Journal of Cranio-Maxillo-Facial Surgery 39 (2011) 615e618

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Journal of Cranio-Maxillo-Facial Surgery

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Review

Delayed soft tissue recurrence after treatment of ameloblastoma in a black African: Case report and review of the literatureq
Ezekiel Taiwo Adebayo*, Benjamin Fomete, Emmanuel Oladepo Adekeye
Etomie Oral and Maxillofacial Surgery Specialist Hospital (Medical Director: Prof. E.O. Adekeye BDS, FDSRCS, FWACS, FMCDS, FICS), Shehu Ibrahim Close, Malali, Kaduna, Nigeria

a r t i c l e i n f o
Article history: Paper received 25 May 2009 Accepted 14 May 2010 Keywords: Ameloblastoma Soft tissue Recurrence Radical resection

a b s t r a c t
Introduction: Ameloblastoma is rare worldwide. Delayed treatment can result in signicant morbidity from facial deformity and inltration of adjacent tissues. Mortality can occur from invasion of vital structures in the head and neck, super infection, recurrent and even distant metastases. Recurrence after radical treatment is not common. Case report: This paper presents a case of soft issue recurrence in the chin 21 years after radical resection of the mandible for ameloblastoma. The iliac crest bone grafted to the site was not involved in the tumour recurrence. Conclusion. Radical surgical resection is accepted treatment for solid/multicystic ameloblastoma. However, the surgical review should be for life as recurrence can occur after a long interval. 2010 European Association for Cranio-Maxillo-Facial Surgery.

1. Introduction Ameloblastoma is a benign, locally aggressive, slow growing neoplasm of the jaws and surrounding tissues (Abu-El-Naaj et al., 2008). It accounts for 1% of all oral tumours. In the latest classication of odontogenic tumours and allied cysts published for the WHO, Barnes et al. (2005) described the tumour as histologically benign, of odontogenic epithelial origin and characterised by odontogenic epithelium with mature brous stroma without odontogenic ectomesenchyme. Despite its benign histological appearance, ameloblastoma is clinically persistent, disguring and can kill from invasion of vital structures, super-infection, recurrence or distant metastases. Ameloblastoma can occur at any age, but most cases are seen between the 3rd and 5th decades (Olaitan et al., 1998; Abu-El-Naaj et al., 2008; Eckardt et al., 2009). The tumour does not show a sexual predilection. Among 47 patients, the female to male ratio was 1.1:1 (Eckardt et al., 2009). The main histological types of ameloblastoma are follicular and plexiform with subtypes acanthomatous, spindle cell, granular cell, basal cell and desmoplastic (Waldron and El-Mofty, 1987; Reichart et al., 1995; Kovcs et al., 1999). While surgical treatment is the acceptable form of treatment, Eckardt et al. (2009) note that opinions vary as to how radical it should be. However, adequate therapy necessitates a compromise

between the least destructive treatment possible for a benign tumour and a suitably radical method for preventing a recurrence (Eckardt et al., 2009). Recurrent ameloblastoma accounts for between 9 and 26% of all cases of the tumour seen (Olaitan et al., 1998; Abu-El-Naaj et al., 2008; Eckardt et al., 2009). Forty-seven patients were seen by Eckardt et al. (2009) between 1982 and 2003. Of these, 12 (26%) were cases of recurrence. Since the last report on recurrent ameloblastoma from Kaduna, Nigeria (Olaitan et al., 1998), to our knowledge no other has been presented. In that series, the longest interval before recurrence was 13 years. In view of the poor response to follow-up visits in our environment, we present a case of soft tissue recurrence 21 years after radical resection of a mandibular ameloblastoma. We review the literature in the light of this case with emphasis on the need for prolonged follow-up after treatment of ameloblastoma using any modality. 2. Case report A 55-year-old black African man was seen in the oral and maxillofacial specialist hospital in December 2008, following advice from relatives for assessment of the teeth that supported his denture. In 1987, when the patient was 34 years old, he was diagnosed with an ameloblastoma of the mandible extending from 35 to 47. Segmental resection from the right angle of the mandible to 36 was performed. The remaining teeth were preserved. Six months after surgery, iliac crest bone reconstruction of the defect was done, followed by sulcoplasty to retain a lower partial denture.

q Sources of grants: nil. * G.P.O. Box 3338, Kaduna 800001, Nigeria. Tel.: 234 803 3309149. E-mail address: taiwo_adebayo@yahoo.com (E.T. Adebayo).

1010-5182/$ e see front matter 2010 European Association for Cranio-Maxillo-Facial Surgery. doi:10.1016/j.jcms.2010.05.010

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E.T. Adebayo et al. / Journal of Cranio-Maxillo-Facial Surgery 39 (2011) 615e618

Fig. 1. Photograph of patient showing bulge in chin due to recurrent ameloblastoma.

Fig. 3. Photomicrograph of section showing peripheral ameloblastoma occurring in skin (400) using haematoxylin/eosin stain.

Fig. 2. Plain postero-anterior radiograph showing intact bone graft 21 years after resection of jawbone for ameloblastoma.

At the new referral, the patient complained of pain and mobility of the abutment teeth supporting the denture. On examination, a chin swelling was noticed which he said had been present for 6 months but did not bother him as the beard present made it aesthetically acceptable (Fig. 1). His medical history was unremarkable. Examination revealed that he was moderately obese, has a blood pressure of 180/90 mmHg. Plain radiographs revealed the iliac crest bone graft placed about 21 years ago was still intact and not involved with the swelling as shown in Fig. 2. A suspicion of soft tissue recurrence of ameloblastoma was made. Aspiration yielded a straw-coloured liquid from the chin swelling. The lesion was excised with the involved soft tissue and skin under general anaesthesia and forwarded for histology. Post-operative antibiotics and analgesics were administered and the post-operative course was uneventful. The patient was discharged from hospital on the 7th day post surgery and followed up every fortnight for 3 months. There have been no further complaints since the last review visit. One of the histology slides (Fig. 3) showed pigmented skin overlying a cystic lesion surrounded by abundant bro-hyaline stroma with foci of lymphocytic inltrates. The cysts are lined by ameloblastic epithelium. At higher magnication, a multicystic follicular ameloblastoma in soft tissue (Fig. 4) was diagnosed. 3. Discussion Ameloblastoma can present at any age but most cases are seen between the 3rd and 5th decades of life. Waldron and El-Mofty

Fig. 4. Photomicrograph of section of soft tissue ameloblastoma showing multicystic lesion using haematoxylin/eosin stain (400).

(1987) report that 83% of cases of ameloblastoma occur in the mandible, of these, 61% are seen in the molar/ramus area. Our patient was 34 years old at his initial presentation, which is within the peak decades, and the tumour was in the incisor, canine and premolar regions of the mandible. There are various clinical, radiological and histological types of the tumour. Clinically, there are solid/multicystic, unicystic and peripheral forms Gardner (1996). While the peripheral type is rare, they can be primary or secondary soft tissue tumours. The secondary tumours appear after attempted surgical removal (Gurol and Burkes, 1995). In sub-Saharan Africa, solid/multicystic types are more frequent than the unicystic type. The reverse proportion is observed in Latin America (Ledesma-Montes et al., 2007). There are unilocular, multilocular and polycystic without clear-cut distinguishing features seen radiologically. Associating the various types with recurrence, the unicystic ameloblastoma is reported to be less aggressive than the solid/ multicystic type (Gardner and Corio, 1983; Olaitan and Adekeye, 1997). In the work of Ueno et al. (1989), follicular ameloblastoma gives rise to more recurrences than the plexiform type while the unicystic is more common in the younger age group than in older patients.

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It has not been possible to retrieve the earlier histology report of this case but the soft tissue recurrence was solid/multicystic, follicular. Ueno et al. (1986) and El-Mofty et al. (1991) had observed that peripheral ameloblastoma is more often of the plexiform type than follicular. Recently, Leiser et al. (2008) found that ameloblastoma weakly expresses cancerous molecular markers such as p53, MDM2 from the cytoplasm of its reverse polarised cells. The strongly expressed marker was heparanase. Further research is necessary to understand the prognostic signicance of these markers and their relevance to treatment outcome. However, there is insufcient data to associate histological typing, adequate treatment and rates of recurrence (Eckardt et al., 2009). Gardner and Pecak (1980) found that conservative treatments such as curettage and enucleation result in up to 90% rate of recurrence. Curi et al. (1997) have suggested that they can be treated by enucleation followed by liquid nitrogen spray cryotherapy to decrease local recurrence. While unicystic ameloblastomas do well after curettage and enucleation treatment, solid/ multicystic forms are treated radically by resection in our environment and in several other parts of the world. In view of the large sizes of the lesions seen in sub-Saharan Africa, the high likelihood of soft tissue involvement by tumour and the late presentation, we agree with Kovcs et al. (1999) that radical resection is absolutely indispensable in the proper management of these cases; discussions about its necessity are detrimental for the patient. This was recently borne out by the nding that recurrence was higher (60%) after conservative surgical treatment than after radical surgery (20%) from the work by Eckardt et al. (2009). Radical surgical treatment by resection of ameloblastoma gives better results. Instances of recurrence emphasise the need for prolonged follow-up reviews after treatment. Hayward (1973) and Collings and Harrison (1993) have reported recurrence after 30 and 49 years, respectively. While the primary lesion presented by Hayward (1973) had been treated conservatively, that from Collings and Harrison (1993) was initially treated by resection of the involved } ller and Slootweg (1985), most cases of jawbone. According to Mu recurrent ameloblastoma are seen within 5 years of therapy. Reichart et al. (1995) believe that recurrences develop 2.3 years before they are diagnosed. In the series earlier reported by Olaitan et al. (1998), more than 80% of recurrent cases were seen within 5 years of treatment with the longest being after 13 years. The case we are presenting recurred after 21 years. The patient claimed the chin swelling had been present for only 6 months. Considering the insidious nature of the growth of ameloblastoma, the growth may have started earlier. It is likely that as the life expectancy of the population in sub-Saharan Africa continues to increase, greater duration before recurrence will be observed. Histologically benign ameloblastoma can recur at the primary site or in distant organs and tissues. In the classication by Barnes et al. (2005), the latter are called malignant ameloblastoma. Primary recurrence can be in the soft tissue or in the bone used to replace the defect. Soft tissue recurrence as in the case presented is not common. Graft et al. (1970), Stea (1985), Vasan (1995), Choi et al. (2006) and Eckardt et al. (2009) have reported recurrences in bone grafts while Collings and Harrison (1993) and Marx et al. (1993) presented soft tissue recurrences. Among 21 cases of recurrent ameloblastoma reported by Olaitan et al. (1998), only four (19%) were in the soft tissue while the rest 17 (81%) were in bone. In a later multi centre analysis by Arotiba et al. (2007), only four (13.3%) of 30 cases of recurrent ameloblastoma were in soft tissue. Choi et al. (2006) have recorded the complaints of patients with soft tissue recurrence as including ill tting dentures, erythema of the intraoral mucosa and swelling. Our patient had these except that the mucosa was not erythematous while the teeth supporting

the denture were mobile. Tooth mobility could be due to the fact that there were few teeth supporting an almost full lower denture and not necessarily associated with tumour recurrence. Radiographs showed that the bone graft was completely intact, uninvolved by tumour recurrence and this was proven at surgery. Stea (1985) postulates that primary recurrence may be from the stump of the jawbone left after resection, from adjacent soft tissue or from intra-operative seeding of tumour during resection. In view of the wide soft tissue exposure necessary for resection of some tumours and the fact that the recurrence in this case occurred far away from the bone, intra-operative seeding of tumour cells is the most likely cause of this case of recurrence. Some authors have proposed radiotherapy for some forms of ameloblastoma. Akanime (1988) cited a study that showed radiotherapy resulted in 42% recurrence rate. Gardner (1988) believes it should be reserved for inoperable lesions while Pinsolle and Michelet (1995) combined surgery with radiotherapy (50 Grays) for mandibular recurrences when the rst surgery was inadequate; for all recurrences and when there were positive surgical margins after wide resection. Olaitan et al. (1998) managed recurrences as for primary lesions with care and conscious effort to prevent further recurrence. Such measures include delivering the specimen wholly and the excision of suspicious skin. The case reported here was managed by excision of the entire soft tissue tumour and the involved skin. Early cases of recurrent ameloblastoma are asymptomatic. However, they can destroy the bone graft and spread to involve soft tissue. The benets of life-long follow-up after treatment should be emphasised to all patients with radiographs taken to augment clinical examination. Recurrences can only be reduced by individualising treatment, based on tumour site, extent and histology. 4. Conclusion Ameloblastoma is a slow growing, benign but locally aggressive, odontogenic tumour of epithelial origin. It occurs in peripheral and central forms with two main histological types. For the more common multicystic/solid ameloblastoma, radical surgical resection is the accepted treatment modality. Adequate treatment must be individualised based on history, examination, histology and radiological characteristics. However, the post-surgical review should be for life as recurrence can occur after a long time. References
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