ameloblastic fibroma to 9 year old

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Journal of Cranio-Maxillary

Diseases

Volume 2 Issue 1 January-June 2013

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ISSN: 2278-9588

Journal of Cranio-Maxillary Diseases / Vol 2 / Issue 1 / January 201380

An aggressive ameloblastic fibroma in a 9‑year‑old child treated with buccal fat graft

Amina Sultan, Dayashankara Rao JK1, Harsha Jain2

Department of Pediatric and Preventive Dentistry, Faculty of Dentistry, Jamia Millia Islamia, New Delhi, 1Department of Oral and Maxillo Facial Dentistry, S.G.T. Dental College and Hospital, Gurgaon, Haryana, 2Department of Oral and Maxillo

Facial Surgery, Sharda Dental College, Greater Noida, Uttar Pradesh, India

ABSTRACT

Ameloblastic fibroma is a relatively uncommon neoplasm of odontogenic origin which is characterized by simultaneous proliferation of both epithelial and mesenchymal tissue without the formation of enamel and dentin. It is a slow‑growing tumor and commonly located in mandibular molar area, often over an unerupted tooth. It is usually diagnosed in the second decade of life. Its occurrence in posterior maxilla is a rare or uncommon entity and only few cases have been reported so far in children. The management of such lesions in maxilla is to avoid any disfigurement and at the same time not compromising its complete removal as it has tendency to recur and possibility of malignant change into ameloblastic fibrosarcoma. This report is a documentation of a case, of a 9‑year‑old female child with an aggressive ameloblastic fibroma of posterior region of maxilla. The patient has been followed up for 2 years; the outcome has been functionally and aesthetically satisfactory and there have been no signs of recurrence.Keywords: Ameloblastic fibroma, buccal fat pad graft, surgical excision

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DOI:

10.4103/2278-9588.113583

Correspondence to:Dr. Dayashankara Rao JK, Department of Oral and Maxillo Facial Dentistry, S.G.T. Dental College and Hospital, Farukh Nagar Road, Budhera, Gurgaon, Haryana, India. E‑mail: [email protected]

in association with an unerupted tooth or tooth migration or displacement.[2‑5]

It will frequently cause no complaint but pain tenderness or mild swelling of the jaw may induce the patient to seek aid from the dentist.[1]

Nevertheless its importance lies in its tendency to recur and in the possibility of malignant change into ameloblasticfibrosarcoma.[7]

This report is a documentation of a case of a 9‑year‑old female child with clinically aggressive AF with an atypical localization (posterior region of maxilla) treated with surgical excision followed by closure of the defect with the buccal fat pad.

CASE REPORTA 9‑year‑old girl child reported to the department

of Pedodontics, SGT Dental college, Gurgaon,

INTRODUCTIONThe ameloblastic fibroma (AF) is a relatively

uncommon neoplasm of odontogenic origin, which is characterized by simultaneous proliferation of both epithelial and mesenchymal tissue without the formation of enamel and dentin.[1,2]

AF tends to occur in younger patients. Most lesions are diagnosed within first two decades of life. This tumor is slightly more common in males than in females.[1,3‑6] The most common site of occurrence is the posterior site of mandible.[1‑3] It is a slow‑growing tumor, often asymptomatic and usually occurs

Case Report

Sultan, et al.: Ameloblastic fibroma, buccal fat graft

81Journal of Cranio-Maxillary Diseases / Vol 2 / Issue 1 / January 2013

with a complaint of a painless swelling on the right side of maxilla for the last2 years. The patient gave a history of a small nodule, on facial aspect of gingiva in posterior region that gradually increased up to the present size of 5‑6 cm over a period of 2 years. Intraoral examination presented a soft tissue lesion in the right maxillary posterior alveolar region extending from deciduous canine region to soft palate posteriorly and toward mid palatine area and also extraorally. The swelling was large, lobulated, nontender, and pedunculated and 6 × 2 cm in size. It was irregular in shape and soft in consistency. The overlying mucosa was normal and did not bleed on probing. No discharge or redness was present [Figure 1].

Extraoral examination revealed facial asymmetry with an edematous swelling on right side of cheek extending from maxillary right permanent canine to first permanent molar. Skin color was normal. She had no other symptoms and apparently in good health [Figure 2].

Patient gave a history of past treatment involving an incisional biopsy of that area; about 1 year back, at some local hospital along with the medical line of treatment (antibiotics).But the swelling increased in size considerably without any response to the given treatment.

A panaromic radiograph Orthopantamogram (OPG) showed a well‑circumscribed unilocular radiolucent lesion in right maxillary posterior region [Figure 3]. The radiolucency measured 4 × 2.5 cm and it was associated with displacement of second premolar, first and second permanent molars toward the periphery of the lesion (maxillary sinus).

Erupted teeth were not mobile and there was no sign of root resorption.

Based on clinical and radiographic findings, a presumptive preoperative diagnosis of dentigerous cyst and ameloblastic fibrosarcoma, was done.

Under naso endotracheal general anesthesia the complete lesion was excised and curetted via intraoral approach. The cut surface was grey white in appearance. Through horizontal incision along the buccal mucosa the buccal fat pad was released

and spread over the bony defect keeping its pedicle intact [Figure 4]. Primary closure of the oral mucosa along with the buccal fat graft was done by using 3‑0 vicryl [Figure 5]. Right side deciduous canine and lateral incisor were extracted prior to the excisional biopsy while first and second permanent molars were left untouched. After the whole lesion was removed

Figure 1: Intraoral picture showing the extension of the lesion crossing the midline of palate

Figure 2: Extraoral swelling with the diffuse swelling over the right side of the face

Figure 3: OPG radiograph showing soft tissue density lesion in the right maxilla



the resultant bony cavity was smooth and had an intact surface. The excised tissue mass was send for biopsy.

The specimens sent for the pathological examination were in two pieces from different sites of the lesion. The tissues taken were 4 × 2 × 1 cm and 2 × 1 × 6 cm in sizes.

The diagnosis of AFwas confirmed according to the histological findings [Figures 6 and 7].

The postoperative healing was uneventful and there was no incidence of any wound break down. There was no fistula formation. On follow‑up

examinations every month, there has been no evidence of recurrence after surgery [Figure 8]. Both the premolars have erupted into the oral cavity. The postoperative OPG taken after 3 months showed occlusal movement of the permanent molars and eruptive movement of permanent lateral incisor [Figure 9].

DISCUSSIONAmeloblastic fibroma is a true histological

biphasic tumor because the epithelium and mesenchymal component are part of neoplastic process. The two tissue components represent an early stage of odontogenesis before the formation of calcified structures of enamel and dentin.[8]

Figure 4: Complete surgical enucleation of the lesion and exposure of the buccal fat Figure 5: Primary closure of the huge defect with buccal

fat graft

Figure 6: H and E staining, A: Section showing odontogenic epithelium in form of cords and small islands with loosely arranged epithelial cells resembling stellate reticulum in the center and stellate cells in loose matrix 10 (× 4 magnification)

Figure 7: Odontogenic epithelium in form of islands with juxtra epithelial hyalinization of mesenchymal portion.10 × 10 magnification


83Journal of Cranio-Maxillary Diseases / Vol 2 / Issue 1 / January 2013

According to the revised WHO classification AF consists of odontogenic ectomesenchyme resembling the dental papilla and epithelial strands and nests resembling dentallamina and enamel organ. No dental hard tissues are present. Rarely, tumors with the histo morphology of AF may form dysplastic dentin, and are called ameloblastic fibrodentinomas (AFD).[9]

In the present case, the AF was diagnosed to be present in 9‑year‑old female child in the posterior region of maxilla. Ameloblastic fibromatends to occur in younger patient, with average age of patient being 14.6 years and with 40% of the patients under the age of 10 years.[10] The age limits are widespread i.e., from6 months to 42 years.[10,11] Also the sex predication varies from no preference[7,12] to males are more frequently affected than females.[5,10,11,13] Over 80% of the tumors occur in mandible, the usual site being the canine‑molar region. Only four cases of tumors in maxillary anterior region have been reported.[11,14]

Ameloblastic fibroma exhibits somewhat slower clinical growth than simple ameloblastoma and does not tend to infiltrate among trabeculae of bone. Instead it enlarges by gradual expansion so that periphery of the lesion often remains smooth.[1,14] The lesion may displace erupted teeth although the teeth may remain vital.[15]

In our patient, however, rapid growth was observed, probably due to small size of maxilla rather than the biological behavior of tumor.

Most patients present with a painless, slow‑growing and expanding tumor commonly associated with an unerupted tooth. Often the associated tooth is displaced in an apical direction indicating that the origin of lesion is superior to the tooth.[16] The patient, in this case also reported with an asymptomatic, rapidly expanding swelling on right side of maxilla in posterior region. Second premolar and first permanent molar were unerupted, while first premolar was partially erupted. Deciduous right lateral incisor was retained. The child’s medical history was unremarkable.

No constant significant differences between the appearance of the simple ameloblastoma and that of the AF are found radiographically.[1] The tumor appears as a well‑defined radiolucent image, usually unilocular, as in our case, which can be difficult to distinguish from other odontogenic or non‑odontogenic fibroma, odontogenic myxoma, fibrous dysplasia in the osteolytic stage and central giant cell granuloma. The presence of dental inclusions may give a false appearance of follicular cyst; and resorption of tooth roots is very rare.[7] On the basis of review by Trodahl, the radiographic lesion varied in size from 1 to 8.5cm in diameter. Most of the lesion (3:1) were multilocular; only the smaller ones were unilocular.[11] The OPG in this case revealed a large expansile‑ 4 × 2.5 cm well‑corticated unilocular radiolucent lesion located in region of posterior maxilla. The lesion had displaced first and second permanent molars into the maxillary sinus.

Microscopically AF is composed of young cellular mesenchymal tissue that resembles or duplicates the dental papilla of tooth germ. Within this young mesenchyme are thin strands and islands of odontogenic epithelium.[6] Analysis of sections of the lesion taken from our patient showed fragments

Figure 8: Three months post-op picture showing good healing with out any fistula

Figure 9: Three months post-op OPG with out any evidence of the disease



of tumor tissue composed of both mesenchymal and epithelial components. Epithelial cells were arranged in cords, nests, trabeculae, and strands of cuboidal to low columnar cells. No evidence of cellular atypia was seen.

All these histological findings suggested that it was a true mixed tumor.

Proper management of AF has been a recent topic of debate. Although, initially it was believed that AF was innocuous lesion that seldom recurred after simple local excision or curettage, but subsequent reports seemed to indicate a substantial risk of recurrence after conservative therapy.[3] As the lesion is well‑circumscribed recurrence are considered to be the result of inadequate initial removal of what are frequently multilocular lesions.[8] We opted for excision and curettage. A wide excision of the tumor has been recommended unless the extent of surgery would result in significant cosmetic deformity.[7] Other support enucleation or curettage as the initial treatment with a modified block resection reserved for recurrence.[11] In the present case the tumor was massive measuring 5‑6 cm. The location was posterior maxilla, in contrast to its usual site i.e. posterior mandible. It was intra osseous having perforated the buccal cortical plate to present as a soft tissue lesion, an indication of its aggressive nature. First permanent molar was left untouched. Clinically, eruption of unerupted and malpositioned teeth should be evaluated carefully. In this case also, both the premolars and permanent lateral incisor erupted spontaneously while permanent molars showed some drifting movement occlusally.

Although rare, malignant transformation can occur and long‑term follow‑ups is recommended.

Regardless of the form of treatment, patient with AF must be followed up for longer period to enable the early detection of possible recurrence or development of ameloblastic fibrosarcoma.[17]

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Odontogenic origin. In: Shafer's textbook of oral pathology. 5th ed. Philadelphia: Elsevier; 2006. p. 400‑2.2.

Tuna EB, Gulsum AK, Gencay K. Ameloblastic fibroma: A case report with five years follow‑up. Acta Stomatol Croat 2008;42:185‑91.

3. Neville Brad W, Damm Douglas D, Allen Carl M, Bouquot Jerry E. Odontogenic cysts and tumors. In: Oral and Maxillofacial Pathology. 2nd ed. Philadelphia, America: Saunders; 2004. p. 626‑7.

4. Akal UK, Gunhan O, Guler M. Ameloblastic fibrodentinoma. Report of two cases. Int J Oral Maxillofac Surg 1997;26:455‑7.

5. Mcguinness NJ, Faughnan T, Bennani F, Connolly CE. Ameloblastic fibroma of the anterior maxilla presenting as a complication of tooth eruption: A case report. J Orthod 2001;28:115‑8.

6. Bhaskar SN. Odontogenic tumours of the jaws. In: Synopsis of Oral Pathology. 7th ed. St. Louis: Mosby; 1986.p. 285‑6.

7. Junquera LM, Albertos JM, Floriano P, Calvo N, Santos J. Ameloblastic fibroma: Report of two cases. Int J Paediatr Dent 1995;5:181‑6.

8. Sapp JP, Eversole LR, Wysocki GP. Odontogenic tumors. In: Contemporary Oral and Maxillofacial Pathology. 2nd ed. Maryland Heights, Missouri‑America: Mosby; 2004. p. 155‑6.

9. Slootweg PJ. Ameloblastic fibroma/fibrodentinoma. In : World Health Organizat ion Class i f icat ion of Tumors (WHO)‑International Agency for Research on Cancer (IARC). Lyon, France: IARC Press; 2005.

10. Slootweg PJ. An analysis of the interrelationship of the mixed odontogenic tumors‑ ameloblastic fibroma, ameloblastic fibro‑odontoma, and the odontomas. Oral Surg Oral Med Oral Pathol 1981;51:266‑76.

11. Trodahl JN. Ameloblastic fibroma. A survey of cases from the armed forces institute of pathology. Oral Surg Oral Med Oral Pathol 1972;33:547‑58.

12. Pereira KD, Bennett KM, Elkins TP, Qu Z. Ameloblastic fibroma of the maxillary sinus. Int J Pediatr Otorhinolaryngol 2004;68:1473‑7.

13. Regezi JA, Sciubba JJ, Jordan RC. Odontogenic tumors. In: Oral Pathology‑ Clinical Pathologic Correlations. 4th ed. Philadelphia: Saunders Company Ltd.; 2003. p. 284‑6.

14. Takeda Y. A meloblastic fibroma and related lesions: Current pathologic concept. Oral Oncol 1999;35:535‑40.

15. Hegde V, Hemavathy S. A massive ameloblastic fibro‑odontoma of the maxilla. Indian J Dent Res 2008;19:162‑4.

16. Furst I, Pharoah M, Phillips J. Recurrence of an ameloblastic fibro‑odontoma in a 9‑year old boy. J Oral Maxillofac Surg 1999;57:620‑3.

17. Vasconcelos BC, Andrade ES, Rocha NS, Morais HH, Carvalho RW. Treatment of large ameloblastic fibroma: A case report. J Oral Sci 2009;51:293‑6.

How to cite this article: Sultan A, Rao JD, Jain H. An aggressive ameloblastic fibroma in a 9-year-old child treated with buccal fat graft. J Cranio Max Dis 2013;2:80-4.Source of Support: Nil. Conflict of Interest: None declared.Submission: May 04, 2012, Acceptance: December 11, 2012

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ameloblastic fibroma to 9 year old