Transcript of "Basal cell ameloblastoma a rare case report and review of literature"
BASAL CELL AMELOBLASTOMA: A RARE CASE REPORT AND REVIEW OF LITERATURE*Fatema Saify **Nidhi SharmaAbstract:Ameloblastomas are an enigmatic group of oral tumors. The name implies a resemblance tocells of the enamel-forming organ. The general agreement that ameloblastomas areodontogenic in origin occurs largely on the basis of the histologic similarities of the tumorand the developing enamel organ. Basal cell ameloblastoma is believed to be the raresthistologic subtype. It is reported to occur primarily in peripheral location but has been seenintraosseously, albeit rarely. Till date only 6 cases of Basal cell Ameloblastoma has beenreported in literature out of which five were in 3rd – 4th decade and only one case wasreported in 2nd decade. Numerous cases of ameloblastomas have been reported in adults inthe literature. However, only a few articles discuss ameloblastomas in children andadolescents. Considering the rarity of the lesion, we report here an interesting and uniquecase of Basal cell ameloblastoma of the mandible occurring in a very young patient.Key Words: odontogenic tumors, Basal cell ameloblastoma, acanthomatous ameloblastoma,granular cell ameloblastomaIntroduction accounts for 1% of lesions . WHOOdontogenic tumors comprise of a defines it as a locally-invasive polymorphiccomplex group of lesions of diverse neoplasia that often has a follicular orhistopathologic types and clinical plexiform pattern in a fibrous stroma. Itsbehaviour.1. Of all swellings of the oral behavior has been described as beingcavity, 9% are odontogenic tumors, and benign but locally aggressive2within this group, ameloblastomaOral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
Ameloblastomas are benign locally in children differ from adults, with aaggressive, polymorphic neoplasms of higher percentage of unicystic tumors5proliferating odontogenic epithelial origin. Basal cell variant of ameloblastoma is theThe incidence, clinical & radiological least common type. These lesions arefeatures, behavior and histopathology of composed of nests of uniform baseloidameloblastomas have been extensively cells and histopathologically very similarreviewed in numerous publications. to basal cell carcinoma of skin. No stellateSeveral histopathologic patterns of reticulum is present in the centre portionameloblastomas are commonly described of the nest. The peripheral cells aroundand include the follicular, plexiform, the nest tends to be cuboidal rather thanacanthomatous, granular cell and basal cell columnar.1patterns. There appears to be rather Though very little information appears ingeneral agreement that these variations in literature either due to insufficient numberhistopathology patterns do not have any of cases reported or due to variations insignificant bearing on prognosis except the clinical or radiological criteria, theunicystic ameloblastoma because of less pathologist may sometimes fail toaggressive behavior and favorable differentiate it from intraoral basal cellprognosis.3 carcinoma3Ameloblastomas in young people (ie, The purpose of this article is to present athose 19 years old and younger) are case of rarest variant of ameloblastomathought to be rare. They account for (Basal cell ameloblastoma) that hasapproximately 10% to 15% of all reported occurred in an unusual age and to providecases of ameloblastoma.4Ameloblastomas a brief review of literature.Case Report no history of trauma, sinus opening or pusA 12 year old male was referred to the discharge. Extraoral examination revealedgovernment dental college and hospital, facial asymmetry due to swelling on rightRaipur in with the chief complaint of side of the face extending from 2cmpainless swelling in relation to right lower anterior to ear till corner of mouthmandibular 2 nd and 3rd molar region. Past anteroposteriorly. Superioinferiorly, themedical, dental & family history of the swelling extended from infraorbitalpatient was non contributory. There was margin till inferior border ofOral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
mandible.(Fig 1) Clinical examination were all normal. From these clinical andrevealed firm to bony hard swelling in the radiographic findings a possible diagnosisright mandibular region with normal of ameloblastoma or odontogenicoverlying skin. No lymphadenopathy on keratocyst was made. To obtain a specificpalpation was noted. Intra-oral diagnosis an excisional biopsy was done asexamination revealed obliteration of segmental resection of right mandible andbuccal sulcus in the region of 43,44,45,47. the lesion was diagnosed as basal cellHistory of extraction of 46 was given ameloblastoma.during incisional biopsy, the details ofwhich are not available. Poor oral hygiene Microscopy:was noted. There was no intra oral sinus H & E stained sections showed lesionalor ulceration.( Fig 2) tissue composed of uniform baseloid cells. The peripheral cells were columnar withInvestigations: reverse polarity. In few islands, cuboidalRadiographic report revealed multilocular cells were also seen at the periphery. Noradiolucency with root resorption in stellate reticulum was seen in the centralrelation to 43,44,45,47. The inferior portion of the follicles. At one place theseborder of mandible was not traceable nests, tends to form a net like pattern withfrom 43,44,45,47. region. Third molar was interconnecting strands. The lesionalseen in the right ramus area Left side of tissue was covered by fibrous capsule withmandible appears normal. .(Fig.3) fibrous septa intervening into it giving it aHematological investigations [CBC, ESR] lobular pattern. (Fig 4,5,6,7)Fig 1 Extraoral swelling on the right sideof the face.Oral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
Fig 4 The lesional tissue covered by fibrous capsule with fibrous septa intervening into it giving it a lobular pattern (4x)Fig 2 Intraorally obliteration of buccalsulcus in the region of 43,44,45,47 is seen Fig 5 Baseloid cell island (10x)Fig 3 OPG reveals multilocularradiolucency with root resorption inrelation to 43,44,45,47 Fig 6 . The peripheral cells were columnar with reverse polarity(40x)Oral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
Fig 7 uniform baseloid cells with nostellate reticulum seen in the centralportion of the follicles (40x)Discussion basal cell subtype were available for validAmeloblastoma is chiefly a lesion of statistical analysis but recurrence wasadults. It occurs predominantly in 4th – 5th reported. 7decade of life and the age range is very According to published articles on thisbroad6. The average age of patients with variant, 6 cases have been reported inintraosseous ameloblastoma has been literature out of which five were in 3rd –reported to be 39 years2 The rare lesions 4th decade and only one case was reportedoccurring in children are usually cystic and in 2nd decade.8,9,10,11 The average incidenceappear clinically as odontogenic cysts. In of basal cell ameloblastomas was found tothis study, we document the occurrence of be less than 3%.ameloblastoma in a significantly younger Basal cell ameloblastoma tends to grow inage group. an island like pattern. The characteristicThe basal cell ameloblastoma is a rare color gradation seen in othervariant of ameloblastoma, which shows a ameloblastomas is often difficult toremarkable resemblance to basal cell appreciate in the basal cell subtype,carcinoma and published cases of intraoral because baseloid appearing cells ratherbasal cell carcinoma most likely are basal then stellate reticulum like appearing cellscell ameloblastomas. Too few cases of occupy the central portion of the tumorOral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
islands. The baseloid cells tends to stain ability to produce marked deformitydeeply basophilic and are nearly equivalent before leading to serious debilitationin staining intensity with the peripheral probably account for its early recognitionlayer of cells. The cells in the central .Thus we conclude on the availableportion may be polyhedral to spindle information that basal cell ameloblastomashape but stellate reticulum like areas are needs a perfect diagnosis based not onlynotably absent. The typical cellular on clinical and radiographical appearancemorphology and nuclear orientation of but also on histopathological findings.the peripheral cells often are altered. They The lesion in the present case deviatestend to be low columnar to cuboidal and from the usual ameloblastoma in terms ofoften do not demonstrate reverse nuclear age and histological appearance. Thereforepolarity with subnuclear vacuole when the diagnosis in young peopleformation. However, hyperchromatism remains in doubt after clinical andand palisading of the nuclei normally are radiologic examination, a biopsy isretained. 7 necessary. Long term follow-up at regularHistorically ameloblastoma has been intervals after surgery is alsorecognized for over a century and a half. recommended.Its frequency, persistent local growth andReferences analysis of 71 cases Oral Surg Oral Med Oral1. Neville BW, Damn DD, Allen CM, Pathol Oral Radiol Endod 2001;91:649-Bouqout JK,editors. Oral and maxillofacial 653)pathology. 2nd ed. Philadelphia; WB Saunders 5. Ord RA, Blanchaert RH Jr, Nikitakis NG,Co, 2002;.610-615. Sauk JJ. Ameloblastoma in children. J Oral2. Lagares DT, Cossío PI ,José M, Pérez JUL Maxillofac Surg. 2002 Jul;60(7):762-70;Med. oral patol. oral cir.bucal(Ed.impr.) discussion, 770-771.2005.10 ( 3 )Valencia mayo-jul. 6.Regezi, Sciubba, Jordan. editors. Oral3 . Desai H, Sood R, Shah R, Cawda J, Pathology Clinical Pathologic Correlations.4thPandya H. Desmoplastic ameloblastoma : Report ed. Saunders; 2003; 267-268 of a unique case and review of literature. Indian 7. Kessler HP Oral Maxillofacial Surg Clin NJ Dent Res 2006;17:45-49 Am 2004 ;16 (3): 311-315.4. Kim SG, Jang HS, Ju K . Ameloblastoma: 8. Kameyama Y, Takehana S, Mizohata M,A clinical, radiographic, and histopathologic Nonobe K, Hara M, Kawai T, Fukaya M IntOral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
A clinicopathological study of ameloblastomas J 11. Hirota M , Aoki S, Kawabe R , Fujita K.Oral Maxillofac Surg. 1987 Dec;16(6):706- Desmoplastic ameloblastoma featuring basal cell712. ameloblastoma: A case report Oral Surgery, Oral9. Kehinde FA, Vincent IU, Ganiat OO, Medicine, Oral Pathology, Oral Radiology, andKIzito CN, FAdekemi OO. Clinicopathological Endodontology February 2005;99( 2) :Pagesanalysis of histological variants of ameloblastoma 160-164.in a suburban Nigerian population Head &Face Medicine 2006, 2:42;1-8.10. Ide F, Shimoyama T, Horie N. Basaloidsquamous cell carcinoma versus basal cellameloblastoma Oral Oncology 34 (1998) 154. *SENIOR LECTURER, DEPT OF ORAL PATHOLOGY, GOVT. DENTAL COLLEGE, RAIPUR ** SENIOR LECTURER, DEPT OF ORAL PATHOLOGY, GOVT. DENTALCOLLEGE, RAIPUR..Oral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225