article WS

Case Report

Buccal Bifurcation Cyst in a 7-Year-Old: Surgical Management and 14-Month Follow-Up

S. Thikkurissy,*† Kate M. Glazer,*†‡ Kristin K. McNamara,§ and Dimitris N. Tatakisi

Background: The buccal bifurcation cyst (BBC) is a paradental cyst that typically presents on the buc- cal aspect of mandibular permanent first molars in children 6 to 11 years of age. Histopathologic features are non-specific and closely resemble other inflamma- tory odontogenic cysts. The aim of this article is to re- port a case of a BBC and to review the management of such lesions.

Methods: A 7-year-old white male, complaining of lower right tooth pain and swelling, presented to the Nationwide Children’s Hospital Dental Clinic. A clini- cal examination revealed no caries and swelling lo- calized to the area approximating tooth #30 with a 15-mm probing depth on the buccal aspect. Radio- graphs revealed a radiolucency involving the bifurca- tion and root area of tooth #30, accompanied by the distal displacement of unerupted tooth #31. Therapy consisted of a simple surgical enucleation of the cyst.

Results: A microscopic evaluation revealed a chron- ically inflamed cyst lined by a non-keratinized stratified squamous epithelium. The immediate postoperative course was uneventful. At 14 months postoperatively, the development of teeth #30 and #31 was noted as nor- mal. Probing depths around tooth #30 were £4 mm, tooth #31 was no longer displaced, and no recurrence was detected.

Conclusion: The simple surgical enucleation of the BBC provided good short- and long-term outcomes without compromising the development of associated molars. J Periodontol 2010;81:442-446.

KEY WORDS

Case report; furcation defects; jaw cysts; molar; pathology.

T he buccal bifurcation cyst (BBC), a rare type of paradental cyst that typically occurs in children between the ages of 6 to 11 years,1-3

was first defined by Stoneman and Worth1 in 1983 and is included by the World Health Organization (WHO) in their histologic typing of odontogenic tumors.4 The BBC has been referred to by several different names, including a mandibular BBC,5

circumferential dentigerous cyst,6,7 and inflamma- tory collateral cyst.8 Under the 2003 WHO classifi- cation system, paradental cysts, such as the BBC, are listed under the major heading of ‘‘inflammatory cysts,’’ as are periapical cysts.9

The key clinical features of the BBC, as reviewed by the WHO,4 are: an alteration in the eruption of the af- fected tooth (typically the mandibular first molar), so that the tooth ‘‘leans’’ to the lingual side; an abnormal probing depth on the buccal surface; and the vitality of the associated molar. Other features such as pain, swelling, and the presence of a localized abscess are all variable but may be initial presenting signs and symptoms. Radiographically, the periodontal lig- ament space and lamina dura are usually unaffected. There may be displacement of adjacent teeth depend- ing on the orientation of the lesion. Histologically, the BBC is typically composed of a thin non-keratinized stratified squamous cystic epithelium, exhibiting exo- cytosis of leukocytes and areas of hyperplasia, asso- ciated with a cyst wall containing granulation tissue, which are features consistent with an inflammatory odontogenic cyst.10

Despite the fact that the BBC characteristically af- fects the furcation area of mandibular molars, we found only one report11 of this pathologic condition in the periodontal literature. In addition, the preferred treatment for a BBC has changed in the last 10 years12,13 (see Discussion section in this article for details). The purpose of this report is to present a case of a BBC in the transitional dentition and to re- view the diagnostic, prognostic, and surgical consid- erations for this lesion.

* Division of Pediatric Dentistry, College of Dentistry, The Ohio State University, Columbus, OH.

† Dental Clinic, Nationwide Children’s Hospital, Columbus, OH. ‡ Private practice, Durham, CT. § Division of Oral and Maxillofacial Surgery, Anesthesiology, and Oral and

Maxillofacial Pathology, College of Dentistry, The Ohio State University. i Division of Periodontology, College of Dentistry, The Ohio State

University. doi: 10.1902/jop.2009.090511

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CLINICAL REPORT

A white male (age: 7 years and 2 months) presented to the Nationwide Children’s Hospital Dental Clinic in January 2007 with a chief complaint of ‘‘lower right tooth pain with some swelling for a couple of days.’’ The patient had seen a physician 2 days prior for the same complaint and had been prescribed amoxicillin. The medical history was otherwise unremarkable, and the patient was afebrile and behaved age-appropri- ately. The patient and his parents denied any history of trauma in the area.

The parents provided written informed consent. Upon extraoral examination, a mild asymmetry was noted in the area of the right mandibular angle. An in- traoral examination revealed a hard, bony expansion approximating the partially erupted mandibular right permanent first molar (tooth #30). The expansion ex- tended from the distal side of the primary second mo- lar to the distal side of the erupting tooth #30. The overlying mucosa, including the buccal gingiva of tooth #30, appeared within normal limits, and no sup- puration was noted (Fig. 1A). However, the area was very tender to palpation. Probing depths on the sur- faces of tooth #30 were as follows: mesio-buccal, 7 mm; mid-buccal, 15 mm (Fig. 1B); and disto-buccal, 9 mm, with all other surfaces probing £3 mm. No clinical caries were noted on tooth #30, which also tested vital to thermal testing.

Radiographs of the area (panoramic and occlusal radiographs) revealed a well-defined radiolucent le- sion encompassing the bifurcation and distal root area of tooth #30 with posterior displacement of the devel- oping second molar (tooth #31) (Fig. 2A). Buccal ex- pansion with proliferative periostitis of the cortex and lingual displacement of root apices of tooth #30 was evident (Fig. 2B). No other tooth or jaw pathol- ogy was noted on the radiographs. After consultation with an oral and maxillofacial pathologist (Dr. John Kalmar, Division of Oral and Maxillofacial Surgery, Anesthesiology and Oral and Maxillofacial Pathology,

College of Dentistry, The Ohio State University, Co- lumbus, OH), a differential diagnosis of a BBC and odontogenic keratocyst was established. The parents of the child were informed of the need for a biopsy, and the possible risks to the first molar, which included ex- traction as a potential treatment option. Due to sev- eral considerations (behavioral management, the technique sensitive nature of the biopsy, and limited access), the child was treated under general anesthe- sia at the Nationwide Children’s Hospital Dental Sur- gery Center.

After local anesthesia, a full-thickness flap was el- evated on the buccal aspect of tooth #30. Upon flap elevation, a small, soft-tissue–filled space (;1 mm in bucco-lingual width and 6 mm in mesio-distal length) was evident between the buccal aspect of tooth #30 and the mid-buccal alveolar crest. This space was enlarged toward the buccal side to create an access window of sufficient size to enucleate the lesion. The cystic lesion was attached to the mid- buccal cemento-enamel junction (CEJ) of tooth #30 (Fig. 3) and was not attached to any bony wall or other tooth area (e.g., the buccal surface of roots). A 13 · 9 · 2-mm lesion (Fig. 4A) was enucleated and submitted for histopathologic evaluation. The surgi- cal site was irrigated with sterile saline, and the fur- cation was found to be intact; only 2 to 3 mm of the most coronal mid-buccal aspect of the mesial and distal roots of tooth #30 appeared exposed. A blood clot was allowed to form in the empty bony space, and the flap was repositioned and sutured with interrupted sutures. No dressing was applied. The patient had an uneventful postanesthetic recov- ery and was discharged with standard postoperative instructions.

The surgical specimen was submitted for micro- scopic evaluation. A cyst lined by a non-keratinized stratified squamous epithelium and associated with prominent inflammation of the connective tissue wall was identified (Fig. 4B). The cyst lining was thin and exhibited leukocytic exocytosis with focal areas of hyperplasia (Fig. 4C). A dense chronic inflammatory cell infiltrate, composed of lymphocytes and plasma cells, and extravasated erythrocytes were noted in the cyst wall (Fig. 4D). The histologic diagnosis was reported as chronically inflamed granulation tissue and a non-keratinized stratified squamous epithe- lium, consistent with a BBC.

The patient presented for follow-up 1 month later, reporting an uneventful course of recovery. The pa- tient presented again for follow-up at 2 months post- operatively, and radiographic osseous healing was noted on the distal side of tooth #30 (Fig. 2C). At the 14-month follow-up appointment, the patient was asymptomatic, tooth #30 had erupted to the occlusal plane, all probing depths on tooth #30 were

Figure 1. Clinical presentation of the mandibular BBC. A) Preoperative view of tooth #30. B) Periodontal probe demonstrating a 15-mm mid-buccal probing depth.

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£4 mm, and radiographic assessment indicated that tooth #30 had matured apically with normal root de- velopment, whereas tooth #31 had drifted mesially into an appropriate position (Fig. 2D).

DISCUSSION

The present clinical case concerns the diagnosis and treatment of a BBC on the first permanent mandibular

molar of a 7-year-old system- ically healthy child. The BBC was successfully treated by enucleation alone, leaving the tooth intact. This surgical management approach re- sulted in uncomplicated post- operative recovery, a normal eruption pattern, root devel- opment for the first molar, and correction of the position of the unerupted second man- dibular molar.

This BBC case matched typical clinical features of this lesion as reported in the litera- ture,1-3 namely: a patient aged 6 to 11 years, a lesion asso- ciated with vital mandibular permanent first molar, swell- ing in the affected area, and tilting of the tooth so that it appears to lean lingually. Fi- nally, upon intraoperative ex- posure of the buccal surface of tooth #30, it was noted that the cystic lesion was attached to the mid-facial aspect of the tooth, at the level of the CEJ.

Upon microscopic examination, the histologic find- ings were also consistent with previous reports of the BBC:1,2,11,12 a non-keratinized stratified squamous epithelium associated with chronic inflammation of the surrounding connective tissue wall. In this regard, the histopathology of a BBC is non-specific and is microscopically indistinguishable from other inflam- matory odontogenic cysts.12,14 Information regard- ing the clinical setting and radiographic features are required to avoid a non-specific descriptive micro- scopic analysis (such as a ‘‘chronically inflamed gran- ulation tissue and non-keratinized epithelium’’) and to reach a definitive diagnosis of a BBC.

The pathogenesis of the BBC is only speculative. The typical patient age at presentation and the char- acteristic association with the first molar suggests some relationship to the eruption mechanism.15 An association of paradental cysts with third molars exhibiting buccal enamel projections into the bifurca- tion area, promoting pocket formation and inflamma- tion secondary to pericoronitis, was also reported.16

Nevertheless, the origin of the BBC epithelium is not clear.12 In the present case, there was no enamel projection or other enamel developmental anomaly associated with the lesion.

Traditionally, two methods of treatment for a BBC have been reported in the literature: enucleation of the

Figure 2. Radiographic views. A, C, and D) Partial view of panoramic radiographs. B) Partial view of occlusal radiograph. A) Preoperative: note radiolucent area in the furcation and distal aspect of tooth #30 and the distal displacement of tooth #31. B) Preoperative: note expansion of cortical plate with periosteal reaction and lingual inclination of root apices of tooth #30. C) Two months postoperative: note early radiographic evidence of bone fill in previously radiolucent areas. D) Fourteen months postoperative: note eruption and normal progression of root development of tooth #30 and appropriate position of tooth #31.

Figure 3. Intraoperative clinical view of the BBC attached to mid-buccal surface of tooth #30. Note the smooth surface and lack of attachment to bony walls.

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BBC and (historically) extraction of the associated tooth1-3 or enucleation without extraction of the tooth.12,14,17 Pompura et al.12 described 44 cases of BBCs in children aged 5.5 to 11 years who were treated with enucleation without extraction. These cases were followed up for 2.5 to 3 years with no molars being lost as a result of the pathology. In 1998, David et al.13 suggested a more conservative, non-surgical treatment through a combination of periodontal prob- ing and daily saline lavage of the cyst resulting in micromarsupialization of the lesion. This therapy has not been supported in the literature; the major drawback of such a non-surgical approach is that it precludes definitive diagnosis of the lesion and, thus, could result in improper treatment of a potentially more destructive lesion, such as an odontogenic keratocyst. In the present case, the accepted treat- ment (a conservative cystectomy) was performed.

Although extraction of the associated tooth was ini- tially reported as a BBC treatment option,1-3,18 the ex- traction of a permanent first molar in a growing child can have significant impact on the overall dental growth and development and is a treatment that should be avoided whenever possible. In the present case, the parents and patient were prepared for the potential extraction of tooth #30 due to inadequate

periodontal support or surgi- cal complication(s). In a pa- tient of this chronologic and dental age (7 years), extrac- tion of tooth #30 could have been performed with the in- tention of causing the mesial drift of tooth #31 into its place. However, the surgical periodontal findings in the present case (intact furcation and minimal root exposure) indicate no reason to extract tooth #30. Tooth vitality ren- dered endodontic treatment to be unnecessary.

In the present case, the 14- month follow-up shows that the first mandibular molar continued to develop nor- mally, with correction of the lingual tilt, normalized erup- tion to the occlusal plane, radiographic healing of the associated osseous lesion, lack of periapical pathology, normal probing depths, and no recurrence of the lesion. In addition, the initially dis- tally displaced developing

second mandibular molar moved to the expected po- sition. These follow-up findings are consistent with those of Pompura et al.12 on 44 BBC cases treated with enucleation alone and followed for 2.5 to 3 years. These results suggest that routine extraction of the tooth associated with a typical BBC, a practice still occasionally followed today,18 is not warranted.

CONCLUSIONS

The BBC is a paradental cyst, typically involving the buccal aspect of the mandibular first molar and ap- pearing during the first decade of life. Surgical man- agement by enucleation alone allows for normalized continued development and retention of the perma- nent first mandibular molar.

ACKNOWLEDGMENT

The authors report no conflicts of interest related to this case report.

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buccal cyst–molar area. Dent Radiogr Photogr 1983; 56:1-14.

2. Trask GM, Sheller BL, Morton TH Jr. Mandibular buccal infected cyst in a six-year-old girl: Report of case. ASDC J Dent Child 1985;52:377-379.

Figure 4. A) Gross specimen viewed from the buccal aspect. Note the flattened, coronal aspect (top of the picture) and the rounded, apical aspect of the lesion (bottom of the picture). The bluish coloration reflects extravasated erythrocytes (hemorrhage) into the lesional tissue. B through D) Histopathology of the lesional tissue. The luminal cystic cavity is apparent in B. C and D show the non-keratinized cystic epithelial lining and the extensive inflammatory cell infiltrate (depicted by the heavy influx of small blue cells) and extravasated erythrocytes present in the cyst wall. (Hematoxylin and eosin; original magnification: B, ·20; C, ·100; D, ·200.)

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3. Camarda AJ, Pham J, Forest D. Mandibular infected buccal cyst: Report of two cases. J Oral Maxillofac Surg 1989;47:528-534.

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outcomes in 44 sites. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;83:215-221.

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14. Thompson IO, de Waal J, Nortje CJ. Mandibular infected buccal cyst and paradental cyst: The same or separate entities? J Dent Assoc S Afr 1997;52:503-506.

15. Vedtofte P, Praetorius F. The inflammatory paradental cyst. Oral Surg Oral Med Oral Pathol 1989;68:182- 188.

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17. el-Magboul K, Duggal MS, Pedlar J. Mandibular infected buccal cyst or a paradental cyst? Report of a case. Br Dent J 1993;175:330-332.

18. Iatrou I, Theologie-Lygidakis N, Leventis M. Intraos- seous cystic lesions of the jaws in children: A retro- spective analysis of 47 consecutive cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107: 485-492.

Correspondence: Dr. Sarat Thikkurissy, Division of Pe- diatric Dentistry, College of Dentistry, The Ohio State University, 305 West 12th Avenue, Columbus, OH 43210. Fax: 614/292-1125; e-mail: [email protected].

Submitted September 3, 2009; accepted for publication October 27, 2009.

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