Giant ossifying fibroma

J. N. Khanna, N. N. Andrade Department of Maxillofacial Plastic and Oral Surgery, Nair Hospital and Dental College, Bombay, India

Case report on a bimaxillary presentation J. N. Khanna, N. N. Andrade." Giant ossifying fibroma. Case report on a bimaxillary presentation. Int. J. Oral Maxillofac. Surg. 1992; 21." 233-235. Abstract. Ossifying fibroma is a slow-growing, benign neoplasm, but some lesions behave aggressively, reaching massive proportions, thus demanding special treatment. The following case report holds particular interest, because of the simultaneous occurrence of an active ossifying-cementifying fibroma in the maxilla and mandible with the maxillary lesion attaining enormous size.

The fibro-osseous lesions of the jaws comprise a diverse, but challenging group of conditions. Common to all fibro-osseous lesions is the replacement of normal bone by a tissue composed of collagen fibers and fibroblasts that contains varying amounts of mineralized substance, which may be bony or cementum-like or an admixture of both. Although MENZEL in 1842, 23 first described the entity known as ossifying fibroma, it w a s MONTOGOMERY23 w h o in 1927 coined the term "ossifying fibroma". Subsequent to the introduction of the term "fibrous dysplasia" by LICHTENSTEIN 12 in 1938 it was suggested that lesions of the jaws previously designated as fibrous osteoma or ossifying fibroma should be called fibrous dysplasias.

Fig. 1. Pre-operative, frontal view reveals involvement of the maxilla and mandible.

Recently, this trend appears to be reversing and at present most pathologists consider the 2 as separate and distinct clinical entities. It has been shown that there are characteristic histological, radiographic 5 and clinical features that assist in the differentiation between these 2 fibro-osseous lesions. Ossifying fibroma, has been designated as a wellcircumscribed, generally slow-growing benign lesion, with expansile, sharply defined margins often with a radiolucent peripheral component 3'14'19'22. The etiology of ossifying fibroma remains unknown. Trauma-induced stimulation of progenitor cells has been suggested by WENIG et al. 24 but to date this has not been substantiated by other authors. It has been suggested, however, that those ossifying fibromas associated with trauma seem to behave more aggressively than the typical benign lesion usually of moderate size. HAMNER et al. 7'8 in a comprehensive analysis of 249 cases of fibro-osseous lesions of the jaws, suggested that ossifying fibroma should be considered as a tumour of periodontal membrane origin arising from multipotent mesenchymal blast cells, present in the periodontal membrane, that have the capacity to produce cementum, alveolar bone and fibrous tissue. This concept is supported by the work of CHO et al. 4 who demonstrated that the periodontal membrane (PDM) fibroblast is capable of transforming into hard tissue-producing cells. Although no definition of giant ossifying fibromas is to be found in the literature HAMNER et al. 8 arbitrarily defined giant fibro-osseous lesions as those exceeding 2 x 2 cm in size. SHeRMAN • STERNBERG16 reported 12 cases that qualify as giant forms. HEERDEN

Key words: ossifying fibroma; giant ossifying fibroma, multiple presentation. Accepted for publication 25 March 1992

et al. 19 reported a clinico-pathological study of 8 giant ossifying fibromas. The literature also contains isolated cases of large aggressive forms of these lesions 1'7'14.A t present, most authors believe that the large aggressive lesions of either ossifying or cementifying nature vary little with regard to the age or sex of the patient and the location 11,21,22. Reports of cemento-ossifying fibroma as being more aggressive than either the pure cementoid or osteoid type of lesions does not seem to be borne out in subsequent reviews) 3'15'17 This paper reports on a patient who presented with 2 cemento-ossifying fibromas involving the maxilla and mandible.

Fig. 2. PA mandible view shows cortical expansion and a diffuse calcification of the right maxilla and left body of the mandible.

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•Fig. 3. Frontal view on 7th postoperative day.

Case report A 33-year-old male patient was referred to the O M F department with the chief complaint of a giant painless swelling of 3 years duration involving the maxilla. It had rapidly increased in size in the past 3 months. He also had a swelling involving the left body and angle of the mandible (Fig. 1). Past medical and family history was non-contributory. Examination revealed a large, hard, nontender swelling on the right side o f the maxilla and on the left body of the mandible. The maxillary swelling which extended superiorly to the orbital region had pushed the eye cranio-laterally. Inferiorly it extended to the corner of the mouth, medially to the frontonasal suture obliterating the lateral nasal fold and deviating the nares to the left. The swell-

ing reached to the ears and measured approximately 12 cm x 10 cm. A similar hard swelling was seen involving the body of the mandible on the left side, approximately 5 cm x 4 cm in size (Fig. 1). The skin over both swellings had a normal colour but was stretched. The right eye seemed to function reasonably well although vision was blurred but the eye movements were normal. Intraorally, the maxillary alveolus on the right side revealed considerable expansion, obliterating the buccal sulcus. It involved the hard palate beyond the midline and extended posteriorly to the soft palate. The patient was edentulous from the canine backwards with deep indentations on the maxillary swelling due to the opposing teeth. Considerable expansion of the buccal and lingual cortices was also present on the left side of the body mandible. A routine radiographic examination included anterior-posterior and lateral views. It revealed large lesions in the maxilla and body of the mandible. The lesions contained diffuse calcifications (Fig. 2). In the maxilla significant posterior and superior extension was seen with involvement of pterygoid plates, nasal fossa and orbital floor. In the left mandible was considerable cortical expansion with "bending" of the inferior border. Alkaline phosphatase, serum calcium and other routine investigations were all within normal limits. A biopsy was performed on both the maxillary and mandibular tumour masses. The diagnosis ossifying-cementifying fibroma was made for both lesions. The patient was treated under general anaesthesia. The maxillary tumour was exposed via a Weber Fergusson approach. The tumour was seen to have dissected its way through lines of least resistance. It had involved the maxilla, zygoma and the maxillary sinus and had extended well into the nasal cavity. The orbital floor was destroyed. Posteriorly, the tumour had extended into the pterygopalatine fossa, where it had to be

Fig. 4. Photomicrograph showing clear demarcation between fibroosseous tissue (right side) and adjacent cortical bone (left side) (H & E x 125).

carefully dissected. Otherwise the tumour, owing to its capsule, was easily shelled out from the surrounding tissues. A split thickness skin graft was used to cover the defect. The mandibular tumour mass was exposed via a submandibular incision; the tumour was well encapsulated and was extirpated by blunt dissection. A Kirshner wire was primarily used to maintain the contour of the face. The specimens were sent for histopathological examination. It was found that the tumour was covered by a thin layer o f cortical bone (Fig. 4). The composing spindle cells were slender and grouped into bundles and whorls. The nuclei were spherical or oval in shape with low chromatin content. Mitoses were occasionally observed. The tumour contained numerous, well-defined, bizzareshaped bone particles and occasional mineralized structures most of which were spheroid (Fig. 5). Some spherical, lamellar mineralized bodies were also found together with osteoid rimmed, intensely stained bony structures that had only a few cells. The definitive diagnosis of ossifying-cementifying friboma was made. Postoperative ocular mobility was surprisingly good and there was no apparent weakness or diplopia (Fig. 3). The patient was discharged from the hospital after 12 days. The patient was followed up periodically at 3 monthly intervals. An obturator designed to fit the maxillary defect was made. The patient was last seen after a period o f 1 year, when he was in apparent good health, but was subsequently lost to follow up.

Discussion T h e initial a s s e s s m e n t o f o u r case w a s disconcerting. The rapid expansion of the t u m o u r m a s s was c o n s i d e r e d a sign of poor prognosis. The suspicion of mal i g n a n c y was i n c r e a s e d b y t h e d e s t r u c tion seen in t h e o r b i t a l floor, t h e n a s a l

Fig. 5. Photomicrograph showing highly cellular fibrous stroma with various types of mineralized structures (H & E x 125).

Giant ossifying fibroma obstruction and epiphora. Rapid growth, paresthesia, pain, t o o t h m o bility a n d r e s o r p t i o n are also c o m m o n l y associated w i t h osteogenic s a r c o m a 2s. In the p r e s e n t case, overt pain was a b s e n t a n d the p a t i e n t was e d e n t u l o u s in the region involved. R a d i o g r a p h s d e m o n strated the m a g n i t u d e of the lesions, b u t did n o t p e r m i t distinction between b o t h ossifying f i b r o m a a n d oesteogenic sarcoma. Surgery was less complex t h a n was expected a n d h a e m o r r h a g e was far less t h a n anticipated. Despite its size, it was possible to r e m o v e the t u m o u r easily f r o m the o r b i t a l a n d nasal regions. I n the p t e r y g o p a l a t i n e fossa, the fibrous capsule was lacking a n d the t u m o u r h a d to b e carefully dissected out. T h e diagnosis o f ossifying-cementifying f i b r o m a was justified by the clear d e m a r c a t i o n between t u m o u r a n d s u r r o u n d i n g structure including cortical bone. A l t h o u g h , the literature c o n t a i n s n u m e r o u s r e p o r t s o n giant ossifying a n d cementifying fibromas, 3'1°'14'17'19,2°'23only 3 cases o f multiple occurrence o f this lesion h a v e b e e n reported. 2'9'18

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Address:

Prof. J. N. Khanna Department of Oral and Maxillofacial Surgery Nair Hospital Dental College Dr A. L. Nair Road Bombay-400 008, lndia

Giant ossifying fibroma. Case report on a bimaxillary presentation.

Ossifying fibroma is a slow-growing, benign neoplasm, but some lesions behave aggressively, reaching massive proportions, thus demanding special treat...
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