JIndianSocPedodPrevDent302179-1553084_041850

179JOURNAL OF INDIAN SOCIETY OF PEDODONTICS AND PREVENTIVE DENTISTRY | Apr - Jun 2012 | Issue 2 | Vol 30 |

Case RepoRt

Mandibular mass in pediatric patient: A diagnostic dilemma

Ranadive PA, Deshpande MD, Ingole SN, Ankush C1

Department of oral surgery, Nair Hospital Dental College, Mumbai, 1Government Dental College, Mumbai, India

Correspondence: Asst. Prof. P.A. Ranadive, 107, 1st floor, Nair Hospital Dental College, A. L. Nair Road, Mumbai Central, Mumbai- 400008., India. E-mail: [email protected]

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DOI: 10.4103/0970-4388.100013

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Abstract

A case of aggressive Juvenile ossifying fibroma in a child has been discussed. The unique appearance of this particular lesion demonstrates a close relationship between aggressive ossifying fibroma and well-differentiated osteogenic sarcoma. The age of the patient and the location of the tumor in the case presented made it mandatory to utilize multiple diagnostic parameters and adjunctive diagnostic help to resolve the dilemma posed by this tumor. Differentiation of ossifying fibroma from other benign and malignant neoplasms as well as fibrous dysplasia is important in correct management of the lesion.

Key words

Aggressive Juvenile ossifying fibroma

Introduction

In 1872, Menzel first described the entity known as ossifying fibroma,[1] although it can resemble focal cement-osseous dysplasia radiographically and to a certain extent histopathologically ossifying fibroma is a true neoplasm with significant growth potential.[2] Its affliction to the jaws can be related to the vast amount of mesenchymal cellular induction that is required in this region for odontogenesis; the possibility of induction or genetic alteration leading to a neoplasm is therefore greater.[3]

They expand slowly and asymptomatically, and characteristically behave in a benign fashion. Occasionally they may occur in a gigantic form exhibiting an aggressive nature although none have been known to metastasize.[4]

This tumor is mainly found in the tooth bearing areas, with women in their twenties and thirties being commonly affected; however, the younger and older men are also affected.[3]

The term “juvenile ossifying fibroma” has been introduced as a less common andmore aggressive variant that occurs primarily in children and young adults; such lesions have a rapid growth rate with local aggressive tendencies, occasionallywith growth mimicking sarcomas. [5] Histologically two patterns of Juvenille ossifying fibroma are accepted (1) trabecular (2) psammomatoid. Both varieties reveal similar radiographic features and growth patterns, the trabecular form is diagnosed initially in young patient. Both patterns occur in either jaws but reveal a maxillary predominance. Psammomatoid variant frequently appears outside the jaws with over 70% arising in the orbit, frontal bones, and para nasal sinuses.[2]

Case Report

A 5-year-old girl reported to the Department of Oral and Maxillofacial Surgery, Nair Hospital Dental College with a chief complaint of swelling in the anterior region of the mandible. Patient’s mother first noticed the swelling that was progressively

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Ranadive, et al.: Pediatric tumor

180 JOURNAL OF INDIAN SOCIETY OF PEDODONTICS AND PREVENTIVE DENTISTRY | Apr - Jun 2012 | Issue 2 | Vol 30 |

enlarging for 3 months. There was no history of trauma to that area, no pain associated with the mass, no tooth pain or mobility and she also denied numbness to the lower lip. The patient’s medical and dental histroy was non contributory. The swelling was firm and nontender, palpable over the chin and the submental region. Minor discomfort was elicited on palpation; there were no palpable cervical lymph nodes, bruits nor pulsations were demonstrated.Intraorally a firm nontender swelling was seen extending from 72–82 in the buccal vestibule. There was no lingual cortical expansion. Displacement of teeth were seen [Figure 1].

Radiographic examinationOPG and CT scan showed an irregular radiopaque lesion over the symphysis region with intermittent radiolucent areas and ill defined peripheries. There was no evidence of any root resorption, but permanent dentition showed displacement [Figures 2-4].

Subsequent courseAn incisional biopsy was taken under General anesthesia from the submental region [Figure 5].The histologic diagnosis given was osteogenic sarcoma. Because this did not correspond to the surgeons’ clinical evaluation, the specimen was sent for a second opinion to two different places. The diagnosis given was desmoplastic fibroma and a rhabdomyosarcoma. As two out of three histopathology reports were suggestive of malignancy, incisional biopsy was repeated at one of the referral centers. This specimen was subjected to histochemical studies and was found negative to Desmin,MyoD1, CD34, ruling out embryonic rhabdomyosarcoma, spindle cells were negative for S100, SMA, CD34. The final diagnosis made based on the above-mentioned findings was Juvenile ossifying fibroma. A locally aggressive tumor necessitating excision.Based on clinical and radiographic findings, correlated with the histologic appearance, it was considered that the lesion was an aggressive yet probably benign fibro-osseous lesions. The mass was then excised in Toto. The lingual cortex of the mandible appeared to have

Figure 2: Preoperative orthopantomogram

Figure 3: Preoperative axial view CT scan Figure 4: Preoperative coronal view CT scan

Figure 1: Preoperative intraoral view




181JOURNAL OF INDIAN SOCIETY OF PEDODONTICS AND PREVENTIVE DENTISTRY | Apr - Jun 2012 | Issue 2 | Vol 30 |

Figure 6: Postoperative intraoral viewFigure 5: Intraoperative tumor exposed

been eroded superficially due to the pressure of the growing lesion. The patients postoperative course was uncomplicated. Patient was kept under observation for clinical and radiographic features of recurrence. One year postoperatively, the patient has remained asymptomatic [Figure 6].Root development is normal. Radiographs of the patient showed complete filling of the defect with normal bone and without any evidence of recurrence [Figure 7].

Pathologic diagnosisThe final diagnosis was Juvenile ossifying fibroma.

Discussion

In evaluating the aggressive forms of ossifying fibroma, it is important not only to carefully examine the histologic appearance of the tumor but to strongly consider the clinical radiographic and surgical presentation in attempting to differentiate it from a possible malignant entity. Several investigators have reported the problems associated with the differentiation between ossifying fibroma and osteogenic sarcoma.[1,6,7] The patient described

earlier provided the same diagnostic dilemma. Occurrence of rapid growth, paresthesia, pain, and tooth mobility are common features of osteogenic sarcoma.[8,9] In our initial assessment of the patient, the only clinical features that was of concern was the apparent rapi d rate of growth. Although this finding is not a typical characteristics of ossifying fibroma, their presence in the more aggressive form is acknowledged. [1,7,10] Radiographically the lesion exhibited local aggressiveness, a diffuse scattering and calcification. Although these characters are representative of both the aggressive ossifying fibroma and osteogenic sarcoma. The existence of well-defined sclerotic borders and displacement of the tooth structures rather than resorption supported a diagnosis of a benign lesion.Appearance of the tumor during surgery probably represented the most significant support for a benign diagnosis. The tumor mass was easily shelled out and erosion of the cortical plates was seen. Perforation of cortical plates that often is a feature of potentially malignant lesion were not noted. These characteristics in combination with clinical and radiographic findings influenced the decision to consider this a benign lesion.The histologic features exhibited by the lesion also generated a distinct difference of opinion regarding whether it was a benign or malignant . Most sections had microscopic appearance that satisfactorily filled the basic criteria for diagnosis of ossifying fibroma. The cellular soft tissue component had no significant vascularity or pleomorphism and was not particularly anaplastic in appearance. In addition the mitotic index in sections was low.Despite the unusual histologic presentation the classification of this lesion as an aggressive ossifying fibroma is appropriate and consistent with previously reported cases. As is typical of many

Figure 7: Postoperative orthopantomogram




182 JOURNAL OF INDIAN SOCIETY OF PEDODONTICS AND PREVENTIVE DENTISTRY | Apr - Jun 2012 | Issue 2 | Vol 30 |

fibroosseous lesions, proper differentiation between the various pathologic entities is often an exercise in judgment.

References

1. Walter JM Jr, Terry BC, Small EW, Matteson SR, Howell RM. Aggressive Ossifying fibroma of Maxilla: Review of literature and report of case. J Oral Surg 1979;37:276-86.

2. Drad W Neville, Douglas Damm, Carl Allen, Jerry Bouquot. Oral and Maxillofacial pathology second edition saunders 2004.

3. Oral and Maxillofacial pathology a rationale for diagnosis and treatment Robert E. Marx, Diane Stern. Quintessence publishing Co,Inc.

4. Reaume CE, Schmid RW, Wesley RK. Aggressive ossifying fibroma of mandible. J Oral Maxillofac Surg 1985;43:631-5.

5. Sciubba JJ, Younai F. Ossifying fibroma of mandible and maxilla: Review of 18 cases. J oral Pathol Med 1989;18:315-21.

6. Hamner JE 3rd, Scofeild HH, Cornym J. Benign fibro-osseous jaw lesions of periodontal membrane origin.An analysis of 249 cases. Cancer 1968;22:861-78.

7. Small IA, Goodman PA, Giant cemento ossifying fibroma of maxilla: Report of case and discussion. J Oral Surg 1973;31:113-9.

8. Cocke WM Jr, Wade WM Jr. Osteogenic Sarcoma of the Mandible. Oral Surg Oral Med Oral Pathol 1970;30:601-6.

9. Wilcox JW, Dukrat RC, Kolodny SC, Jacoby JK. Osteogenic Sarcoma of mandible: Review of literature and report of a case J. Oral Surg 1973;31:49-52.

10. Hamner JE 3rd, Lightbody PM, Ketcham AS, Swerdlow H. Cemento-ossifying fibroma of the maxilla. Oral Surg Oral Med Oral Pathol 1968;26:579-87.

How to cite this article: Ranadive PA, Deshpande MD, Ingole SN, Ankush C. Mandibular mass in pediatric patient: A diagnostic dilemma. J Indian Soc Pedod Prev Dent 2012;30:179-82.

Source of Support: Nil, Conflict of Interest: None declared.

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JIndianSocPedodPrevDent302179-1553084_041850