Dental Science - Case Report Recurrent peripheral odontogenic fibroma associated with basal cell budding C. Sreeja, N. Vezhavendan1, F. Shabana, D. Vijayalakshmi, M. Devi, N. Arunakiry

Departments of Oral and Maxillofacial Pathology, Adhiparasakthi Dental College, Melmaruvathur, Chennai, Tamil Nadu, 1 Indra Gandhi Dental College, Puducherry, India

ABSTRACT

Address for correspondence: Dr. C. Sreeja, E‑mail: sreeja.bds@gmail. com

Peripheral odontogenic fibroma (POdF) is a rare benign odontogenic neoplasm. It represents the soft tissue counterpart of central odontogenic fibroma. The embryonic source of POdF has been suggested by many as arising from the rest of dental lamina that has persisted in the gingiva following its disintegration. It presents clinically as a firm, slow growing and sessile gingival mass, which is difficult to distinguish with more common inflammatory lesions. Very few cases of recurrence have been documented. It has been stated that histological budding of basal cell layer of the surface squamous epithelium is associated with higher recurrence and the presence of calcification in direct apposition to the epithelial rest is associated with lower recurrence. Hereby, we present a case which histologically exhibited budding of the basal cell layer, which could have been the reason for its recurrence.

Received : 18-04-14 Review completed : 18-04-14 Accepted : 23-04-14

KEY WORDS: Budding, dental lamina, peripheral odontogenic fibroma, recurrence

T

he World Health Organization (WHO) describes odontogenic fibroma as a rare benign odontogenic neoplasm.[1] The peripheral odontogenic fibroma (POdF) is considered to be the mucosal analog to the central odontogenic fibroma.[2] Cases reported in the literature under the terms “odontogenic gingival epithelial harmartoma” “hamartoma of the dental lamina” and “peripheral ameloblastic fibrodentinoma” are actually examples of POdF.[1,3] They are rare and comprise only 0.05% of all biopsy specimens, but they are the most common peripheral odontogenic tumor. The ratio of occurrence of the central to peripheral odonrtogenic fibroma is 1.4:1.4. About 207 cases are reported in the English literature with follow‑up information on 31 cases. A very inadequate data on the biologic behavior and recurrence of this lesion exists. POdF should be considered Access this article online Quick Response Code:

Website: www.jpbsonline.org DOI: 10.4103/0975-7406.137470

a mixed odontogenic tumor because it is composed of active odontogenic epithelial and ectomesenchymal components. It has been stated that budding of the basal cell layer of the surface squamous epithelium was associated with higher recurrence. The presence of calcification in direct apposition to epithelial rests was associated with lower recurrence.[4] The purpose of this article is to report the clinical and histopathological features of a case of POdF in a 53‑year‑old male patient who exhibited recurrence.

Case Report A 53‑year‑old male patient reported with the complaint of a painless swelling on upper back tooth region since 6 months. Patient gave a history of similar swelling 1 year back for which he had undergone excision in a private clinic, no other records was obtained from the patient regarding the same. On intraoral examination, a localized, sessile, erythematous growth was present in the maxillary alveolar region of size 3 cm × 2 cm, extending from buccal gingiva of 15-16 [Figure 1] to the palatal gingiva of 15-16 by traversing the interdental papilla [Figure 2] with mild tooth displacement. The swelling was firm, nontender and exhibited bleeding on palpation. The periodontal examination revealed generalized periodontitis with the recession and Grade II mobility. No other relevant medical and family history was given by the patient.

How to cite this article: Sreeja C, Vezhavendan N, Shabana F, Vijayalakshmi D, Devi M, Arunakiry N. Recurrent peripheral odontogenic fibroma associated with basal cell budding. J Pharm Bioall Sci 2014;6:S204-7.



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Sreeja, et al.: Recurrent peripheral odontogenic fibroma

Investigations An excisional biopsy was performed and the specimen was sent for histopathological investigations. The gross specimen was greyish white, nodular, firm, measuring about 2.5 cm × 2 × 1.7 cm. A gritty texture was felt on sectioning. The hematoxylin and eosin stained section of the specimen under the light microscope exhibited stratified squamous parakeratinized surface epithelium associated with a fibromyxoid connective tissue. The surface epithelium exhibited slender, branching, and deep penetrating rete ridges with budding. The sub‑basilar connective tissue showed abundant capillaries [Figures 3 and 4]. Islands and strands of odontogenic epithelium were found to be scattered throughout the connective tissue [Figure 5], which was more cellular in the fibrous areas [Figure 6] than the myxoid areas [Figure 7]. Few foci showed cementoid and osteoid calcifications [Figure 8]. Considering these features, the case was finally diagnosed as POdF with a comment on its chances to recur.

Differential diagnosis On account of the firm, sessile swelling in the gingiva a provisional diagnosis of pyogenic granuloma (PG), peripheral ossifying fibroma (POF), and peripheral giant cell granuloma (PGG) were given. Commonly given differential diagnosis of peripheral swellings are PG, POF, PGG, peripheral ameloblastoma, parulis, POdF, and fibroma.[1]

Treatment Oral prophylaxis was done and a complete surgical excision of the lesion was performed. The patient was advised for a periodic follow‑up.

Outcome and follow‑up Our patient exhibited recurrence of a similar lesion of the same size and site after a period of 1 month [Figure 9].

Figure 1: Buccal aspect showing localized, sessile, erythematous growth in the maxillary gingival region of size 3 cm × 2 cm, extending buccally from 14 to 16 regions by involving the interdental papilla Journal of Pharmacy and Bioallied Sciences July 2014 Vol 6 Supplement 1

The recurrence rate of POdF has been reported to be from very low to high as 38.9%.[2] Ritwik and Brannon have reported a 50% recurrence rate in their study of 151 cases and also stated that neither the size of the lesion nor the race of the patient was associated with recurrence. They concluded that the recurrence was associated with the histological presence of budding in basal cell layer of the surface epithelium and lower recurrence was associated with the presence of calcification in opposition to the odontogenic epithelial rests.[4] Our patient’s histopathology exhibited budding of the basal cell layer, which could have been the reason for recurrence in our case.

Discussion The World Health Organization defined odontogenic fibroma as “a benign odontogenic neoplasm of fibroblastic origin characterized by relatively mature collagenous fibrous tissue and varying amounts of odontogenic epithelium with potential to occur in either a central or an extraosseous location. The extraosseous counterpart is designated as POdF.”[5] “The recent WHO emphasized more on the central odontogenic fibroma and briefly mentioned about the POdF as being the extraosseous counterpart of the central odontogenic fibroma. Lesions so‑called as odontogenic gingival epithelial hamartoma, odontogenic epithelial hamartoma, hamartoma of dental lamina, and peripheral ameloblastic fibrodentinoma added to the controversy surrounding POdF and are currently included under the histologic spectrum of the POdF.”[4] Several studies from different places around the world show differences in the relative frequency of odontogenic tumors. In a study of 127 cases of odontogenic tumors diagnosed at an oral diagnosis center, only one case of POdF was reported.[6] Peripheral odontogenic tumors are very rare. In a study with 1113 odontogenic tumors, only 4% were composed by PODF. Four out of the 207 cases are reported in the English literature with follow‑up information on 31 cases recurred.[4]

Figure 2: Palatal aspect showing localized, sessile, firm growth extending from 15 to 16 by involving the interdental papilla S205 

Sreeja, et al.: Recurrent peripheral odontogenic fibroma

Peripheral odontogenic fibroma can affect a wide age range, but its peak incidence occurs between the second and fourth decades of life with a slight female predilection. A higher incidence in the mandibular region, particularly on its anterior area has been

reported.[7,4,8] Our case was a male patient of 53 years with the lesion in the maxillary premolar region. POdF appears as a firm, slow growing, and usually sessile gingival mass covered by normal

Figure 3: Surface epithelium exhibiting slender, branching, deep penetrating rete ridges with basal cell budding. The sub-basilar connective tissue showing abundant capillaries

Figure 4: Surface epithelium exhibiting slender, branching, deep penetrating rete ridges with basal cell budding. The sub-basilar connective tissue showing abundant capillaries

Figure 5: Islands and strands of odontogenic cell rests

Figure 6: Highly cellular fibrous connective tissue

Figure 7: Less cellular myxoid areas

Figure 8: Focal areas of calcification



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Histologically, PGG shows more quantity of multinucleate giant cells when compared with POdF. It also presents areas of bone or dystrophic calcifications. However, PGG mass has a characteristic hemorrhage, deposits of hemosiderin, and a high quantity of ovoid spindle cells – commonly on its peripheral region. A study on histological predictors of recurrence concluded that budding of basal cell layer of the surface squamous epithelium was associated with higher recurrence and the presence of calcification in direct apposition to the epithelial rest was associated with lower recurrence.[4] Our case exhibited budding of the basal cell, which might have been the reason for its recurrence.

Learning points/take home messages Figure 9: Recurrent lesion

appearing mucosa. Most lesions range between 0.5 and 1.5 cm in diameter and they rarely cause displacement of the teeth.[9] Our patient also exhibited a painless, firm, sessile swelling, which measured 3 cm × 2 cm with duration of 6 months. The embryonic source of POdF has been suggested by many as arising from the rest of dental lamina that has persisted in the gingiva following its disintegration.[10]

• POdF should be included in the differential diagnosis of swellings involving the gingiva • Presence of basal cell layer budding is associated with increased recurrence • These issues necessitate regular postoperative follow‑up for early detection of recurrence.

 References 1.

The histopathological features of the POdF is similar to that of the WHO type of central odontogenic fibroma.[9] It exhibits interwoven fascicles of cellular fibrous connective tissue, which may be interspersed with areas of less cellular myxoid connective tissue. Islands or strands of odontogenic epithelium are scattered throughout the connective tissue. The epithelial cells may show vacuolization with the presence of cementoid, dentinoid, or osteoid areas.[9,11] Similar features were evident in the histopathology of our case. The clinical differential diagnosis of POdF includes all nodular lesions which occur on the gingiva. The most common being PG, POF, and PGG.[12,13]

2.

Histologically, it is important to differentiate between these lesions. PG shows a vascular proliferation similar to granulation tissue. The mass also presents numerous endothelium‑lined channels filled with red blood cells and a mixed inflammatory infiltrate with lymphocytes, neutrophils, and plasma cells. The surface is usually ulcerated. However, the odontogenic epithelium, a fibromyxomatous connective tissue and occasionally, sites of calcification (dentinoid, cementicles, or osteoid) which are typically presented by POdF is not seen in PG.

8.

In peripheral ossifying fibroma, fibroblastic proliferation admixed with mineralization foci are commonly observed, presenting a cementum‑like, bone or dystrophic calcification. The presence of the odontogenic epithelium seen only in POdF and the highly cellular connective tissue seen in POF are the main differences between POF and POdF.

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3. 4.

5. 6. 7.

9. 10. 11. 12.

Shafer WG, Hine MK, Levy BM. Text Book of Oral Pathology. 5th ed. Reed Elsevier India Private Limited; 2006. p. 441. Michaelides PL. Recurrent peripheral odontogenic fibroma of the attached gingiva: A case report. J Periodontol 1992;63:645‑7. Buchner A. Peripheral odontogenic fibroma. Report of 5 cases. J Craniomaxillofac Surg 1989;17:134‑8. Ritwik P, Brannon RB. Peripheral odontogenic fibroma: A clinicopathologic study of 151 cases and review of the literature with special emphasis on recurrence. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;110:357‑63. Weber A, van Heerden WF, Ligthelm AJ, Raubenheimer EJ. Diffuse peripheral odontogenic fibroma: Report of 3 cases. J Oral Pathol Med 1992;21:82‑4. Santos JN, Pinto LP, de Figueredo CR, de Souza LB. Odontogenic tumors: Analysis of 127 cases. Pesqui Odontol Bras 2001;15:308‑13. Buchner A, Merrell PW, Carpenter WM. Relative frequency of peripheral odontogenic tumors: A study of 45 new cases and comparison with studies from the literature. J Oral Pathol Med 2006;35:385‑91. Garcia BG, Johann AC, da Silveira‑Júnior JB, Aguiar MC, Mesquita RA. Retrospective analysis of peripheral odontogenic fibroma (WHO‑type) in Brazilians. Minerva Stomatol 2007;56:115‑9. Neville BW, Douglas D. Damm, Allen CM, Jerry E. Bouquot. Oral and Maxillofacial Pathology, 3rd ed.(Restricted South Asia Edition) Saunders Publishers; 2009. p. 727. de Villiers Slabbert H, Altini M. Peripheral odontogenic fibroma: A clinicopathologic study. Oral Surg Oral Med Oral Pathol 1991;72:86‑90. Re i c h a r t   A , H a n s   P. O d o n t o g e n i c Tu m o r s a n d A l l i e d Lesion.  Quintessence books, Quintessence Publishing Co. Ltd original from the university of Michigan; 179. Manor Y, Merdinger O, Katz J, Taicher S. Unusual peripheral odontogenic tumors in the differential diagnosis of gingival swellings. J Clin Periodontol 1999;26:806‑9. Silva BS, Yamamoto FP, Da Costa RM, Cruz e Silva BT, De Carvalho WR, Pontes HA. Peripheral odontogenic fibroma: Case report of a rare tumor mimicking a gingival reactive lesion. Rev Odontol UNESP 2012;41:64‑7.

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

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Recurrent peripheral odontogenic fibroma associated with basal cell budding.

Peripheral odontogenic fibroma (POdF) is a rare benign odontogenic neoplasm. It represents the soft tissue counterpart of central odontogenic fibroma...
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