Vol. 114 No. 3 September 2012

CLINICOPATHOLOGIC CONFERENCE

Asymptomatic nodule in the tongueJoão Artur Ricieri Brito, DDS, MSD,a Fabrício Tinôco Alvim de Souza, DDS, MSD,a

Júlio César Tanos de Lacerda, DDS, MSD,b Vanessa Fátima Bernardes, DDS, PhD,a

Carolina Cavaliéri Gomes, DDS, MSD, PhD,c and Ricardo Santiago Gomez, DDS, MSD, PhD,a Belo Horizonte, BrazilUNIVERSIDADE FEDERAL DE MINAS GERAIS AND ODILON BEHRENS HOSPITAL

(Oral Surg Oral Med Oral Pathol Oral Radiol 2012;114:281-283)

A 28-year-old woman, with noncontributory social andmedical histories, was referred to the Oral MedicineService of a public hospital in Belo Horizonte, Brazil;she complained of an asymptomatic nodule at the pos-terior dorsal surface of the tongue. She reported that thelesion had been present for the preceding 6 months.Intraoral examination revealed a well-defined nod-ule, measuring approximately 0.6 � 0.7 cm, anteriorto the circumvallate papillae on the left posteriortongue (Figure 1, A and B).

DIFFERENTIAL DIAGNOSISConsidering the clinical presentation and localization ofthe lesion, we included neurofibroma, neurilemmoma,granular cell tumor, reactive lesions, such as giant cellfibroma or focal fibrous hyperplasia, lingual thyroid, andother lingual papillae alterations, in the differential diag-nosis.

The tongue is a common site for benign connectivetissue lesions. Neurilemmoma, or schwannoma, is anencapsulated submucosal mass, typically localized asan asymptomatic lump in the tongue.1-3 Although theycan arise at any age, the peak incidence is between 25and 55 years of age. Only 25% of all extracranialschwannomas are located in the head and neck, withonly 1% originating in intraoral regions.3 Notably, theseintraoral lesions show a predilection for the tongue, withthe tip being the least affected part. The mobile portion,such as the dorsum, of the tongue is the most commonlyaffected site; schwannoma that affects the base of thetongue is rare.4

aDepartment of Oral Pathology, Faculty of Dentistry, UniversidadeFederal de Minas Gerais, Belo Horizonte, Brazil.bDepartment of Oral Diagnosis and Surgery, Odilon Behrens Hospi-tal, Belo Horizonte, Brazil.cDepartment of Pathology, Biological Sciences Institute, Universi-dade Federal de Minas Gerais, Belo Horizonte, Brazil.Received for publication Aug 15, 2011; returned for revision Oct 21,2011; accepted for publication Nov 5, 2011.© 2012 Elsevier Inc. All rights reserved.2212-4403/$ - see front matter

doi:10.1016/j.oooo.2011.11.003

Another neural tumor usually found in the tongue isneurofibroma. This lesion also manifests as an asymp-tomatic mass. It can appear as multiple lesions or as asolitary lesion, and may arise at any age. Neurofibro-mas are most commonly observed in the skin, and theyrarely affect the oral cavity; however, their presencewithin the oral cavity is frequently associated withneurofibromatosis type 1. The tongue, buccal mucosa,gingiva, and lips have been reported to be affected byneurofibromas, with the tongue being the most commonintraoral site.5,6

The oral granular cell tumor is a benign tumor prob-ably of nerve sheath origin, and can also be found in thetongue. Clinically, it is observed as an asymptomaticmass with intact overlying epithelium; the color of thetumor surface varies from the color of a normal mucosato yellowish. The tongue and the buccal mucosa arecommonly affected intraoral sites, with the posteriorportion of the tongue being the least affected region. Thistumor generally occurs in middle-aged or older adults.7,8

Other lesions that should be considered in the differ-ential diagnosis are giant cell fibroma and focal fibroushyperplasia. Giant cell fibroma is a non-neoplastic orallesion that usually presents as an asymptomatic mass. Itcommonly occurs in patients younger than 30 years,has a slight female predilection, and is found mostfrequently on the gingiva and, to a lesser extent, on thetongue and the buccal mucosa.9 The focal fibrous hy-perplasia, or irritation fibroma, typically presents as a pinknodule with a smooth surface and has a color similar tothat of the surrounding mucosa. The labial mucosa,tongue, and gingiva are common sites. They range in sizefrom tiny lesions to large masses; however, most of themare 1.5 cm or smaller in diameter. The peak incidence isbetween the fourth and sixth decades of life.10

Lingual thyroid may be seen as a tumoral lesionbetween the foramen cecum and epiglottis. As the patientin the present case presented a lesion very anterior to thissite, we have not investigated functioning thyroid tissue inthe neck.

The clinical appearance of the lesion may also lead to

the diagnosis of enlarged or ectopic circumvallate pa-

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pillae, as well as prominent lingual papillae. Anotherpathologic condition that can be included in the differ-ential diagnosis is the subgemmal neurogenous plaque.This alteration typically has a subgemmal location andcan be associated with an erythematous area, ulcer,white patch, or hyperplastic papule, usually located inthe lateral border of the tongue.

DIAGNOSIS AND MANAGEMENTOn the basis of the diagnostic possibilities and consid-

Fig. 1. A and B, Clinical presentation. A well-defined nodulis observed.

Fig. 2. A, Histologic examination revealed a mucosal fragmneural proliferations underneath the taste buds (hematoxylexamination revealed spindle cells organized in cords parallecation �400). C, Immunopositivity for S-100 protein in spinmagnification �100). D, Immunopositivity for S-100 in spindbuds (streptavidin-biotin, original magnification �400).

ering the probable benign nature of the lesion, an ex-

cisional biopsy was performed. The histopathologicalevaluation revealed a mucosal fragment composed ofnormal epithelium, abounding with taste buds, and neu-ral proliferations underneath taste buds. The superficiallayer of the connective tissue consisted of a fibrillarneurofibromalike pattern, with spindle cells organizedin cords parallel to the epithelium, whereas the deepzone was composed of ganglion cells in a neuromalikepattern (Figure 2, A and B). After histopathologicalanalysis, immunohistochemical reaction for S-100 pro-

ior to the circumvallate papillae on the left posterior tongue

posed of normal epithelium abounding with taste buds andeosin stain, original magnification �100). B, Histologic

e epithelium (hematoxylin and eosin stain, original magnifi-lls of the superficial component (streptavidin-biotin, originals involved in the neural proliferation associated with the taste

e anter

ent comin andl to thdle cele cell

tein was performed. This reaction highlighted the bi-

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phasic pattern of the plaque. Both the spindle cells ofthe superficial component and the neural proliferationassociated with the taste buds were S-100 positive(Figure 2, C and D). The findings of both the histologicevaluation and immunohistochemistry were consistentwith the diagnosis of subgemmal neurogenous plaque.

DISCUSSIONThe subgemmal neurogenous plaque was first describedby McDaniel11 in 1999 as a subepithelial nerve plexusassociated with the taste buds and was classified as a smallneural proliferation. Some studies show the presence oftaste buds, lingual foliate papillae, and lymphoid follicleson the posterolateral border of the tongue12; however,little is known about the biological characteristics andclinical relevance of subgemmal neurogenous plaques.

Microscopically, the subgemmal neurogenous plaque ischaracterized by subepithelial aggregates of ganglion cellsand nerve plexus, usually associated with taste buds ofthe tongue.11,12 Immunohistochemistry was performedfor S-100 protein to confirm the biphasic pattern of theplaque, because most of the neural proliferations weresubjacent to taste buds.

Patients who present with subgemmal neurogenousplaques usually do not show clinical signs or symp-toms, although this subgemmal neurogenous plaque isoccasionally associated with an erythematous area, ul-cer, a white patch, a hyperplastic papule, and a burningsensation in the tongue.11-13 Although subgemmal neu-rogenous plaques usually do not have significant clin-ical consequences, their removal is necessary for thedifferential diagnosis with other lesions that affect thisregion, such as benign or malignant neoplasm.13-15

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Ther 2006;2:76-8.3. Patnayak R, Anuradha SV, Uppin SM, Sundaram C, Raju GS,

Jena A. Schwannoma of tongue—a case report and short review

of literature. Acta Oncol 2007;46:265-6.

4. Nakasato T, Kamada Y, Ehara S, Miura Y. Multilobular neurilem-moma of the tongue in a child. AJNR Am J Neuroradiol 2005;26:421-3.

5. Jouhilahti EM, Visnapuu V, Soukka T, Aho H, Peltonen S, Hap-ponen RP, et al. Oral soft tissue alterations in patients with neuro-fibromatosis. Clin Oral Investig 2012;16:551-8.

6. Zwane NP, Noffke CE, Raubenheimer EJ. Solitary oral plexi-form neurofibroma: review of literature and report of a case. OralOncol 2011;47:449-51.

7. Nagaraj PB, Ongole R, Bhujanga-Rao BR. Granular cell tumorof the tongue in a 6-year-old girl—a case report. Med Oral PatolOral Cir Bucal 2006;11:E162-4.

8. Andrade ES, Filho JR, Rocha NS, Neto IC, Camargo IB. Isolatedintra-oral granular cell tumor: report of two cases and review ofthe literature. Acta Odontol Latinoam 2010;23:99-104.

9. Kuo RC, Wang YP, Chen HM, Sun A, Liu BY, Kuo YS.Clinicopathological study of oral giant cell fibromas. J FormosMed Assoc 2009;108:725-9.

10. Toida M, Murakami T, Kato K, Kusunoki Y, Yasuda S, FujitsukaH, et al. Irritation fibroma of the oral mucosa: a clinicopatholog-ical study of 129 lesions in 124 cases. Oral Medicine & Pathol-ogy 2001;6:91-4.

11. McDaniel RK. Subepithelial nerve plexus (with ganglion cells)associated with taste buds. Oral Surg Oral Med Oral Pathol OralRadiol Endod 1999;87:605-9.

12. Gueiros LA, León JE, Leão JC, Lopes MA, Jorge J, de AlmeidaOP. Subgemmal neurogenous plaque: clinical and microscopicevaluation of 7 cases. Oral Surg Oral Med Oral Pathol OralRadiol Endod 2009;108:920-4.

13. Gueiros LA, Leon JE, Lopes MA, de Almeida OP, Jorge J.Subgemmal neurogenous plaque associated with burning tongue:report of two cases and review of the literature. Int J OralMaxillofac Surg 2008;37:773-6.

14. Val-Bernal JF, Rivadulla I, Garijo MF. Lingual subgemmalneurogenous plaques with pseudoepitheliomatous hyperplasia:incidental pseudomalignant condition. Pathol Int 2006;56:462-5.

15. Triantafyllou A, Coulter P. Structural organization of subgemmalneurogenous plaques in foliate papillae of tongue. Hum Pathol2004;35:991-9.

Reprint requests:

Ricardo Santiago Gomez, DDS, MSD, PhDUniversidade Federal de Minas GeraisFaculdade de OdontologiaSala 3202D Av. Antônio Carlos 6627, PampulhaCEP 31270-901Belo Horizonte, Minas Gerais, Brazil

rsgomez@ufmg.br