unique pericoronal orocutaneous fistula: sequelae of chronic subclinical inflammation

meningitis can occur in lesions that rupture, likelybecause of the irritating effects of cholesterol in thecellular debris.7 Enophthalmos and diplopia mayoccur post-operatively in cases where the lesionhas invaded and destroyed part of the orbital archi-tecture.

Intradiploic epidermoid tumors are rare entitiesthat require aggressive surgical treatment. This le-sion should be included in all differential diagnosesof osteolytic lesions of the orbit. The case describedin this report is an example of how locally destruc-tive these lesions can be if left untreated. Thepossibility of malignant transformation, althoughuncommon, also mandates total excision.

References1. Blanco G, Esteban R, Galarreta D, et al: Orbital intradiploic

giant epidermoid cyst. Arch Ophthalmol 119:771, 20012. Baxter JW, Netsky MG, Osborne DR, et al: Epidermoid and

dermoid tumors, in Wilkins RH, Rengachary SS: Neurosurgery.New York, NY, McGraw-Hill, 1985, pp 655-673

3. Niederhagen B, Reich RH, Zentner J: Temporal dermoid withintracranial extension: Report of a case. J Oral Maxillofac Surg56:1352, 1998

4. Arana E, Latorre FF, Revert A, et al: Intradiploic epidermoidcysts. Neuroradiology 38:306, 1996

5. Eijpe AA, Koorneef L, Verbeeten B, et al: Intradiploic epider-moid cysts of the bony orbit. Ophthalmology 98:1737, 1991

6. Rumelt S, Harsh GR, Rubin PA: Giant epidermoid involvingthree cranial bones. Arch Ophthalmol 115:922, 1997

7. Fukuta F, Jackson IT: Epidermoid cyst and cholesterol granu-loma of the orbit. Br J Plast Surg 43:521, 1990

8. Dutt SN: Radiologic differentiation of intracranial epidermoidsfrom arachnoid cysts. Otol Neurol 23:84, 2002

9. De Vries J, Freihofer H, et al: Successful surgical repair ofprogressive exophthalmos caused by a meningocele in apatient with neurofibromatosis type 1. J Neurosurg 89:1032,1998

10. Demirci H, Shields CL, Shields JA, et al: Bilateral sequentialorbital involvement by eosinophilic granuloma. Arch Ophthal-mol 120:978, 2002

11. Meyer DR, Lessner AM, Yeatts RP, et al: Primary temporal fossadermoid cysts: Characterization and management. Ophthalmol-ogy 106:342, 1999

12. Holds JB, Anderson RL, Mamalis N, et al: Invasive squamous cellcarcinoma rising from asymptomatic choristomatic cysts of theorbit. Ophthalmology 100:1244, 1993

13. Bretschneider T: Squamous cell carcinoma arising in an intra-diploic epidermoid cyst. Neuroradiology 41:570, 1999

J Oral Maxillofac Surg63:1676-1679, 2005

Unique Pericoronal OrocutaneousFistula: Sequelae of Chronic

Subclinical InflammationAndrew Cheung, DDS,* John-Wallace Hudson, DDS,† and

Tate Viehweg, DMD‡

It is more desirable to remove third molars at ayounger age, if indicated, for prevention of futurecomplications and associated pathology. Specifi-cally, mandibular third molar teeth show the high-est incidence of impaction and have been held

responsible for several pathologic entities such aspericoronitis, periodontal defects posterior to thesecond molars, caries in the second and third mo-lars, neurogenic and myofascial pain, odontogeniccysts and tumors, and primary and secondarycrowding of dentition.1 Studies indicate that thereis a higher incidence of infection of retained thirdmolars in patients over the age of 30, with agebeing a major causative factor for complication.2

Other studies suggest that age alone increases sur-gical morbidity and has a direct correlation withpain affecting the patient’s quality of life.3-5 It isrecommended that early extraction of fully im-pacted third molars, if indicated for removal, is thebest approach. The following is a presentation of aunique case of third molar pathosis involving fistu-lization through the parotid gland with potentialatypical postoperative morbidity.

Received from the Department of Oral and Maxillofacial Surgery,

University of Tennessee Medical Center, Knoxville, TN.

*Chief Resident.

†Professor.

‡Former Chief Resident.

Address correspondence and reprint requests to Dr Hudson:

University of Tennessee Medical Center, 1930 Alcoa Highway,

Building A, Suite 335, Knoxville, TN 37920; e-mail: jwhudson@

mc.utmck.edu

© 2005 American Association of Oral and Maxillofacial Surgeons

0278-2391/05/6311-0017$30.00/0

doi:10.1016/j.joms.2005.07.014

1676 UNIQUE PERICORONAL OROCUTANEOUS FISTULA

Report of a Case

A 75-year-old man was referred because of a drainingorocutaneous fistula extending through a parotid glandfrom infected impacted mandibular right third molar asevidenced by the clinical photograph (Fig 1). The patienthad been examined and treated initially by a general dentist.After review of the situation, the patient was referred to anoral and maxillofacial surgeon, who had recommended ex-traction approximately 2 years earlier. The tooth was symp-tomatic for approximately 3 to 4 months, including painand purulent exudate. Per patient history, longstandingantibiotic therapy was initiated by the general dentist andcontinued for several months. The panoramic radiographicevaluation showed a mesioangular impacted mandibularright third molar, with a radiolucent region surrounding theroots (Fig 2). Extraoral examination disclosed a right man-dibular angle orocutaneous fistula. The oral mucosa overthe region in question appeared grossly intact but withevidence of inflammation. No paraesthesia was evident inthe distribution of the inferior alveolar nerve on the affectedside. No cranial nerve VII paralysis was apparent on theaffected side. The panoramic radiograph showed abscessedmandibular right third molar, full bony impacted mandibu-lar left third molar, and generalized periodontal disease of

the surrounding dentition. The differential diagnosis in-cluded an orocutaneous fistula associated with an impactedabscessed tooth, folliculitis, osteomyelitis associated withan abscessed tooth, and possible inflammatory induced neo-plasia, based on the chronicity and aggressive nature of thelesion.

The patient was subsequently taken to the operatingroom, and the fistulous tract and impacted third molar wereremoved using both an extraoral and an intraoral approach(Figs 3-5). The procedure involved dissection through thesuperficial portion of the parotid gland. The associatedgranulation tissue was extremely adherent to the inferioralveolar nerve and was carefully dissected to avoid injury.

Sectioning revealed the opening to extend 1.3 cm intothe subcutaneous tissue. A gelatinous cystic structure wasidentified adjacent to the opening and measured 0.4 cm inthe greatest dimension. At the time of the initial evaluation,because of the aggressive nature and tract of the fistula,adherence to the nerve, and size, a neoplastic lesion washigh on the differential diagnosis. The pathology report was

FIGURE 1. Cutaneous portion of fistula as seen preoperatively.

Cheung, Hudson, and Viehweg. Unique Pericoronal Orocutane-ous Fistula. J Oral Maxillofac Surg 2005.

FIGURE 2. Impacted mesioangular third molar with surrounding largeradiolucency.


FIGURE 3. Excision with margin of cutaneous fistula.


FIGURE 4. Orocutaneous fistulous tract specimen in its entirety.


CHEUNG, HUDSON, AND VIEHWEG 1677

acute and chronic inflammation negative for dysplasia orneoplasia.

Cultures of the specimen yielded no growth of bacteria.This can be attributed to the history of antibiotic therapy.

Postoperatively, the patient was followed weekly overthe course of 2 months (Fig 6). One concern was that thepatient might have sustained V3 paresthesia or facial nerveinjury due to the proximity and management of the lesion.However, postoperative visits revealed no evidence of thiswith the respective involved nerves.

Discussion

The types of bacteria typically implicated in peri-coronal abscess are Streptococcus viridans, mixedoral flora, spirochetes, and fusobacteria.6 Other bac-teria associated with pericoronitis include Prevotellaintermedia, Peptostreptococcus micros, F. nucleam,A. actinomycetemcomitans, Veillonella, and Capno-cytophaga spp.6 A recent study by White et al7

showed that clinical and microbial changes associatedwith the initiation of periodontitis may present first inthe third molar region, even in young adults. Thebacteria involved most often exist in complexes, par-ticularly Bacteroides forsythus, Porphyromonas gin-givalis, and Treponema denticola. Despite the youngage and healthy periodontal status of the participantsin the study, the presence of these bacteria in asymp-tomatic individuals with third molars places them atrisk for future complications both locally and system-ically. Recent clinical studies on some of these patho-gens, such as P. gingivalis and B. forsythus, havefound these organisms present in atheromas post–myocardial infarction via polymerase chain reactionand account for sustained elevation of inflammatoryindices such as C-reactive protein. Pathogen-inducedinflammatory mediators have been associated withplaque rupture as related to myocardial infarction and

cerebrovascular accident.8 Some of the pathogenshave also been linked to premature birth and low-birth-weight infants.7 The local environment with in-creased serum transudate seen as increased gingivalcrevicular fluid flow, bringing protein and heme, cou-pled with low oxygen tension facilitates the contin-ued multiplication and resulting destructive activity ofthe pathogenic bacteria. Furthermore, if one region ofthe mouth could be singled out as a prime candidatefor such a scenario, it is the mandibular third molarregion. In addition to being difficult for patients toreach to keep clean, the horizontal shelf of thickcortical bone of the mandibular external obliqueridge and the easily traumatized covering mucosalends itself to the accumulation of plaque and result-ing inflammation.7

The majority of these bacteria are anaerobic. Anaer-obic bacteria produce a variety of proteolytic en-zymes, endotoxins, and exotoxins, which results in asignificant amount of tissue destruction and lysis.9

Over time, the chronicity of infection and inflamma-tion has the potential to develop from metaplasia to aneoplasm.

For many neoplasms, the initial histologic manifes-tation may be in the form of a hyperplasia. In contrast

FIGURE 5. Closure of orocutaneous fistula.


FIGURE 6. Two month follow-up clinical photograph.


1678 UNIQUE PERICORONAL OROCUTANEOUS FISTULA

to hyperplasia, metaplasia involves the abnormaltransformation of one fully differentiated, adult tissueinto another kind of differentiated tissue.10

In this case, the aggressive nature of this lesion hadcharacteristics mimicking metaplasia leading to neo-plastic disease. The chronic inflammation caused bythe anaerobic pathogens led to tissue destruction andtransformation as well as potential systemic decom-pensation (ie, cardiac, vascular, and respiratory dis-ease). At some point in the future it may be possibleto vaccinate people against the organisms that causethese problems in a similar manner to elective sple-nectomy, with regard to encapsulated organisms.Once teeth are exposed in a region difficult to keepclean and with qualitatively poorer soft tissue, peri-odontal pathogens can proliferate. The end result ofthis process was evident by the formation of an ab-scess cavity with suppuration forming a fistulous tractthrough alveolar bone that exited into the surround-ing integumental soft tissue envelope.

The initial treatment of this lesion was chosen be-cause of the potential presence of neoplasia and in-vasive properties. Many patients procrastinate intreatment, subjecting themselves to complicationssuch as delayed healing, increased risk for furthersurgery, acute and chronic infection, postoperativebleeding, nerve injury, and pathologic fracture as theycontinue to delay.

An alternative treatment option for this patient couldhave been removal of the infected and abscessed tooth,allowing for the infection to resolve, and then a second-ary closure of the fistulous tract, if the infection resolvedand the drainage ceased. The shortcomings of this pro-cedure would include the possibility of missing a dys-

plastic or neoplastic lesion developing along the clinicalaspect of the fistulous tract that was very chronic andmature in nature or creating an inclusion cyst within thebody of the parotid gland with incomplete removal ofthe mature epithelial lining.

References1. Chiapasco M, Crescentini M, Romanoni G: Germectomy or

delayed removal of mandibular impacted third molars: Therelationship between age and incidence of complications.J Oral Maxillofac Surg 53:418, 1995

2. Yoshii T, Hamamoto Y, Muraoka S, et al: Incidence of deepfascial space infection after surgical removal of the mandibularthird molars. J Infect Chemother 7:55, 2001

3. Bruce RA, Frederickson GC, Small GS: Age of patients andmorbidity associated with third molar surgery. J Am Dent Assoc101:240, 1980

4. de Boer MP, Raghoebar GM, Stegenga B, et al: Complicationsafter mandibular third molar extraction. Quintessence Int 26:779, 1995

5. Capuzzi P, Montebugnoli L, Vaccaro MA: Extraction of im-pacted third molars. A longitudinal prospective study on fac-tors that affect postoperative recovery. Oral Surg Oral Med OralPathol 77:341, 1994

6. Trummel C, Behnia A: Periodontal and pulpal infections, inTopazian RG, Goldberg MH, Hupp JR (eds), Oral and Maxillo-facial Infections (ed 4). Philadelphia, PA, WB Saunders, 2002,pp 142–146

7. White, Madianos, Offenbacher, et al: Microbial complexes de-tected in the second/third molar region in patients with asymp-tomatic third molars. J Oral Maxillofac Surg 60:1234, 2002

8. Beck JD, Offenbacher S: The association between periodontaldiseases and cardiovascular diseases: A state-of-the-science re-view. Ann Periodontol 6:9, 2001

9. Peterson L: Principles of surgical and antimicrobial infectionmanagement, in Topazian RG, Goldberg MH, Hupp JR (eds),Oral and Maxillofacial Infections (ed 4). Philadelphia, PA,WB Saunders, 2002, p 103

10. Leach S, Pearson A, Beauchamp R: Tumor biology, in Green-field LJ, Mulholland MW, Oldham KT (eds), Surgery, ScientificPrinciples and Practice (ed 3). Philadelphia, PA, LippincottWilliams and Wilkins, 2001, pp 468–469

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unique pericoronal orocutaneous fistula: sequelae of chronic subclinical inflammation

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