Oral Oncology EXTRA (2004) 40 14

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CASE REPORT

Malignant transformation of oral lichen planus

in lingual location: report of a case

rcio Diniz Freitasb, Pilar Ga

Manuel Ga

ndara-Reya,*, Ma

ndara Vilab,
Jose

s Blanco Carrio

na, Jose

Manuel Sua

rez Pen
arandac,
Andre
Abel Garcia Garciad
a

University of Santiago de Compostela, Facultad de Odontologia, Calle Entrerrios s/n,

15705-Santiago de Compostela, Spain
b
Oral Surgery and Oral Medicine Unit, School of Dentistry, University of Santiago de Compostela, Spain
c
Departament of Pathology, Complejo Hospitalario Universitario de Santiago, Spain
d
Departament of Maxillofacial Surgery, Complejo Hospitalario Universitario de Santiago, Spain

Received 26 September 2003; accepted 29 September 2003

KEYWORDS

Summary One of the most controversial aspects of oral lichen planus (OLP) is its
malignant potential. There have been a few well-documented reports of malignant
transformation in the absence of known exposure to exogenous carcinogens. The aim
of this study is to report the case of a patient with a lesion previously diagnosed
as 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.

Oral lichen planus;

Squamous cell carcinoma;
Malignant transformation


Introduction

Lichen planus is a chronic mucocutaneous

inflammatory disease of unknown etiology, frequently occurring in oral locations, with distinctive
though not entirely diagnostic clinical and histopathological characteristics.1
One of the most controversial aspects of oral
lichen planus (OLP) is its malignant potential.2;3
Various studies have suggested that OLP has potential for malignant transformation, although
there is consensus that the probability of such
transformation is low.3 There have been a few
*

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

+34-981-563-100.

ndara-Rey).
E-mail address: cigandar@usc.es (J.M. Ga

well-documented reports of malignant transformation in the absence of known exposure to

exogenous carcinogens.4;5
Here, we report the case of a patient with a
lesion previously diagnosed as oral lichen planus,
who developed squamous cell carcinoma in the
same location, in the absence of known exposure
to exogenous carcinogens.

Case report
A 45-year-old woman who in June 1995 was referred to the Oral Medicine Unit of the Dentistry
Faculty of the University of Santiago de Compostela
for evaluation of a red and white lesion in lingual
location. Oral examination revealed a reddish
atrophic lesion with a small central erosion

1741-9409/$ - see front matter c 2003 Elsevier Ltd. All rights reserved.
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2
surrounded by raised whitish streaks, approximately 2 cm in diameter, located on the dorsal
surface of the right half of the tongue (Fig. 1). In
addition, atrophic zones with whitish streaks were
present on the alveolar mucosa of the upper jaw.
The patient reported itchiness in tongue and gums,
first noted about 3 months previously. She was a
non-smoker and moderate social drinker with no
relevant antecedents. Following a clinical diagnosis
of atrophic-erosive lichen planus, biopsies were
taken for histopathological studies. These revealed
(a) stratified squamous epithelium with areas of
acanthosis and hyperkeratosis, (b) the presence of
a dense lymphocytic inflammatory infiltrate along
the epithelium/connective-tissue interface, (c)
hydropic degeneration of the basal layer, and (d)
some necrotic keratinocytes (Fig. 2). Dysplasia was
not observed. Immunofluorescence tests for
detection of IgG, IgA, IgM, IgE and C3 were negative. The diagnosis of atrophic-erosive lichen
planus was thus confirmed, and treatment was
commenced with 0.3% aqueous triamcinolone
acetonide as mouthwash, three times daily for 15
days. At next appointment the patient showed

Figure 1 (a) Dorsal surface of the patients tongue,

showing atrophic-erosive zones surrounded by whitish
streaks. (b) Detail of Fig. 1a, showing an atrophic-erosive
lesion on the dorsal surface of the right half of the tongue. Note the whitish streaks with reticular distribution,
characteristic of lichen planus.

ndara-Rey et al.
J.M. Ga
symptom improvement, but examination revealed
erosive zones, so that we commenced topical
treatment with 0.05% clobetasol propionate in orabase paste, three times daily for another 15 days.
This examination also revealed that the ulcerated
areas had disappeared, leaving an atrophic epithelium with whitish streaks. After 15 days, the
treatment was stopped. The patient returned for
regular check-ups every 3 months, showing alternating lesion recurrence and remission on treatment. In June 1998 the patient stopped attending
check-ups.
In October 2002, the patient consulted again
with a whitish exophytic lesion on the back of the

Figure 2 (a) Stratified flat epithelium showing acanthosis and slight hyperkeratosis. Note also the hydropic
degeneration of the basal layer, and the dense lymphocytic inflammatory infiltrate along the epithelium/
connective-tissue interface. Dysplasia is not seen.
(Hematoxylin-eosin, 10). (b) Higher-magnification of
the micrograph in (a), showing the hydropic degeneration of the basal layer, and the inflammatory infiltrate
along the epithelium/connective-tissue interface. Some
necrotic keratinocytes can be seen. (Hematoxylin-eosin,
40).

Malignant transformation of OLP

3
University Clinical Hospital of Santiago de Compostela, where the lesion was removed and the
patient underwent radiotherapy (total dose 70 Gy,
in a megavoltage cobalt-60 unit). The patient currently shows no sign of relapse, although radiotherapy has led to an oral mucositis of difficult
management.

Discussion
Figure 3 Squamous cell carcinoma on the back of the
tongue, arising 7 years after the initial diagnosis of oral
lichen planus.

tongue, with areas of redness and ulceration,

approximately 3 cm in diameter (Fig. 3). Biopsy
revealed squamous cell carcinoma (Fig. 4), at
clinical stage I (T2 N0 M0). The patient was referred to the Maxillofacial Surgery Service of the

Figure 4 (a) Biopsy of the squamous cell carcinoma

lesion, showing well-differentiated squamous cells.
(Hematoxylin-eosin, 10). (b) Higher-magnification detail of the micrograph in (a). (Hematoxylin-eosin, 40).

The World Health Organization (WHO) defines

oral lichen planus as a precancerous condition,
associated with an increase in the risk of oral
cancer.6 As noted, various authors have suggested
that OLP has malignant potential,3;7;8 but other
authors have disputed this view.2;911 In this connection, Krutchkoff et al.,4 in a review of 223 cases
published between 1950 and 1976, pointed out that
only 15 met strict requirements for consideration
as cases of malignant transformation of an existing
OLP lesion.
More recently, van der Meij et al.5 have published a meta-analytical review of articles dealing
with malignant transformation of oral lichen planus
in the English-language literature over the period
19771998. Of the 98 documented cases, the authors found that only 33 (34%) met the criteria of
Krutchkoff et al.4 Of the 65 cases that did not meet
these criteria, 20 were inadequately documented
as regards histopathology, one as regards clinical
characteristics and course, 33 as regards both histopathology and clinical characteristics; in addition, four had been followed up for less than 2
years, and seven patients were smokers.
Here we have reported a case in which the patient developed a squamous cell carcinoma in the
same location as a lesion diagnosed 7 years previously as OLP. Clinical appearance was an isolated
single unilateral lesion, with absence of bilateral
lesions in the jugal mucosa. This is not the typical
presentation of OLP;2 however, the diagnosis was
confirmed by the combination of the clinical
appearance (atrophic lesion surrounded by raised
whitish streaks) and histopathological findings in
line with those characteristic of OLP.2;11
The patient showed the atrophic-erosive form of
OLP, which some authors have suggested to be that
with highest risk of malignant transformation.3;12
Some researchers have suggested that these
atrophic-erosive forms predispose the oral mucosa
to the effects of other carcinogenic agents;13;14 as
far as we know, however, our patient does not
have a history of exposure to any major exogenous
carcinogen. In any case, malignant transformation
does not appear to be exclusive to atrophic-erosive

4
OLP: Silverman et al.,15 in a study of 214 cases of
OLP followed up on average for 7.5 years, found
five of malignant transformation, three affecting
erosive lesions, one affecting an atrophic lesion,
and one affecting a reticular lesion. Lo Muzio
et al.16 reported 14 cases of squamous cell carcinoma associated with OLP lesions, which in 12 of
the cases were plaque-like.
The body-location statistics of OLP-associated
squamous cell carcinomas (SCCs) are of course
different from those of SCCs in general. SCCs on
the back of the tongue are very infrequent,
accounting for less than 5% of all oral carcinomas.17
However, malignant transformation of OLP in this
location is relatively frequent, and some authors
have suggested that this location is a significant
risk factor.18
Malignant transformation of OLP appears to
be independent of exogenous risk factors. Thus
Garcia-Pola et al.19 reported 4 cases of SCC in
the same location as OLP lesions, but only in one
of these cases was the patient a smoker. Similarly,
in the present case (in which the OLP was conclusively diagnosed, and in which we confirmed that
the SCC developed in exactly the same location as
the OLP) the patient had no history of exposure to
exogenous carcinogens. The present case thus
supports the view that OLP may undergo malignant
transformation, and that this does not require
exogenous carcinogens. In conclusion, despite the
current uncertainty about the probability of
malignant transformation of OLP, the fact that
such transformation may occur seems increasingly
certain.16;19;20 This argues for a need to perform
routine monitoring of some subsets of OLP patient,
including patients with atrophic-erosive lesions on
the tongue.

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