Oral Oncology EXTRA (2004) 40 1–4

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CASE REPORT

Malignant transformation of oral lichen planusin lingual location: report of a case

Jos�e Manuel G�andara-Reya,*, M�arcio Diniz Freitasb, Pilar G�andara Vilab,Andr�es Blanco Carri�ona, Jos�e Manuel Su�arez Penarandac,Abel Garcia Garciad

aUniversity of Santiago de Compostela, Facultad de Odontologia, Calle Entrerrios s/n,15705-Santiago de Compostela, SpainbOral Surgery and Oral Medicine Unit, School of Dentistry, University of Santiago de Compostela, SpaincDepartament of Pathology, Complejo Hospitalario Universitario de Santiago, SpaindDepartament of Maxillofacial Surgery, Complejo Hospitalario Universitario de Santiago, Spain

Received 26 September 2003; accepted 29 September 2003

Summary One of the most controversial aspects of oral lichen planus (OLP) is itsmalignant potential. There have been a few well-documented reports of malignanttransformation in the absence of known exposure to exogenous carcinogens. The aimof this study is to report the case of a patient with a lesion previously diagnosedas oral lichen planus, who developed squamous cell carcinoma in the same location,in the absence of known exposure to exogenous carcinogens.

�c 2003 Elsevier Ltd. All rights reserved.

KEYWORDSOral lichen planus;

Squamous cell carcinoma;

Malignant transformation

Introduction

Lichen planus is a chronic mucocutaneousinflammatory disease of unknown etiology, fre-quently occurring in oral locations, with distinctivethough not entirely diagnostic clinical and histo-pathological characteristics.1

One of the most controversial aspects of orallichen planus (OLP) is its malignant potential.2;3

Various studies have suggested that OLP has po-tential for malignant transformation, althoughthere is consensus that the probability of suchtransformation is low.3 There have been a few

* Corresponding author. Tel.: +34-981-56-31-00x12357; fax:+34-981-563-100.

E-mail address: cigandar@usc.es (J.M. G�andara-Rey).

1741-9409/$ - see front matter �c 2003 Elsevier Ltd. All rights reserdoi:10.1016/S1741-9409(03)00002-5

well-documented reports of malignant transfor-mation in the absence of known exposure toexogenous carcinogens.4;5

Here, we report the case of a patient with alesion previously diagnosed as oral lichen planus,who developed squamous cell carcinoma in thesame location, in the absence of known exposureto exogenous carcinogens.

Case report

A 45-year-old woman who in June 1995 was re-ferred to the Oral Medicine Unit of the DentistryFaculty of the University of Santiago de Compostelafor evaluation of a red and white lesion in linguallocation. Oral examination revealed a reddishatrophic lesion with a small central erosion

ved.

2 J.M. G�andara-Rey et al.

surrounded by raised whitish streaks, approxi-mately 2 cm in diameter, located on the dorsalsurface of the right half of the tongue (Fig. 1). Inaddition, atrophic zones with whitish streaks werepresent on the alveolar mucosa of the upper jaw.The patient reported itchiness in tongue and gums,first noted about 3 months previously. She was anon-smoker and moderate social drinker with norelevant antecedents. Following a clinical diagnosisof atrophic-erosive lichen planus, biopsies weretaken for histopathological studies. These revealed(a) stratified squamous epithelium with areas ofacanthosis and hyperkeratosis, (b) the presence ofa dense lymphocytic inflammatory infiltrate alongthe epithelium/connective-tissue interface, (c)hydropic degeneration of the basal layer, and (d)some necrotic keratinocytes (Fig. 2). Dysplasia wasnot observed. Immunofluorescence tests fordetection of IgG, IgA, IgM, IgE and C3 were nega-tive. The diagnosis of atrophic-erosive lichenplanus was thus confirmed, and treatment wascommenced with 0.3% aqueous triamcinoloneacetonide as mouthwash, three times daily for 15days. At next appointment the patient showed

Figure 1 (a) Dorsal surface of the patient’s tongue,showing atrophic-erosive zones surrounded by whitishstreaks. (b) Detail of Fig. 1a, showing an atrophic-erosivelesion on the dorsal surface of the right half of the ton-gue. Note the whitish streaks with reticular distribution,characteristic of lichen planus.

symptom improvement, but examination revealederosive zones, so that we commenced topicaltreatment with 0.05% clobetasol propionate in ora-base paste, three times daily for another 15 days.This examination also revealed that the ulceratedareas had disappeared, leaving an atrophic epi-thelium with whitish streaks. After 15 days, thetreatment was stopped. The patient returned forregular check-ups every 3 months, showing alter-nating lesion recurrence and remission on treat-ment. In June 1998 the patient stopped attendingcheck-ups.

In October 2002, the patient consulted againwith a whitish exophytic lesion on the back of the

Figure 2 (a) Stratified flat epithelium showing acantho-sis and slight hyperkeratosis. Note also the hydropicdegeneration of the basal layer, and the dense lympho-cytic inflammatory infiltrate along the epithelium/connective-tissue interface. Dysplasia is not seen.(Hematoxylin-eosin, 10·). (b) Higher-magnification ofthe micrograph in (a), showing the hydropic degenera-tion of the basal layer, and the inflammatory infiltratealong the epithelium/connective-tissue interface. Somenecrotic keratinocytes can be seen. (Hematoxylin-eosin,40·).

Figure 3 Squamous cell carcinoma on the back of thetongue, arising 7 years after the initial diagnosis of orallichen planus.

3Malignant transformation of OLP

tongue, with areas of redness and ulceration,approximately 3 cm in diameter (Fig. 3). Biopsyrevealed squamous cell carcinoma (Fig. 4), atclinical stage I (T2 N0 M0). The patient was re-ferred to the Maxillofacial Surgery Service of the

Figure 4 (a) Biopsy of the squamous cell carcinomalesion, showing well-differentiated squamous cells.(Hematoxylin-eosin, 10·). (b) Higher-magnification de-tail of the micrograph in (a). (Hematoxylin-eosin, 40·).

University Clinical Hospital of Santiago de Com-postela, where the lesion was removed and thepatient underwent radiotherapy (total dose 70 Gy,in a megavoltage cobalt-60 unit). The patient cur-rently shows no sign of relapse, although radio-therapy has led to an oral mucositis of difficultmanagement.

Discussion

The World Health Organization (WHO) definesoral lichen planus as a precancerous condition,associated with an increase in the risk of oralcancer.6 As noted, various authors have suggestedthat OLP has malignant potential,3;7;8 but otherauthors have disputed this view.2;9–11 In this con-nection, Krutchkoff et al.,4 in a review of 223 casespublished between 1950 and 1976, pointed out thatonly 15 met strict requirements for considerationas cases of malignant transformation of an existingOLP lesion.

More recently, van der Meij et al.5 have pub-lished a meta-analytical review of articles dealingwith malignant transformation of oral lichen planusin the English-language literature over the period1977–1998. Of the 98 documented cases, the au-thors found that only 33 (34%) met the criteria ofKrutchkoff et al.4 Of the 65 cases that did not meetthese criteria, 20 were inadequately documentedas regards histopathology, one as regards clinicalcharacteristics and course, 33 as regards both his-topathology and clinical characteristics; in addi-tion, four had been followed up for less than 2years, and seven patients were smokers.

Here we have reported a case in which the pa-tient developed a squamous cell carcinoma in thesame location as a lesion diagnosed 7 years previ-ously as OLP. Clinical appearance was an isolatedsingle unilateral lesion, with absence of bilaterallesions in the jugal mucosa. This is not the typicalpresentation of OLP;2 however, the diagnosis wasconfirmed by the combination of the clinicalappearance (atrophic lesion surrounded by raisedwhitish streaks) and histopathological findings inline with those characteristic of OLP.2;11

The patient showed the atrophic-erosive form ofOLP, which some authors have suggested to be thatwith highest risk of malignant transformation.3;12

Some researchers have suggested that theseatrophic-erosive forms predispose the oral mucosato the effects of other carcinogenic agents;13;14 asfar as we know, however, our patient does nothave a history of exposure to any major exogenouscarcinogen. In any case, malignant transformationdoes not appear to be exclusive to atrophic-erosive

4 J.M. G�andara-Rey et al.

OLP: Silverman et al.,15 in a study of 214 cases ofOLP followed up on average for 7.5 years, foundfive of malignant transformation, three affectingerosive lesions, one affecting an atrophic lesion,and one affecting a reticular lesion. Lo Muzioet al.16 reported 14 cases of squamous cell carci-noma associated with OLP lesions, which in 12 ofthe cases were plaque-like.

The body-location statistics of OLP-associatedsquamous cell carcinomas (SCCs) are of coursedifferent from those of SCCs in general. SCCs onthe back of the tongue are very infrequent,accounting for less than 5% of all oral carcinomas.17

However, malignant transformation of OLP in thislocation is relatively frequent, and some authorshave suggested that this location is a significantrisk factor.18

Malignant transformation of OLP appears tobe independent of exogenous risk factors. ThusGarcia-Pola et al.19 reported 4 cases of SCC inthe same location as OLP lesions, but only in oneof these cases was the patient a smoker. Similarly,in the present case (in which the OLP was conclu-sively diagnosed, and in which we confirmed thatthe SCC developed in exactly the same location asthe OLP) the patient had no history of exposure toexogenous carcinogens. The present case thussupports the view that OLP may undergo malignanttransformation, and that this does not requireexogenous carcinogens. In conclusion, despite thecurrent uncertainty about the probability ofmalignant transformation of OLP, the fact thatsuch transformation may occur seems increasinglycertain.16;19;20 This argues for a need to performroutine monitoring of some subsets of OLP patient,including patients with atrophic-erosive lesions onthe tongue.

References

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