Accepted Manuscript

Surgical Management of the Buccal Bifurcation Cyst: Bone Grafting as a TreatmentAdjunct to Enucleation and Curettage

Rachel Levarek, DMD Mauricio Wiltz, DDS Robert Kelsch, DMD Richard A. Kraut,DDS

PII: S0278-2391(14)00460-1

DOI: 10.1016/j.joms.2014.04.028

Reference: YJOMS 56310

To appear in: Journal of Oral and Maxillofacial Surgery

Received Date: 23 January 2014

Revised Date: 17 April 2014

Accepted Date: 21 April 2014

Please cite this article as: Levarek R, Wiltz M, Kelsch R, Kraut RA, Surgical Management of the BuccalBifurcation Cyst: Bone Grafting as a Treatment Adjunct to Enucleation and Curettage, Journal of Oraland Maxillofacial Surgery (2014), doi: 10.1016/j.joms.2014.04.028.

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Surgical Management of the Buccal Bifurcation Cyst: Bone Grafting as a Treatment Adjunct to Enucleation and Curettage

Rachel Levarek, DMD,* Mauricio Wiltz, DDS,† Robert Kelsch, DMD‡ Richard A. Kraut, DDS§

*Resident, Division of Oral and Maxillofacial Surgery, Montefiore Medical Center, Bronx, NY.

†Attending Surgeon, Division of Oral and Maxillofacial Surgery, Montefiore Medical Center, Bronx, NY.

‡Assistant Professor, Departments of Dental Medicine and Pathology and Laboratory Medicine, NSLIJ Health System, New Hyde Park, NY; Attending, Departments of Dentistry and Pathology, Montefiore Medical Center, Bronx, NY.

§Chairman, Department of Dentistry; Director, Oral and Maxillofacial Surgery Residency Program, Montefiore Medical Center, Bronx, NY.

Address correspondence to Dr. Kraut: Department of Dentistry, Montefiore Medical Center, 111 E 210th Street, Bronx, NY 10467-2490; e-mail: loreilly@montefiore.org, T:718.920.5993, F:718.515.5419

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Introduction

The buccal bifurcation cyst (BBC) is a rare inflammatory odontogenic cyst of unknown

etiology. It typically develops on the buccal aspect of the permanent mandibular first molar, and

occasionally on the permanent mandibular second molar, in children aged four to 14 years.1,2 In

1983, Stoneman and Worth published the largest study on the entity and were the first to

describe the specific clinical and radiographic characteristics of the lesion they named the

“mandibular infected buccal cyst-molar area”.2,3 There have been several reports in the literature

describing similar lesions under a variety of names, such as “circumferential dentigerous cyst”,

“inflammatory collateral dental cyst”, and “inflammatory paradental cyst”.4 The term,

“mandibular infected buccal cyst” was used by Camarda et al5 to identify lesions with

characteristics described by Stoneman and Worth.5 In 1992, a revision to the World Health

Organization classification of odontogenic cysts and tumors included the addition of

“mandibular infected buccal cyst” under the category of “paradental cyst”.1,2,3 Pompura et al6

suggested a more specific and descriptive term that emphasizes the relationship to the buccal

bifurcation and named the lesion, “mandibular buccal bifurcation cyst”.6

The distinct clinical findings of the BBC include involvement of a vital partially- or fully-

erupted mandibular first or second molar in children, swelling in the affected mandibular molar

region, delayed or altered eruption pattern of the involved tooth, and increase in periodontal

pocket depth when the affected tooth is partially erupted.5,6 The specific radiographic features

include a radiolucent lesion on the buccal aspect of the tooth involving the roots to a variable

extent, tilting of the involved molar so that the root apices are toward the lingual cortical plate,

an intact periodontal ligament space and lamina dura, a periosteal reaction on the buccal surface,

and an intact inferior border of the mandible.5,6 The histopathology of the lesion has been

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described as similar to a radicular or inflammatory odontogenic cyst.3 The specific clinical and

radiographic features of the lesion aid in determining a definitive diagnosis.1,6 The treatment of

BBCs has changed significantly over the years and remains controversial; however, the majority

of the current literature supports simple enucleation and curettage of the cyst without extraction

of the involved tooth as the treatment of choice.1,3 This report presents three cases of BBCs that

were treated with enucleation and curettage without extraction of the involved tooth, in addition

to a bone graft placed primarily or secondarily as an adjunctive treatment approach to the current

therapies.

Report of Cases

CASE 1

A healthy 7-year-old male was referred by his pediatric dentist for evaluation of a left

mandibular intraoral swelling that was present for approximately one week. The pediatric dentist

prescribed oral antibiotics one week prior for the swelling. On extraoral examination, there was

minimal left facial swelling and the patient denied any pain or parasthesia. Intraoral examination

revealed a firm swelling, buccal to the left permanent mandibular first molar, with no associated

pain or loss of tooth vitality. The lower left permanent first molar was fully erupted and

displayed a slight Class I mobility. The buccal surface was expanded and the overlying mucosa

was intact with similar color and texture as the surrounding tissue.

A panoramic radiograph did not reveal the lesion in the lower left quadrant (Fig 1). Cone-

beam computed tomography (CBCT) showed a well-circumscribed radiolucency of

approximately 1.0 cm in diameter on the buccal aspect of the left permanent mandibular first

molar extending from the alveolar crest to mid-root with furcation involvement (Fig 2). Bony

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cupping on the buccal surface, slight tilting of the permanent mandibular first molar with the root

apices toward the lingual cortical plate, and an intact inferior border of the mandible were also

seen.

An excisional biopsy was performed under intravenous sedation. A mucoperiosteal flap

was elevated (Fig 3), the lesion was enucleated, and the associated tooth was not extracted. The

defect was grafted with a demineralized bone matrix and a resorbable membrane was placed

(Keystone Dental, Burlington, MA). Histopathologic examination showed a cyst lined by non-

keratinized stratified squamous epithelium with areas of epithelial hyperplasia and an

inflammatory infiltrate in the connective tissue wall (Fig 4). Based on these findings, along with

the clinical and radiographic findings, a diagnosis of BBC was rendered. The patient returned for

a follow-up examination five months later, at which time no evidence of residual or recurrent

pathology was found, there were no signs of tooth mobility, and the periodontium was intact. A

panoramic radiograph showed complete bone regeneration in the area. No evidence of recurrence

was present at the two-year follow-up.

CASE 2

A healthy 6-year-old female was referred for evaluation of a painless intraoral swelling of

the left posterior mandible that was present for a period of one month. On extraoral examination,

there was mild left facial swelling and no parasthesia. Intraoral examination demonstrated a non-

tender, firm, buccal expansion adjacent to a partially erupted lower left permanent first molar.

The mucosa overlying the expanded buccal surface was normal and intact.

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A panoramic radiograph revealed a well-demarcated, unilocular radiolucency of

approximately 1.0 cm in diameter about the roots, and furcation of the partially erupted left

permanent mandibular first molar. CBCT showed a lytic lesion on the buccal aspect of the lower

left permanent molar with lingual displacement of the roots, buccal cortical expansion and an

onion-skin appearance of the periosteum on the buccal surface (Fig 5). Slight tilting of the

permanent mandibular first molar with the root apices toward the lingual cortical plate and an

intact inferior border of the mandible were also seen.

Under intravenous sedation, a mucoperiosteal flap was elevated, the lesion was enucleated,

and the involved tooth was not extracted. Histopathologic examination showed a cyst lined by

non-keratinized stratified squamous epithelium with an inflammatory infiltrate in the connective

tissue wall compatible with BBC.

The patient reported mild left mandibular swelling and pain at a one-month postoperative

follow-up visit. On extraoral examination, there was mild left mandibular swelling, tenderness to

palpation, and no parasthesia. Intraorally, a firm, tender, vestibular swelling was noted at the site

of prior treatment. Under intravenous sedation, a full thickness flap was elevated and the surgical

site was debrided of the granulation-like tissue. Cancellous bone (Stryker Corporation,

Kalamazoo, MI) was placed in the defect and a resorbable membrane was placed over the graft

site. The patient returned for a follow-up examination nine months later, with no further evidence

of pain or swelling, there were no signs of tooth mobility, and the overlying mucosa was intact

and similar in appearance to the surrounding tissue. A panoramic radiograph showed complete

bone regeneration in the area.

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CASE 3

A 7-year-old female was referred for evaluation of a painful intraoral swelling of the left

posterior mandible that was noticed approximately two weeks prior by the patient’s mother. On

extraoral examination, there was mild left facial swelling that was tender to palpation with no

parasthesia. Intraoral examination revealed a moderately large vestibular expansion, measuring

approximately 2.0 cm in diameter, buccal to the unerupted left permanent mandibular first molar.

The swelling was collapsible with sufficient pressure and the mucosa overlying the expansion

was intact with similar color and texture as the surrounding mucosa.

A panoramic radiograph revealed a well-defined, unilocular radiolucency of approximately

2.5 cm in diameter associated with the unerupted lower left permanent first molar (Fig 6). CBCT

displayed a large lytic lesion on the buccal aspect of the lower left permanent first molar

extending to the adjacent primary teeth. Buccal expansion with an eggshell thin buccal cortex

and slight tilting of the permanent mandibular first molar with the root apices displaced toward

the lingual cortical plate were also depicted (Fig 7).

An incisional biopsy was performed under intravenous sedation. Multiple pieces of the

lesion were collected and sent for histopathologic examination. Microscopic examination

showed a cyst lined by non-keratinized stratified squamous epithelium with areas of epithelial

hyperplasia and an inflammatory infiltrate in the connective tissue wall. Based on these findings,

along with the clinical and radiographic findings, a diagnosis of BBC was rendered.

Complete excision of the lesion under general anesthesia was performed. The area was

thoroughly curetted and the involved tooth was saved. The defect was grafted with cancellous

bone (Stryker Corporation, Kalamazoo, MI) and a resorbable membrane was placed. The patient

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tolerated the procedure well and returned for a follow-up examination three weeks later, at which

time no evidence of residual lesion was found and a panoramic radiograph showed the lower left

permanent first molar starting to erupt.

The patient reported mild left mandibular pain at the site of prior treatment at a five-month

postoperative follow-up visit. On extraoral examination, there was no mandibular swelling.

Intraorally, a vestibular swelling was noted and purulent drainage was expressed with

manipulation at the site of prior excision. There were no necrotic bone pieces noted. A

panoramic radiograph displayed adequate consolidation around the periphery of the grafted site

and there was no gross bone loss (Fig 8). The patient was prescribed one week of oral antibiotics

and returned for a follow-up examination two weeks later, with no further evidence of drainage,

no signs of tooth mobility and adequate soft tissue attachment.

Discussion

The buccal bifurcation cyst (BBC) is an uncommon lesion that presents in the first or

second decade and is often associated with a partially-erupted or fully-erupted permanent

mandibular first or second molar.1,2,7 It most often presents unilaterally, however, bilateral

occurrence has been reported.2,6 The lesion exhibits distinct clinical and radiographic features

described by Stoneman and Worth, which differentiates it from other inflammatory odontogenic

cysts.6 The clinical and radiographic findings in our three cases were consistent with those

reported by Stoneman and Worth. Histologic characteristics of the BBC are similar to those seen

in a radicular/inflammatory odontogenic cyst, demonstrating a non-keratinized proliferative

stratified squamous epithelium with areas of epithelial hyperplasia and an inflammatory infiltrate

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in the connective tissue wall peripheral to the cyst lining.4,5,6 Microscopic examination of the

lesions in our cases uniformly exhibited those findings.

Management of the BBC has changed significantly over the years.1 Initial and prior studies

have reported successful surgical treatment through enucleation of the cyst and extraction of the

involved tooth, endodontic treatment of the tooth and curettage of the cyst, and enucleation of

the cyst without tooth extraction.1,2,7 A non-surgical treatment approach was described by David

et al4, which presented three cases with a total of five cysts, and suggested that periodontal

probing of the buccal pocket or daily irrigation of the buccal pocket with saline may result in

regression of the lesion.4 The authors proposed that manipulation induced a small opening in the

cyst lining, inducing a “micro-marsupialization” which allowed the cyst to depressurize and heal

without surgical intervention.1,3,4,7 Recently, Corona-Rodriguez et al1 and Zadick et al7, each

reported a case of BBCs that self-resolved.

The majority of the current literature supports simple enucleation and curettage of the cyst

without extraction of the involved tooth as the treatment of choice.3 All three of our cases were

enucleated and curetted without extraction of the affected tooth. Due to the size of the bony

defects, ranging from approximately 1.0-2.5 cm in diameter, all three of the subjects received a

bone graft either immediately after enucleation of the cyst or secondarily. The bone grafts were

placed to enhance bone regeneration, provide stability and adequate root and furcation coverage

of the involved tooth and to re-establish the alveolar crest to the level of the cemento-enamel

junction. Panoramic radiographic follow-up revealed good bone fill as well as sufficient alveolar

crest levels in all three cases. Clinical follow-up examinations demonstrated an intact overlying

mucosa and sufficient soft tissue attachment.

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The decision to place a bone graft as an adjunctive therapy differs from the non-surgical

and observational treatment approaches for the BBC. Factors that predict which lesions may be

more likely to need a bone graft are unknown. There are no guidelines on which defects need to

be managed surgically with or without a bone graft; however, our rationale is that at least some,

especially large, expanding lesions, may benefit from this approach either primarily or

secondarily. To the best of our knowledge, this is the first report of using a bone graft as a

treatment adjunct to enucleation and curettage of the BBC and appears to be successful.

Conclusion

The current recommended therapy for the buccal bifurcation cyst (BBC) is enucleation

and curettage with maintenance of the involved tooth. However, there is no consensus on the

treatment of the BBC. We believe that small lesions measuring approximately 1.0 cm or less,

without or with little cortical bone disruption, can be successfully treated by enucleation and

curettage if adequate bone is present. The question in these cases is the treatment of large,

expansile lesions greater than 1.0 cm in diameter with considerable alveolar crest and buccal

cortical bone loss. These cysts were treated successfully with enucleation and curettage in

conjunction with a bone graft placed either primarily or secondarily as a means of providing

interim stability to the involved tooth within the large bony defect, enhancing bone regeneration,

re-establishing alveolar bone height and enabling a means for proper soft tissue re-attachment to

a normal level. Panoramic radiographic assessment of all three cases after bone graft

reconstruction demonstrated adequate bone fill and re-establishment of the alveolar bone height.

The clinical findings during follow-up examination in the three cases correlated with the post-

operative radiographic features and showed an intact, normal overlying mucosa with adequate

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soft tissue re-attachment and non-mobile teeth. A future study determining factors that predict

which lesions may be more likely to need a bone graft as an adjunct to enucleation and curettage

may be beneficial.

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References

1. Corona-Rodriguez J, Torres-Labardini R, Tizcareno-Velasco M, et al: Bilateral buccal

bifurcation cyst: Case report and literature review. J Oral Maxillofac Surg 69:1694-1696, 2011

2. Ramos LMA, Vargas PA, Coletta RD, et al: Bilateral buccal bifurcation cyst: Case report and

literature review. Head and Neck Pathol 6:455-459, 2012

3. Shohat I, Buchner A, Taicher S: Mandibular buccal bifurcation cyst: enucleation without

extraction. Int J Oral Maxillofac Surg 32:610-613, 2003

4. David LA, Sandor GK, Stoneman DW: The buccal bifurcation cyst: is non-surgical treatment

an option? J Can Dent Assoc 64:712, 1998

5. Camarda AJ, Pham J, Forest D: Mandibular infected buccal cyst: report of two cases. J Oral

Maxillofac Surg 47:531, 1989

6. Pompura JR, Sandor George KB, Stoneman DW: The buccal bifurcation cyst: a prospective

study of treatment outcomes in 44 sites. Oral Surg Oral Med Oral Pathol 83:215-221, 1997

7. Zadik Y, Yitschaky O, Neuman T, et al: On the self-resolution nature of the buccal bifurcation

cyst. J Oral Maxillofac Surg 69:e282-e284, 2011

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Legends

FIGURE 1. Case 1. Panoramic radiograph showing no radiolucent lesion in the lower left

quadrant.

FIGURE 2. Case 1. Axial CBCT showing a well-defined radiolucency of approximately 1.0 cm

in diameter about the roots and furcation of the lower left permanent first molar with slight

buccal expansion.

FIGURE 3. Case 1. Intraoral buccal view of lesion.

FIGURE 4. Case 1. A photomicrograph showing a cystic process lined by thin proliferative

squamous odontogenic epithelium with an associated chronic inflammatory infiltrate in the

fibrous connective tissue wall. (Hematoxylin and eosin; magnification, x10)

FIGURE 5. Case 2. Axial CBCT displaying lingual displacement of the roots of the first molar.

Arrow reveals an onion-skin appearance of the periosteum on the buccal surface.

FIGURE 6. Case 3. A panoramic radiograph showing a well-demarcated radiolucency of

approximately 2.5 cm in diameter about the roots and involving the furcation of an unerupted left

permanent mandibular first molar.

FIGURE 7. Case 3. Coronal CBCT showing buccal expansion with an eggshell thin buccal

cortex and slight tilting of the permanent mandibular first molar with the root apices toward the

lingual cortical plate.

FIGURE 8. Case 3. A panoramic radiograph demonstrating sufficient bone consolidation around

the periphery of the permanent mandibular first molar and no gross bone loss at a five-month

post-operative follow-up visit.

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