CASE REPORT

Primary Gingival Squamous Cell Carcinoma: A Case Reportof the Clinical Presentation and Management

David J. Meister,* Gregory R. Caldwell,* Lisa M. Masters,* Thomas W. Sterio, and Michael P. Mills*

Introduction:Oral squamous cell carcinoma is the eighthmost common cancer in the world. In the United States, it isestimated that 41,380 people will be diagnosed with oral or pharyngeal cancer in 2013, with 7,890 deaths expected. Poten-tially malignant (premalignant/precancerous) oral lesions commonly present as areas of leukoplakia (white patch), erythro-plakia (red patch), or a mixture of both. Early diagnosis is critical to the successful management of the lesion.

Case Presentation: This case report documents the detection and management of a gingival squamous cell carci-noma in a 69-year-old white male who recently completed comprehensive periodontal and restorative therapy. Early detec-tion and diagnosis were paramount in limiting the potential invasiveness and extent of the surgical resection for this patient.

Conclusion: This case report illustrates the importance of maintaining vigilance and thoroughly evaluating the patientthroughout all phases of therapy. Clin Adv Periodontics 2014;4:1-7.

Key Words: Carcinoma, squamous cell; erythroplasia; gingiva; leukoplakia; periodontitis.

BackgroundOral squamous cell carcinoma is the eighth most commoncancer in the world.1 In North America, the highestincidence of oral cancer is found in middle-aged African-American males and in white males older than 65 years.2

The incidence in males is reported to be three times higherthan in females.2 In the United States, it is estimated that41,380 people will be diagnosed with oral or pharyngealcancer in 2013, and, of these, 7,890 deaths are expected.3

The estimated 5- and 10-year relative survival rates for oraland pharyngeal cancers are 62% and 51%, respectively.3

In descending order of appearance, themost common lo-cations for oral squamous cell carcinoma are: 1) the lowerlip, 2) tongue, 3) floor of the mouth, 4) soft palate, 5) alve-olar ridge and gingiva, and 6) buccalmucosa. Symptomatic

oral squamous cell carcinomas are often not difficult to di-agnose because patients may complain of pain, an ulcer,a swelling or growth, or difficulty in chewing or swallow-ing. However, early presentations of these lesions are oftenasymptomatic and can easily be overlooked.Oral potentially malignant lesions commonly present as

areas of leukoplakia (white patch), erythroplakia (redpatch), or a mixture of both.2 Other potentially malignantlesions include: 1) submucous fibrosis, 2) lichen planus, 3)actinic keratosis, 4) discoid lupus erythematosus, 5) kera-tosis congenita, and 6) epidermolysis bullosa.2 They mayalso present clinically as ulcers or nodular growths. Theestimated global prevalence of oral potentially malignantlesions ranges from 1% to 5%.2 The subtle changes ofgingival carcinomamaymimic candidiasis, denture stoma-titis, reaction to an allergen, vesiculobullous disorders,periodontal disease (i.e., mobility of affected teeth), or areactive epulis.2,4,5

This case report describes a patient who had receivedcomprehensive periodontal and restorative therapy beforethe initial appearance of a leukoplakic lesion on the gin-giva, detected during a periodontal maintenance visit.The lesion was subsequently biopsied and diagnosed asa gingival squamous cell carcinoma.

* Department of Periodontology, University of Texas Health Science Centerat San Antonio Dental School, San Antonio, TX.

Private practice, San Antonio, TX.

Private practice, Swampscott, MA.

Submitted March 1, 2013; accepted for publication May 28, 2013

doi: 10.1902/cap.2013.130021

Clinical Advances in Periodontics, Vol. 4, No. 1, February 2014 1

Clinical Presentation, Management,and OutcomesIn September 2008, a 69-year-old white male was referredto the Graduate Periodontics Clinic at the University ofTexas at San Antonio Health Science Center (UTHSCSA),San Antonio, Texas, for comprehensive periodontal anddental implant treatment. The patient reported a medicalhistory of hypertension, hypercholesterolemia, low tes-tosterone, and erectile dysfunction. Medications includedvalsartan, atorvastatin, testosterone injections, sildenafil,and 81 mg aspirin daily. He reported consuming two tothree alcoholic beverages per week but denied use of anytobacco product or recreational drugs. Previous dentalhistory included routine restorative treatment and non-surgical periodontal therapy. Family medical history wasnegative for systemic illnesses or conditions.At his initial evaluation visit, extraoral findings were

found to be unremarkable, and an intraoral screening forsigns consistent with malignancy was negative. The com-prehensive periodontal examination revealed poor oralhygiene with an OLeary plaque index6 (PI) of 60%(Fig. 1). Clinical parameters included: 1) probing depthsranging from 1 to 7 mm and clinical attachment loss from2 to 7 mm; 2) gingival recession of 1 to 3 mm; 3) bleedingon probing at 22%of sites; 4) Glickman7 Class 1 and 2 fur-cation invasions on all remaining molars; and 5) a Millermobility index8 of Class I for teeth #7 through #10 and#25. There were multiple failing restorations with cari-ous lesions and a self-reported history of tooth grinding.

Occlusal analysis revealed: 1) bilateral Angle Class I canineand molar relationships; 2) bilateral canine disocclusion inworking excursions; 3) a non-working contact betweenteeth #5 and #28; and 4) crossbite of teeth #12 and #21.The intraoral radiographic survey revealed generalizedmoderate to localized severe horizontal bone loss (Fig. 2).The initial diagnoses included: 1) generalized moderateand localized severe chronic periodontitis; 2) gingival reces-sion; 3) caries; 4) bruxism; 5) secondaryocclusal trauma; and6) partial edentulism.Over a 28-month period, the patient received both com-

prehensive periodontal and restorative therapy for whichappropriate verbal andwritten informed consentwas givenand obtained. After the non-surgical phase of treatment,oral hygiene had improved to an OLeary PI of 20%.The periodontal surgical phase included three posteriorsextants of osseous surgery, functional crown lengtheningof teeth #6 through #11, and extractions of teeth #10 and#28 with subsequent dental implant placement at bothsites. All surgical sites healed without sequellae. At 6.5months after crown-lengthening surgery, the gingiva atteeth #6 through #11 appeared normal without signs ofleukoplakia or erythroplasia (Fig. 3). Final restorations in-cluded full-coverage metal-ceramic crowns on teeth #6through #9 and #11 and the implant crown at site #10(Fig. 4). A protective occlusal guard was also made and de-livered. At the completion of definitive therapy in January2011, the patientwas transferred to the private periodontaloffice of one of the authors (LMM) for maintenance care,where he was placed on a 4-month maintenance schedule.At his fourth maintenance appointment in March 2012,leukoplakic lesions were observed on the facial and palatalgingival margins of teeth #7 through #9 (Fig. 5). Periapicalradiographs taken at that time were unremarkable (Fig. 6).On additional questioning, the patient denied a family his-tory of cancer and reported that he had never been diag-nosed and treated for cancer himself. A semilunarexcisional biopsy of the marginal and papillary gingivaon the facial and palatal surfaces of teeth #7 through #9was subsequently performed, fixed in 10% formaldehyde,and submitted to a local oral and maxillofacial pathologyFIGURE 1 Initial clinical presentation in 2008.

FIGURE 2 Initial complete radiographic survey in 2008.

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laboratory for histologic examination and diagnosis. Be-cause somemargins of this initial biopsy specimenwere notdistinct, a second more extensive excisional biopsy involv-ing only the facial tissue from the same teeth was per-formed 1 week later. A final diagnosis of a superficiallyinvasive, moderately differentiated squamous cell carci-noma was made (Fig. 7).The patient was notified of the diagnosis and referred to

the UTHSCSA Department of Otolaryngology for addi-tional evaluation and treatment. In May 2012, the otolar-yngology report described the lesion as velvety gingivalmucosa surrounding tooth #8 and extending to tooth #7and #9. Leukoplakia was also noted along the gingivalborder of tooth #8. The lesion did not appear to extend intothe vestibule. There was no evidence of peripheral lymph-adenopathy, bone erosion, or metastatic disease to theneck. A partial en blocmaxillectomy from canine to caninewas performed, and an interim dental prosthesis was fix-ated to the palate with one bone screw (Fig. 8). Intraoper-ative frozen sections of the specimen revealed superficiallyinvasive, moderately differentiated squamous cell carci-noma of the maxillary gingiva around teeth #7 and #8

with clear margins. During the initial healing period,several small sequestra of bone were removed withoutconsequence. Amaxillary rotational path removable den-tal prosthesis was fabricated as the patients final pros-thesis (Fig. 9). Currently, the patient is seen every 4months in the office of the private periodontist (LMM)for periodontal maintenance and by the Department ofOtolaryngology every 6 months.

DiscussionSquamous cell carcinoma develops from the epithelial lin-ing of the oral cavity and accounts for>90% of malignantlesions in the mouth. The incidence for the most commonlocations for oral squamous cell carcinoma are reported as

FIGURE 3a and 3b Clinical presentation 6.5 months after crown-lengthening surgery with provisional restorations.

FIGURE 4 Final restorations for teeth #6 through #11.

FIGURE 5 Leukoplakia observed at the gingival and papillary margins onthe facial (5a) and palatal (5b) surfaces of teeth #7 through #9.

FIGURE 6a and 6b Periapical radiographs of teeth #7 through #9 taken atthe time of biopsy.

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FIGURE 7 Hematoxylin and eosin histomicro-graphs of moderately differentiated squamouscell carcinoma demonstrating an abundant in-flammatory cell infiltrate and nuclear atypia of theepithelial cells. 7a Original magnification 10. 7bOriginal magnification 40.

FIGURE 8a Prior to the maxillectomy. 8b Afterremoval of teeth #6 through #9 and #11 anddental implant for site #10. 8c Partial en blocmaxillectomy for teeth #6 through #11 com-pleted. 8d Interim removable dental prosthesisfixated to the palate with one bone screw.

FIGURE 9 Facial and occlusal views aftermaxillectomy (9a and 9b) and with the finalprosthesis inserted (9c and 9d).

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35% for the lower lip, 25% for the ventral surface of thetongue, 20% for the floor of the mouth, 15% for the softpalate, 4% for the alveolar ridge and gingiva, and 1% forthe buccal mucosa.9 The majority of gingival squamouscell carcinomas occur in the mandible, specifically in theposterior region (69%).10

Historically, the twomajor etiologic factors in squamouscell carcinoma in the oral cavity are the social habits of to-bacco use and alcohol consumption. More recent epidemi-ologic data suggest that other risk factors may also belinked to the development of squamous cell carcinoma, in-cluding: 1) age, 2) sex, 3) human papilloma virus (HPV16),4) malnutrition, 5) weakened immune system, 6) betelquid, 7) solar radiation, 8) alcoholic mouthrinses, and 9)irritation from dentures.2 Fitzpatrick et al.11 reported thatas many as 25% of 127 gingival squamous cell patientshad received previous surgical treatment, of which 13 hadrecent periodontal surgical treatment and another 67 hadrecent extractions. More recently, Moergel et al.12 retro-spectively investigated the occurrence of squamous cell car-cinomas in the vicinity of dental implants in their ownpatient population and by a systematic review of publishedliterature. Of the 15 patients identified in their population,six had no previous history of carcinoma. Data analyzedfrom 17 selected publications revealed 26 patients diag-nosedwith carcinomas around dental implants, 14 ofwhichhad no previous history of malignancy.12 Risk factors in thiscase report are identified as smoking, alcohol consumption,and a previous history of carcinoma.12

In the present case, the patient reported no tobacco usebut did consume two to three alcoholic drinks perweek, forwhich his relative risk might be calculated to bez3.5 ac-cording to Mashberg and Samit.13 He also falls within theage range, ethnicity, and sex categories for patients athigher risk for squamous cell carcinoma. In addition, hehad received periodontal and dental implant surgical ther-apy in the maxillary anterior as part of his comprehensivecare. What part this therapy may have had in the develop-ment of the carcinoma, if any, cannot be determined. Keyfactors in this patients treatment outcome were early

detection, biopsy, and timely referral. The presence ofa suspicious leukoplakic lesion found during a routine peri-odontal maintenance visit prompted the immediate atten-tion of the treating periodontist (LMM). The diagnosisrendered from the two biopsies performed was a superfi-cially invasive, moderately differentiated squamous cellcarcinoma. In a systematic review of 23 primary studies,Petti14 estimated the true global prevalence of leukoplakiato be from 1.7% to 2.7%. He further estimated the annualoral cancer incident rate attributable to leukoplakia to be6.2 to 29.1 per 100,000people.14On referral to theDepart-ment of Otolaryngology, the lesion had transformed andwas at that time clinically described as velvety gingivalmucosa. This fits the description of erythroplakia, whichis defined by the World Health Organization as any lesionof the oral mucosa that presents as bright red velvety pla-ques that cannot be characterized clinically or pathologi-cally as any other recognizable condition.15 A review byReichart and Philipsen16 reported the prevalence of eryth-roplakia to range from 0.02% to 0.83%. Persistent asymp-tomatic erythroplasia is the earliest and most consistentsign of oral carcinoma.13 If, after removal of all possibleetiologic factors, these lesions persist for >14 days, theyshould be biopsied for histologic evaluation. Nine percentof erythroplastic lesions have shown dysplasia, another40% presented as carcinoma in situ, and 51% were diag-nosed as squamous cell carcinoma.16

Treatment for oral squamous cell carcinoma includeschemotherapy, radiation, and surgical resection alongwithpossible radical neck dissection. Surgical resection is typi-cally the primary mode of treatment but can be combinedwith radiation and/or chemotherapy.17 If the lesion is

Summary

Why is this case new information? j This is a case report of a primary gingival squamous cell carcinomathat appeared after comprehensive periodontal and restorativetreatment.

j Leukoplakic lesions are increasingly being associated with thedevelopment of oral squamous cell carcinoma.

j This case emphasizes the periodontists and hygienists role as a firstobserver.

What are the keys to successfulmanagement of this case?

j Continued vigilance and thorough examination of all oral tissues foraberrant changes are necessary.

j There should be early detection, biopsy, and submission to pathologyfor histologic examination and diagnosis.

j Timely referral to the appropriate health care specialty is necessary.j There must be interdisciplinary collaboration in all phases oftreatment.

What are the primary limitations tosuccess in this case?

j Subtle changes in the appearance of oral tissue may be difficult todetect or may even be masked during the course of comprehensiveperiodontal therapy.

AcknowledgmentsThe authors thank Dr. Juliana Robledo, Oral andMaxillo-facial Pathologist at South Texas Oral Pathology, SanAntonio, Texas, and Dr. Frank R. Miller, Department ofOtolaryngology, University of Texas Medicine, Universityof Texas Health Science Center, San Antonio, Texas, fortheir expertise in diagnosis and treatment of the patientscarcinoma. The authors report no conflicts of interest re-lated to this case report.

CORRESPONDENCE:Dr. Michael Mills, 703 Floyd Curl, San Antonio, TX 78229. E-mail:millsm@uthscsa.edu.

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12. Moergel M, Karbach J, Kunkel M, Wagner W. Oral squamous cellcarcinoma in the vicinity of dental implants [published online ahead ofprint March 16, 2013]. Clin Oral Investig; doi:10.1007/s00784-013-0968-5.

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indicates key references.

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