PÁGINA DEL RESIDENTE

What should the diagnosis and treatment be?

¿Cuál sería su diagnóstico y su manejo terapéutico?

Female patient, 68 years old, presented with an ovarian mass suggestive of ovarian cancer. She needed to be operated urgently by the gynecological service in our hospital. The issue of an interconsultation was raised, as the patient had a large intraoral mass that made intubation impossible.

On examination, a mass was observed that occupied the entire palatal vault. It was pedicled to the gingiva of the alveolar crest by teeth 15 to 17. It measured 5x5 cm, it was pinkish and similar to the rest of the mucosa. It had a smooth surface, a fibroelastic consistency and it was closely interwoven with a removable partial prosthesis(Figs.1 y 2). The patient reported that it had been evolving for 10 years during which time it had been impossible to remove and clean the prosthesis. There were no other related symptoms.

Given the pressurized circumstances, and with the presumed diagnosis of benign neoplasm of the maxilla vs. mucosal hyperplasia, a decision was made to operate urgently.

Peripheral ossifying fibroma

Fibroma osificante periférico

E. Charro Huerga¹, I. Vázquez Mahía², G. Gómez Oliveira¹, S. Sironvalle Soliva¹, J.L. López Cedrún³

1 Médico Residente.
2 Médico Adjunto.
3 Jefe de Servicio.
Servicio de Cirugía Oral y Maxilofacial. Hospital Juan Canalejo. A Coruña, España

Correspondence

The patient underwent surgical excision of the lesion under local anesthesia based on clinical data and the examination. The excision of the lesion was carried out using an electric bistoury. The prosthesis was removed, exodontia of the relevant root remains was carried out together with curettage of the bony bed. The area was partially sutured with resorbable material (Fig.3).

The histological study of the specimen revealed the presence of microcalcifications and areas with a myxoid appearance (Figs.4). The microscopic analysis showed the existence of a mucosal lining with a flat, multilayered, non-keratinized epithelium. There were signs of reactive hyperplasia with papillomatosis and acanthosis. The stoma was made up of fibroelastic tissue, with sparse cellularity and there was no evidence of mitosis. Scattered calcifications corresponding to poorly defined bony trabeculae were observed. The histological features corresponded to a peripheral ossifying fibroma.

The patient evolved satisfactorily and, after a followup of eight months, she was asymptomatic and there were no signs of the lesion recurring.

Discussion

In this case, due to the clinical presentation of a pedicled mass in the gingiva that was asymptomatic and which had been developing for a long time, the diagnostic options pointed basically to benign hyperplasia or neoplasms of the oral mucosa. For this type of lesions, authors such as Regezzi y cols.¹ differentiate two subgroups, epithelial and conjuntival. Hyperplasias include irritation fibroma (repetitive trauma) epulis fisuratum, (badly adjusted prosthesis), pyogenic granuloma in pregnancy, peripheral ossifying fibroma and peripheral granuloma of giant cells. On the other hand the fibroma, lipoma, hemangioma, lymphangioma, schwannoma, neurofibroma, rhabdomyoma and leiomyoma fall into neoplasms.¹

Benign fibro-osseous lesions of the maxilla have presented numerous problems with regard to diagnosis and classification.² In the past, different terms were used for referring to this lesion: osteofibroma, fibro-osteoma, cementifying fibroma, fibroosseous benign lesion of the periodontal ligament... creating great confusion with regard to terminology. This is due firstly to it being impossible to differentiate them clinically and radiologically, and secondly to the difficulty in distinguishing the origin of the calcified material (bone tissue or cement), the latter being a point of great controversy.⁶ Therefore, the ossifying fibroma was considered an osteogenic tumor and it was classified separately from the cementifying fibroma that was thought to be odontogenic.⁴

There is currently general agreement as to their both being just one osteogenic neoplasm, with great expressive variety from the histological point of view: some tumors only contain calcifications with a cement-like appearance, others only bony material, and a third group has a mixture of both calcifications. The separation of these entities is extremely subjective, and this has led to it being referred to as a cemento- ossifying fibroma by the WHO in 1992^2,5,6,8 within nonodontogenic tumors.³ This tumor’s mesodermic origin has been accepted, and it is a fibro-osseous neoplasm originating in multipotential cells of the periodontal ligament.^2,7

The ossifying fibroma is a relatively rare lesion and there is a greater prevalence in women in the third and fourth decades in life. These tumors can be related to a history of trauma and irritation of the gingiva, exodontia and periodontitis, ² as in this case of ours, which could have been induced by constant trauma from a mal-adjusted prosthesis.

It evolves very slowly, a characteristic indicating that the process is benign.³

The ossifying fibroma presents mainly as an intraosseous neoplasm, although cases have been described, such as the one we are dealing with, that affect the gingiva and soft tissues. ⁷ Those that are intraosseous are described as having a predilection for the mandibular molar area, followed by the maxillary area, zygomatic bone, ethmoid, frontal bone and petromastoid region.^2,5 Our case appeared in the upper maxilla, as have other peripheral ossifying fibromas.⁷

With regard to the differential diagnosis, which concerns fundamentally intraosseous lesions, we should include fibrous dysplasia, osteoid osteoma, osteoblastoma, low-grade osteosarcoma, cementoblastoma, periapical and focal cemento- osseous dysplasia, juvenile cemento-ossifying fibroma, chronic osteomyelitis and Garré’s sclerosing osteomyelitis.^3,7

It tends to be asymptomatic in the initial phases, and it is frequently diagnosed in advanced stages, as a localized volume increase that is painless, and which usually leads to considerable aesthetic and functional deformity.^3,4 With regard to our patient, given the large size of the lesion, which was taking up the entire palatal vault, intubation maneuvers were impossible.

As previously mentioned, histologically this lesion is described as a stroma of fibrous tissue with cellular variability and calcifications that consist in rounded (cementum-like) masses, osteoid trabeculae or a combination of both.³ In this case of ours, bone trabeculae predominated.

Radiologically, when the cemento-ossifying fibroma is intraosseous, it tends to be described as a well-defined lesion with areas of calcification and a sclerotic margin. It may appear as radiolucid, radiopaque or mixed, depending on the quantity of mineralized tissue in its interior. It tends to be a unilocular lesion that is well-defined and that can be related to root resorption, or it can cause neighboring roots to shift.¹

Treatment is surgical, and it consists in the enucleation of the tumor and curettage of the bony bed. It tends to be a well-defined lesion, surrounded by a fibrous, relatively hypovascular capsule,³ which facilitates exeresis, as occurred in this case of ours. This permits differentiating between intraosseous lesions and fibrous dysplasia.⁴

Prognosis is good and recurrence is rare when adequate treatment including enucleation and curettage of the bony bed is carried out.^2,4,8 Cases of malignant transformation of this type of tumor have not been reported.^2,4 The clinical follow- up should be over various years due to the slow growth of the tumor and panoramic radiographs should be carried out.⁶

Conclusion

The clinical and histological characteristics of the case described led us to the final diagnosis of peripheral cemento- ossifying fibroma of the upper gingiva. Although the lesion was enucleated completely, the patient should be followed yearly clinically and radiologically.

 Correspondence:
Esther Charro Huerga
Hospital Teresa Herrera
Complexo Hospitalario Juan Canalejo
As Xubias De Arriba 84
15006 A Coruña, España
E-mail: esch26@hotmail.com

References

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2. Martín-Granizo R, de Pedro Marina M. Lesión mixta radiolúcida-radiopaca mandibular en paciente con ameloblastoma previo. Casos clínicos. SECOM on line.        [ Links ]

3. Galdeano M, Crespo Pinilla JI, Álvarez Otero R, Espeso A, Terrier A. Fibroma cemento-osificante gingival mandibular: presentación de un caso. Med Oral Patol Oral Cir Bucal 2004;9:176-9.        [ Links ]

4. De Vicente Rodríguez JC, González Méndez S, Santamaría Zuazua J, Rubiales B. Tumores no odontogénicos de los maxilares: clasificación, clínica y diagnóstico. Med Oral 1997;2:83-93.        [ Links ]

5. Waldrom CA. Fibro-osseus lesions of the jaws. J Oral Maxillofacial Surgery 1993;51:828-35.        [ Links ]

6. Marx RE, Stern D. Benign neoplasm of the bone. En: Oral and Maxillofacial pathology. ed Quintessence 2003;(17):789-96.        [ Links ]

7. Martín Granizo R, Sánchez- Cuellar LA, Falahat F. Cemento-ossifying fibroma of the upper gingivae. Otolaryngol Head Neck Surg 2000; 122:755.        [ Links ]

8. Ertug E, Meral G, Saysel M. Cemento-ossifying fibroma: a case report. Oral Pathol 2004;35:808-10.        [ Links ]