical approach remains controversial, it remains the favored treatment modality in our center. Conservative surgical options include enucleation, curettage, cryotherapy, electrocautery, marsupialization, or any combination of the above while radical treatment involves various forms of resection. According to Philip et al.,  resection involves segmental or rim resection of the mandible or maxilla.  In this study, enucleation (8.8%) was the only conservative modality utilized. Various forms of resection were performed for the rest as shown in [Table 5]. Treatment form was selected based on the extent of the lesion and available facilities in our environment as previously reported by Olaitan et al.;  Adebayo et al.  Our treatment protocol is supported by Kahairi et al.,  who state that while unicystic ameloblastomas do well after conservative measures like curettage and enucleation treatment, solid/multicystic forms are treated radically by resection.
Surgical approach to the tumor was such as to give good exposure for complete tumor removal with low prospect of post-operative morbidity. In our center, for mandibular lesions, lip splitting alone or in combination with Risdon incision was done for 32% of patients. Derderian et al.,  have advocated combining Risdon with an intra-oral incision claiming it gives less post-operative morbidity and better cosmetic outcome. Shirani et al.  in a series of seven patients introduced a new technique of removal of large ameloblastoma with immediate reconstruction by using only an intra-oral incision. The stated advantages were tumor removal and repositioning of the mandible intra-orally, removal of the lesion and reconstruction procedure to be done simultaneously. Facial scar and damage to the marginal mandibular nerve that innervate the lips are also avoided. Our center used the technique of Shirani et al.,  16.2% of the treated patients. However, it was observed that tissue torquing and bleeding with more edema accompanied the technique.
Sahoo et al.  and Deepak and Roma  report that tumor growth distorts the facial contour leading to inadequate mask seal precluding proper bag-mask ventilation during induction. They also noted that adequate jaw thrust may not be possible while intraoral extension of the growth may not allow insertion of laryngoscope blade with the inability to visualize the vocal cords. Oral airway manoeuvres may result in aspiration due to bleeding from mucosa overlying the growth. To assist intubation in our patients, endotracheal (80.88%), fibreoptic (11.76%) and tracheostomy (4.41%) and blind intubation (2.94%) were utilized. Other methods of securing airway include blind nasal intubation, use of lightwand, transtracheal jet ventilation and tracheostomy are more traumatic to the patient.  The most widely used method was endotracheal (nasal mostly) even though with difficulties such as bleeding as a result of trauma to the mucosa. A few patients had orotracheal intubation, which was inconvenient in operating jaw tumors. Tracheostomy was rarely used (4.41%) due to its attendant problems even in the hands of experienced surgeons. This was the method mostly used by Chukwuneke et al.  with 27 out of 32 patients. According to Chukwuneke et al.,  tracheostomy has morbidity of 14-45% with a mortality rate of 2-16% while Amusa et al.  reports morbidity of laryngeal stenosis in 6.9% and mortality (25%). According to Gyoh and Emery,  there are circumstances peculiar to Northern Nigeria, which make tracheostomy itself a danger to life. During the dry season (October-March), there are strong, dry, dust-laden harmattan winds blowing from the Sahara desert and humidity is very low; cold and upper respiratory infections are common. The dust load in the atmosphere is so heavy on some days that it hangs like a thick fog, reducing visibility to a mere 100 m. Dryness of the skin, lips and throat is quite serious, and mucus becomes thick and tenacious. For the tracheostomy patient, mucus rapidly dries and cakes in the trachea; it may block the lumen, or reduce effective coughing and ciliary action.
In the immediate post-operative period the ability to maintain patency of the airway is challenged by the loss of the attachment of the muscles of the tongue (genioglossus) in cases of anterior, subtotal and total resections of the mandible.  They also believe that the newly reconstructed mandible with bilateral insufficient attachment of muscle weakens the floor of the mouth predisposing to the tongue falling backward. In cases with respiratory distress, jaw thrust cannot be applied effectively and edema of the airway may compound the airway problems. To avoid these, our center has the policy of keeping patients intubated in the post-operative period after anterior, subtotal and total mandibular resection until return to full consciousness is ascertained by the anesthesiologist. Meanwhile, a tongue stitch is inserted by passing a larger size >or = 1/0 silk or any other suture through the substance of the tongue at the junction of anterior two-third and posterior one-third then knotting it at about 2 cm away from the tongue. It can then be secured on the patients' cloth or on a syringe by opening the hub and tying round it. The hub is then pushed back and plaster is used to prevent it from pulling out. [Figure 4] shows a patient for whom this technique was utilized. Chukwuneke et al.,  have also advocated the use of Oji spatula to maintain patency of the airway.
The ultimate goal of the treatment of ameloblastoma is restoration of function and aesthetics. After treatment for ameloblastoma, reconstruction and rehabilitation are necessary. According to Chukwuneke et al.,  in the developing world there is a dearth of modern equipment and materials to achieve successful jaw reconstruction, especially in cases of total mandibulectomy. This is the situation in our center also where maxillectomy patients are reconstructed using only obturators. According to earlier authors, the procedures used for jaw reconstruction vary widely. Although some have practiced immediate reconstruction (primary reconstruction), others have delayed it (secondary reconstruction). Lawson et al.,  in 1982 found 48% overall success rate with primary reconstruction with most failure due to salivary leak. They also reported 90% of success rate if the procedure was delayed. Komisar  also found that secondary reconstruction had a higher rate of success. With the availability of reliable, single-stage reconstructive procedures and the often-practiced two-team approach, head and neck reconstruction can be performed expeditiously and with predictable outcome. Improved quality-of-life with primary reconstruction is now considered an overriding argument for primary reconstruction. Availability of high-definition anatomic and metabolic imaging studies also facilitates detection of local recurrence and counters the argument to delay reconstruction. 
Presently, secondary reconstruction has almost become obsolete with a few exceptions. In circumstances when there is a lack of microvascular surgical expertise or due to inherent patient factors, it may still be necessary to employ alternative methods of reconstruction and/or delay definitive reconstruction.  This is often the case in our environment where both the surgical expertise and equipment are lacking.
Different materials and methods have been used for the rehabilitation of patients after maxillectomy. In 1912, Scudder described the use of hard rubber prostheses to rehabilitate patients after having jaw tumor excision. Two percent of the patients in the literature had rehabilitation with dental prosthetic devices alone.  Today better materials and techniques extend the possibilities of patient rehabilitation. Wire mesh, acrylic in 1950's, polyurethane and silastic have been used. 
Recent notable advances in head and neck reconstruction include sensate free tissue transfer, osseo integrated implant and dental rehabilitation, motorized tissue transfer and vascularized growth center transfer for pediatric mandible reconstruction.  Previously, alloplasts such as spacers (Kirschner wires and Steinmann pins), gap-bridging appliances (mesh cribs) and reconstruction plates were used to prevent collapse of the overlying tissues. They were abandoned due to lack of stability, external extrusion and unacceptably high rate of infection. 
Though most of our patients needed jaw bone reconstruction, many refused another surgery for its placement. Their reason was inadequate financial capacity to afford surgery, most of them being peasant farmers and petty traders. Financial empowerment to ensure access to hospital care has become a serious issue in Nigeria. Despite the introduction of the National Health Insurance Scheme in 2006, access to hospital-based health services remains very poor. A likely reason is that the scheme only covers federal government employees presently. There is urgent need for pro-poor health-care financing scheme(s) in Nigeria to improve access. Compared with Chukwuneke et al.,  who had no reconstruction out of 32 cases, in this series 9 cases (8 maxillectomies and one mandibulectomy) were reconstructed using obturators for maxillectomy and bone graft (delayed reconstruction following subtotal mandibulectomy where the right rib was used). Presently, she is awaiting sulcoplasty. We also noted that due to the peculiarity of our environment, some patients find a way of covering their defect by either using a veil for female [Figure 5] or a turban for male patients. Some males also grow beards to cover the post-operative jaw defect. Patients with very large tumors that cause expansion of soft tissues after surgery, present with a little soft-tissue defect compare with those with a small tumor with a marked defect. The shrinkage of the remaining soft-tissues leaving them with a defect that is esthetically acceptable, although functionally inadequate [Figure 6].
Mastication is a learned, volitional and automatic process, which can often readjust following surgery. 
Subjective evaluation of masticatory function was assessed by asking patients to evaluate their ability to chew boiled corn, roasted corn and sugar cane. It was observed that patients who had less than segmental mandibular resection had better function than those with hemi mandibulectomy, who also performed better than those with segmental from angle to angle and total mandibulectomy. This agrees with Obiadazie et al.  who found that mastication was absent completely only in subtotal and total mandibulectomy patients. Incidentally, patients with IMF reported better functions than those not placed in IMF post-operatively, which agrees with obiadazie et al. 
Our patients experienced initial difficulties with swallowing in the immediate post-operative period up to a week. This however improved by the day and only one patient so far presented with poor swallowing. Obiadazie et al.  found four patients in whom swallowing was poor.
In our study, speech was found to better in patients with segmental, hemi-mandibulectomy than those with angle to angle and total mandibulectomy and maxillectomy. Those with maxillectomy had improved speech after the obturator was fitted in.
Drooling of saliva was observed in all the patients in the immediate postoperative period. They however regained control of salivary secretions within 7 days postoperatively and sometimes a little more. Our observation agrees with Obiadazie et al.  who also highlighted some of the reason for the leakage (inability of patients to swallow saliva as a result of trauma from endotracheal intubation, nasogastric tube discomfort, diminution of volume of oral cavity due to dentoalvealar and soft-tissues loss; and marked postoperative edema of the tongue and floor of the mouth).
Eckardt et al.  , reported that 26% of ameloblastomas seen are recurrences. Out of 68 treated cases seen in this series, there were 2 recurrences (2.9%).Patients were observed from 6 weeks post-operative to 5 years. The recurrences were observed 2 and 4 years after treatment. Mϋller and Slootweg found that most cases of recurrent ameloblastoma are seen within 5 years of therapy. Reichart et al. however believed that recurrences develop 2.3 years before they are diagnosed. According to Olaitan et al.,  80% of recurrences from our center occurred within 5 years though a report of recurrence after a period of 21 years is also available. 
| Conclusion|| |
Ameloblastoma even though benign, is very destructive and leaves patients who present late with large defect that require extensive reconstruction and expertise, which is still lacking in our environment. Decreasing financial strength also prevented patients from coming for reconstruction even though facilities not available in our center for the extensive ones.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
[Table 1], [Table 2], [Table 3], [Table 4], [Table 5]