Oral focal mucinosis associated with cervical external root resorption: A case report Eran Gabay, DMD, PhD,a Sharon Akrish, DMD,b and Eli E. Machtei, DMD,a Haifa, Israel RAMBAM HEALTH CARE CAMPUS, AND TECHNION
Background. Oral focal mucinosis (OFM) and cervical external root resorption are both rare lesions with an unknown etiology. In this article we report on a rare occurrence of OFM associated with cervical external root resorption. Methods. A 44-year-old female patient presented with sharp pain while drinking cold beverages and a firm localized gingival overgrowth adjacent to the buccal aspect of tooth #19. An excisional biopsy of the gingival lesion was taken and a deep cavity was found in the buccal aspect of the root. The cavity was restored with an amalgam filling. Results. The histological findings were of a well-circumscribed lesion composed of myxomatous connective tissue, which contained numerous stellate-shaped fibroblasts. Based on the findings, a diagnosis of OFM was made. Conclusion. A rare case of OFM and cervical external root resorption occurring in the same site was described. A possible link between these 2 phenomena was hypothesized. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;110:e75-e78)
Oral focal mucinosis (OFM), a rare lesion with unknown etiology, was first described in 1974 by Tomich.1 Clinically, the lesion presents as an elevated, rounded, asymptomatic mass, and is pink in color, similar to normal mucosa.1 Rarely, the lesion may become secondarily ulcerated.2 The histological presentation is a well-circumscribed yet unencapsulated submucosal mass and is composed of a loose myxomatous or mucinous connective tissue stroma. The fibroblasts located within this loose connective tissue are plump and stellate shaped, often demonstrating delicate fibrillar processes. Few inflammatory cells are noted within the lesion.1,3,4 Most cases of OFM are located on keratinized mucosa such as the gingiva or palate,1,3 although 3 cases of OFM were reported on the tongue as well.5 Cervical external root resorption is a relatively rare lesion with an uncertain etiology, and was first reported in 1930 by Mueller and Rony.6 Cervical external root resorption usually occurs in an inflamed site, which may be attributable to periodontal tissue injury, pressure induced by orthodontic forces, or from a neoplasm.7 Its location is typically located apical to the epithelial attachment on the root, adjacent to the connective tissue attachment.8 Because of its subgingival location, diagnosis by clinical examination is difficult. Radiographically, OFM appears a
Department of Periodontology, School of Graduate Dentistry, Rambam Health Care Campus, and Technion, Faculty of Medicine, Haifa, Israel. b Department of Pathology, Rambam Health Care Campus, Haifa, Israel. Received for publication Jun 14, 2009; returned for revision Mar 28, 2010; accepted for publication Apr 5, 2010. 1079-2104/$ - see front matter © 2010 Mosby, Inc. All rights reserved. doi:10.1016/j.tripleo.2010.04.002
as a well-defined, homogeneous radiolucent lesion. Cervical external root resorption usually progresses slowly, without clinical symptoms. In untreated lesions, significant loss of root structure may occur, usually without pulpal involvement owing to a thin layer of dentin, which serves as a barrier.7,9 In this article, we report the first case of OFM that presented with cervical external root resorption, a rare combination of 2 uncommon lesions. CASE REPORT A 44-year-old female patient presented with a chief complaint of thermal sensitivity and sharp pain from the left mandibular first molar (tooth #19). The patient was otherwise in good general health and without any known systemic diseases. Intraoral examination revealed good oral hygiene with overall good periodontal health. A firm and localized gingival overgrowth was found on the buccal aspect of tooth #19 (Fig. 1). According to the patient, the gingival lesion was first noted 3 years earlier, and since that time, remained static and asymptomatic. On clinical examination, tooth #19 showed marked thermal pain (without overshoot) in response to cold test (ethyl chloride application). The patient was asymptomatic when a percussion test was applied. Radiographic examination revealed a round radiolucent lesion with a clear outline, located on the mesial root of tooth #19, apical to the cementoenamel junction (Fig. 2). A previous periapical radiograph (taken 4 years earlier) already exhibited the radiolucent lesion on the mesial aspect of tooth #19 (Fig. 3). Comparison of the different radiographs revealed a slow progression in the size of the radiolucent cervical lesion. Based on these clinical findings, the following entities were suggested in the differential diagnosis: fibrous hyperplasia, peripheral giant cell granuloma, peripheral odontogenic fibroma and peripheral ossifying fibroma, or initial stage orga-
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Fig. 1. A preoperative photograph, demonstrating a firm localized gingival overgrowth at the buccal aspect of tooth #19.
Fig. 2. Radiograph, taken July 2008, demonstrating a round radiolucent lesion with clear outline located on the mesial root of tooth #19 (white lined circle).
nizing epulis granuloma. Although rare, malignant and metastatic lesions could not be ruled out.10 Under local anesthesia, the patient was treated with a full-thickness mucoperiosteal flap that was raised at the buccal aspect of tooth #19; following debridement, a deep, noncarious cavity on the buccal aspect of the root of tooth #19 was revealed. The cavity was restored with an amalgam filling (Fig. 4). Minimal osteoplasty of the adjacent marginal alveolar bone was needed to avoid insult to the biological width. The buccal flap was then apically positioned and sutured. A biopsy specimen of the gingival lesion was sent for pathological analysis. Postoperative treatment included suture removal 1 week after surgery and the patient was scheduled for bimonthly examination for the first 6 months. Further follow-up examinations were scheduled biannually. Histologically, this was a well-circumscribed yet unencapsulated lesion composed of myxomatous and mucoid connec-
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Fig. 3. Radiograph, taken January 2004, demonstrating the examined radiolucent lesion, located on the mesial root of tooth #19 (white lined circle). No marked changes can be noticed along the 4-year period.
Fig. 4. Photograph taken during surgery, demonstrating the amalgam filling at the cleaned external resorption formed cavity.
tive tissue in the submucosa (Fig. 5, A), which contained numerous stellate-shaped fibroblasts and small blood vessels (Fig. 5, B). The excisional biopsy was incomplete. The histological differential diagnosis included odontogenic myxoma and nerve sheath myxoma. Special stains were applied, which included S-100, reticulin, and alcian blue. S-100 and reticulin showed negative staining. Alcian blue was strongly and diffusely positive, which confirmed the abundant mucin dispersed throughout the connective tissue (Fig. 6). Based on these findings, a pathologic diagnosis of oral focal mucinosis was made. The follow-up for our patient was uneventful. Six months after surgery, the surgical site showed good healing with no recurrence of the lesion (Fig. 7). Tooth #19 remained vital
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Fig. 6. Positive alcian blue staining showing the mucinous material to be hyaluronic acid within the connective tissue (alcian blue, pH 2.5, ⫻40).
Fig. 5. A, Photomicrograph showing a well-demarcated lesion within the connective tissue (hematoxylin and eosin [H&E], ⫻40). B, Photomicrograph showing myxomatous connective tissue containing numerous fibroblasts with finely fibrillar processes (H&E, ⫻100).
without thermal sensitivity. The patient was scheduled for biannual follow-up and periapical radiographs were taken annually.
MATERIALS AND METHODS The tissue specimen was put in formaldehyde 4% and sent for histopathological examination. Four-micrometerthick sections were deparaffinized and rehydrated in a graded series of alcohols. The streptavidin-biotin peroxidase complex (SABC) method was used by means of an automated stainer (NexES and Ventana, Ventana Medical Systems, Inc., Tucson, AZ). Reaction products were visualized by chromagenic reaction with diaminobenzidine tetrachloride (DAB⫹) and hematoxylin staining. Special stains and inmmunohistochemistry included S-100 protein (dilution 1:6000), alcian blue (pH 2.5), and reticulin. DISCUSSION This article presents a rare case of 2 uncommon lesions presenting simulataneously. Both lesions are of
Fig. 7. Photograph, taken 6 months after surgery, demonstrating good healing with no recurrence of the lesion.
uncertain etiology.1,7 This apparently circumstantial finding may not be a random outcome. A possible causative factor for the cervical external root resorption may be mechanical pressure against the root’s external wall that was caused by the OFM tissue mass. The mechanism for root resorption is a sterile inflammatory process, initiated by external force application.11 Constant mechanical pressure may promote external root resorption as occasionally seen in orthodontic therapy,6,11 cysts, and benign tumors.6 In the present case, the resorption rate was slow, as evidenced by the progressive changes noted by comparing the radiographs from 2004 and 2008, and from the clinical pattern of the lesion. These findings are in accordance with previous reports.10 The clinical features of OFM in the present report are similar to those described in the literature: firm to palpation,3 a rounded asymptomatic elevated mass of normal gingiva without ulceration,1,3,4,12 and located in the buccal posterior gingiva in the left mandibular quadrant.3
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The histological findings in the present case demonstrated a well-circumscribed mass that was surrounded by fibrotic tissue. The mass was composed of a myxomatous, loose connective tissue with stellate-shaped fibroblasts, without inflammation. These findings are in correlation with previous reports of OFM.1,3,4,13 The histological differential diagnosis included nerve sheath myxoma and odontogenic myxoma. To differentiate between OFM and odontogenic myxoma we stained for reticulin14 and alcian blue. Alcian blue, which stains the hyaluronic acid, is a predominant feature of OFM, but is not characteristic of odontogenic myxomas.3 Reticulin stain, positive in odontogenic myxoma, is not found in OFM.12 Differentiating between OFM and nerve sheath myxoma is accomplished by staining for S-100 protein, which is positive in nerve sheath myxoma yet absent in OFM.15 Based on the histological presentation and special stains, absence of reticulin fiber and S-100 protein labeling and alcianophilic connective tissue appearance, a final diagnosis of OFM was made. As mentioned before, most of the clinical findings support OFM diagnosis. Yet, cervical external root resorption suggests a locally aggressive behavioral pattern. This feature is more common in odontogenic myxoma,16 rather than OFM. The discrepancy between the histological findings and clinical behavior led us to look for a common pathological pathway for both OFM and external cervical root resorption. The pathogenesis of OFM is thought to be attributable to increased amounts of hyaluronic acid produced by fibroblasts at the expense of connective tissue elements. Aldred et al.3 suggested that myxomatous change may occur in inflammatory lesions such as fibrous hyperplasia and fibrous epulides. Inflammatory lesions may also play a role in cervical external root resorption,6 as seen in gingival overgrowth,17 peripheral giant cell granuloma,18 and pyogenic granuloma.19 We propose that a localized inflammatory environment may be the etiology for both OFM and cervical external root resorption in this case. Although we did not accomplish a complete excision of the OFM, recurrence is not expected and there was no need for supplementary excision.3 According to previous reports on cervical external root resorption, pulpal involvement is not expected because of a protective thin dentin layer.6,9 In the present case, the tooth was vital, without any clinical signs of irreversible pulpitis or perforation into the pulp chamber or the root canal. Hence, conservative treatment such as plastic restoration was the treatment of choice. This conservative approach is advocated in the literature unless there is pulpal involvement.6 In conclusion, a rare case of OFM and cervical external root resorption occurring simultaneously was
described and treated successfully using a conservative approach. REFERENCES 1. Tomich CE. Oral focal mucinosis. A clinicopathologic and histochemical study of eight cases. Oral Surg Oral Med Oral Pathol 1974;38:714-24. 2. Talacko AA, Lacy MF, Besly WJ, Aldred MJ. Oral focal mucinosis: report of two cases with ulceration. Pathology 2004;36:582-3. 3. Aldred MJ, Talacko AA, Ruljancich K, Story RD, Newland S, Chen ST, et al. Oral focal mucinosis: report of 15 cases and review of the literature. Pathology 2003;35:393-6. 4. Buchner A, Merrell PW, Leider AS, Hansen LS. Oral focal mucinosis. Int J Oral Maxillofac Surg 1990;19:337-40. 5. Soda G, Baiocchini A, Bosco D, Nardoni S, Melis M. Oral focal mucinosis of the tongue. Pathol Oncol Res 1998;4:304-7. 6. Mueller E, Rony HR. Laboratory studies of an unusual case of resorption. J Am Dent Assoc 1930;17:326-34. 7. Fuss Z, Tsesis I, Lin S. Root resorption— diagnosis, classification and treatment choices based on stimulation factors. Dent Traumatol 2003;19:175-82. 8. Tronstad L. Root resorption— etiology, terminology and clinical manifestations. Endod Dent Traumatol 1988;4:241-52. 9. Nikolidakis D, Nikou G, Meijer GJ, Jansen JA. Cervical external root resorption: 3-year follow-up of a case. J Oral Sci 2008;50:487-91. 10. 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. 11. Weiland F. External root resorptions and orthodontic forces: correlations and clinical consequences. Prog Orthod 2006;7:156-63. 12. Iezzi G, Rubini C, Fioroni M, Piattelli A. Oral focal mucinosis of the gingiva: case report. J Periodontol 2001;72:1100-2. 13. Saito I, Ide F, Enomoto T, Kudo I. Oral focal mucinosis. J Oral Maxillofac Surg 1985;43:372-4. 14. Perrotti V, Rubini C, Fioroni M, Piattelli A. Soft tissue myxoma: report of an unusual case located on the gingiva. J Clin Periodontol 2006;33:76-8. 15. Nishioka M, Aguirre RL, Ishikawa A, Nagumo K, Wang LH, Okada N. Nerve sheath myxoma (neurothekeoma) arising in the oral cavity: histological and immunohistochemical features of 3 cases.Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107: e28-33. 16. MacDonald-Jankowski DS, Yeung RW, Li T, Lee KM. Computed tomography of odontogenic myxoma. Clin Radiol 2004;59:281-7. 17. Patel K, Darbar UR, Gulabivala K. External cervical resorption associated with localized gingival overgrowth. Int Endod J 2002; 35:395-402. 18. Nedir R, Lombardi T, Samson J. Recurrent peripheral giant cell granuloma associated with cervical resorption. J Periodontol 1997;68:381-4. 19. Vossoughi R, Takei HH. External cervical resorption associated with traumatic occlusion and pyogenic granuloma. J Can Dent Assoc 2007;73:625-8. Reprint requests: Eran Gabay, DMD, PhD Department of Periodontology School of Graduate Dentistry Rambam Health Care Campus P.O. Box 9602 Haifa 31096, Israel
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