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kazuistyka / case report

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Oral erythroleukoplakia apotentially

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malignant disorder

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Erytroleukoplakia jamy ustnej schorzenie

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ocharakterze potencjalnie zoliwym

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Natalhia Gabriela de Barros Vieira Guilgen 1, Sylvia Kang1, Maria Helena Martins Tommasi1, Iran Vieira1,
Maria ngela Naval Machado1, Antonio Adilson Soares de Lima1

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Department of Stomatology, School of Dentistry, Universidade Federal do Paran UFPR, Curitiba/PR, Brazil.

Article history: Received: 30.10.2014 Accepted: 18.11.2014 Published: 28.02.2015

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ABSTRACT Erythroleukoplakia or speckled leukoplakia is a painless lesion characterized by an association of white areas inter-
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spersed with red areas, resistant to scratching and with potential for malignancy. It appears most frequently in the oral
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mucosa, vermilion and gums. The etiology of erythroleukoplakia is variable, but may be related to smoking, alcohol-
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ism, microorganisms, and other agents. The objective of this work was to report a case of erythroleukoplakia in a male
patient who was a smoker and an alcoholic. The lesion was detected in the retrocomissural region during a routine
dental examination. The diagnosis was established after histopathological examination of the tissue specimen col-
lected by the excisional biopsy. Otolaryngologists and dentists performing an oral examination should be alert to the
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patients habit of smoking and drinking. Oral biopsy is mandatory to recognize the presence and the severity of epithe-
lial dysplasia, which is a decisive factor for subsequent treatment planning.
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KEYWORDS: Erythroleukoplakia, Leukoplakia, Smoking, Oral Mucosa.

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STRESZCZENIE Erythroleukoplakia or speckled leukoplakia is apainless lesion characterized byan association of white areas inter-
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spersed with red areas, resistant to scratching and with potential for malignancy. It appears most frequently in the oral
mucosa, vermilion and gums. The etiology of erythroleukoplakia is variable, but may be related to smoking, alcohol-
ism, microorganisms, and other agents. The objective of this work was to report acase of erythroleukoplakia in amale
patient who was asmoker and an alcoholic. The lesion was detected in the retrocomissural region during aroutine
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dental examination. The diagnosis was established after histopathological examination of the tissue specimen col-
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lected bythe excisional biopsy. Otolaryngologists and dentists performing an oral examination should be alert to the
This copy is for personal use only - distribution prohibited.

patients habit of smoking and drinking. Oral biopsy is mandatory to recognize the presence and the severity of epithe-
lial dysplasia, which is adecisive factor for subsequent treatment planning.
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SOWA KLUCZOWE: Erytroleukoplakia, leukoplakia, nikotynizm, bona luzowa jamy ustnej.

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develop in the oral mucosa, penis or vulva. According to the World

INTRODUCTION
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Health Organization, leukoplakia is described as a white lesion

that cannot be removed by scraping. It is caused by local irritating
Leukoplakia (leukos meaning white; plakia meaning patch) is a factors (smoking and alcohol) which stimulate the formation of
disease characterized by the presence of a white plate which con- hyperkeratosis. The incisional biopsy is essential for diagnosis [1].
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stitutes an expression of alteration in the keratinization process

[1]. In 1877, Schwimmer was the first author that used this term The natural history of oral leukoplakia remains poorly understo-
to describe a white plaque on the tongue [2]. However, it can also od [4]. Oral leukoplakia can develop into a carcinoma. Malignant
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20 DOI: 10.5604/20845308.1132406 WWW.OTORHINOLARYNGOLOGYPL.COM

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transformation rates of oral leukoplakia range from 0.13 to 17.5%.

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The risk factors of malignant transformation in the buccal muco-
sa and labial commissure are male gender with chewing tobacco
or smoking in some countries such as India, or older age and/or

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being a non-smoking female in other countries [5].

According to the clinical appearance, leukoplakia can be classi-

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fied into four types: early or thin, homogenous or thick, granular

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or verruciform and speckled or erythroleukoplakia. Each sub-

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division has a different malignant transformation potential. For
example, thin leukoplakia often becomes malignant without cli-
nical changes. Thick leukoplakia undergoes malignant transfor-

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mation in 17% of cases [6].
Figure 1. Erythroleukoplakia - White plaque with red areas in the buccal mucosa
Histologically, leukoplakia can show several epithelial changes.

The presence of dysplasia with varying degrees of severity is very
common. A study developed by Waldron and Shafer demonstra-
ted that 19.9% of leukoplakia cases had some degrees of dysplasia,
3.1% were frank carcinoma, 4.6% showed severe dysplasia or car-
cinoma in situ, and 12.2% showed mild to moderate dysplasia [7].
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During the anamnesis, the patient reported tenderness in the po-
sterior teeth and a temporo-mandibular joint pain. Physical exa-
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mination revealed the presence of a removable partial denture
that was not adapted. Dental caries, residual roots, periodontal
disease, bad breath, poor oral hygiene, nicotine stomatitis, and

Erythroplakia is another oral lesion considered as a potentially smokers melanosis were observed during oral examination. In
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malignant disorder which precedes the development of cancer. addition, white lesions interspersed with the reddish ones were
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Erythroplakia is a red lesion characterized by patches, plaques or located bilaterally in the retrocomissural region. The lesions had
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erosions of various dimensions that affect regions of the hard and a triangular shape and were painless. Clinically, the lesion on the
soft palate, floor of the mouth, tongue and retromolar region. It right side was more apparent during the examination (Figure 1).
can also be caused by local irritating factors, such as tobacco and
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alcohol [8]. Erythroplakia, when associated with leukoplakia, has Clinically, the lesion was a plate with approximate size of 20 mm,
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a higher malignant potential and shall be called erythroleukopla- and white color interspersed with red areas. It had a slightly ro-
kia [3]. In order to establish the diagnosis, it is also necessary to ugh consistency with sessile insertion and absence of symptoms.
perform a biopsy and this should be done in the perilesional re-
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gion. The histopathological features of erythroleukoplakia include The diagnosis of chronic hyperplasic candidiasis was conside-
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epithelial atrophy with varying degrees of atypia [9]. red initially because of the clinical appearance of the lesions, and
smoking and drinking habits. The patient was not aware of the
The treatment of choice for oral leukoplakia and erythroplakia is existence of lesions. Therefore, it was not possible to establish the
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surgery. There exists a possibility of recurrence so monitoring of time of lesion development. During medical interview, the patient
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the patient should be permanent. The first step in the treatment reported that he had drunk alcohol every day. In addition, he had
of oral leukoplakia and eritroplasias is advising the patient to stop smoked on average two packs of cigarettes a day for over 20 years.
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his/her habits (alcohol and tobacco). The treatment of these lesions

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may be conservative or surgical [10]. Oral precancerous lesions Before dental treatment, the patient underwent an incisional biop-
can be eradicated by surgical excision [11], laser surgery [12], or sy of the lesion. However, it was decided to completely remove the
photodynamic therapy [13]. The aim of this paper was to report lesions because of their location and size. The diagnostic hypothe-
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on a case of erythroleukoplakia that developed bilaterally in the ses were chronic hyperplasic candidiasis or erythroleukoplakia.
buccal mucosa of an alcoholic and smoker.
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The patient was medicated with 200 mg of Ibuprofen in a thera-

peutic regimen of doses administered every 12 hours for 3 days.
CASE REPORT Seven days later, he returned for suture removal. No change was
observed during the repair process.
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Male, 37 years old, presented to the clinic of the School of Den-

tistry (Universidade Federal do Paran, Curitiba, Brazil) com- The lesions were sent for histopathological examination. The ma-
plaining of pain in the posterior teeth and difficulty in chewing. terial was processed in the laboratory and stained by two stains:
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POLSKI PRZEGLD OTORYNOLARYNGOLOGICZNY, TOM 4, NR 1 (2015), s. 20-24 21

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hematoxylin and eosin and Grocott. The hematoxylin and eosin

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revealed a fragment of oral mucosa lined by parakeratinized epi-
thelial tissue with areas of hyperplasia, acanthosis, and exocytosis.
Some cells of the basal layer exhibited enlarged nuclei with con-

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densed chromatin, nuclear atypia and mitotic figures (Figures 2
and 3). The underlying connective tissue showed chronic inflam-
matory infiltrate with a predominance of lymphocytes. No fungal

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structures were identified with Grocott staining (Figure 4). That

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finding excluded the possibility of fungal infection of the lesion.

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According to those histological findings, the diagnosis of mild
epithelial dysplasia was established.

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The patient was advised to stop or reduce the consumption of ci-
garettes and alcohol because of the diagnosis of epithelial dyspla-
sia and nicotine stomatitis. Periodic examinations were planned

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Figure 2. Epithelial tissue with hyperplasia, acanthosis, and exocytosis (HE x100)
but the patient did not return for the follow-up.

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DISCUSSION
Oral leukoplakia is defined as a white plate which cannot be clini-
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cally or histologically characterized as any other disease [3]. Thus,

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leukoplakia is essentially a clinical entity. Its diagnosis is based on

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exclusion of other injuries, because this entity has no specific hi-

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stopathological features [14]. It is considered the most common

potentially malignant disorder that develops in several areas of the
oral mucosa. However, the buccal mucosa, lower lip and edge of
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the tongue are the most affected areas. Erythroplakia is defined

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as no red plate diagnosed histologically or any other condition [8].

This lesion has a high degree of malignancy compared with oral
leukoplakia or nevus [15].
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Figure 3. Nuclear changes and cromatin condensation in epithelial cells of the When the lesion presents with red and white mucosal alterations
basal stratum (HE x400). concomitantly, the term erythroleukoplakia is used. However, in
erythroleukoplakia lesions, the red or erythroplakia areas have
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been shown to be most likely to demonstrate dysplastic changes

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compared to the white hyperkeratotic areas [16].

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It is believed that the etiology of this lesion is related to habits such

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as smoking and when associated with alcohol consumption, the

probability of malignant transformation is increased. They are
generally considered idiopathic lesions and without apparent ori-
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gin. The erythroleukoplakias are more prevalent in men, due to

an increased association with smoking [17].
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A study by Feller et al. [18] revealed that the prevalence of eryth-

roleukoplakias is approximately 11.2%. The same study observed
that it mainly affects males and individuals in their seventh deca-
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de of life. In our case report, the patient was male, 37 years of age,
smoked cigarettes (40 cigarettes a day) and drank alcohol (daily).
Figure 4. No fungal structure in epithelial tissue (Grocott x100). The anatomical regions most affected by potentially malignant
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22 WWW.OTORHINOLARYNGOLOGYPL.COM
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disorders are: buccal mucosa (28.8%), floor of the mouth (18.3%), The histopathological findings that characterize leukoplakia are

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alveolar ridge and gums (17.3%), and tongue (12.0%). The patient hyperkeratosis, dysplasia, chronic inflammatory infiltrate, acan-
reported here had a lesion within the buccal mucosa. thosis, and atrophy. According to the classification of dysplasia
(mild, moderate and severe), two lesions with severe dysplasia are

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The initial diagnosis of this case was chronic hyperplasic candi- already considered carcinoma in situ [8]. However, histopatho-
diasis due to the clinical aspect of the lesion and patients habits of logical examination revealed that this lesion presented hyperpa-
cigarette smoking and alcohol drinking. Some white lesions need rakeratosis, acanthosis, atrophy, mild dysplasia, and chronic in-

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to be considered in the differential diagnosis of oral potentially flammatory infiltrate. In addition, staining by Grocott ruled out
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malignant disorders with white coloration, i.e.: pseudomembrano- the possibility of chronic hyperplastic candidiasis.

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us and chronic hyperplasic candidiasis, linea alba, lichen planus,
leukoedema, reactive hyperkeratosis due to oral mordiscamento Erythroleukoplakia can be treated by surgical removal at the time
and spongy white nevus [8]. Initially, pseudomembranous can- of the biopsy, or surgical laser, photodynamic therapy, and chemo-

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didiasis was excluded because it was not possible to remove the preventive agents [11,12,13,19]. Thus, it is necessary that the dia-
lesion with scraping. In addition, the lesions of the patient had no gnosis is made in advance and safely. In cases where moderate to
clinical features consistent with the diagnosis of other possibili- severe dysplasia is evident, complete surgical removal and periodic

ties mentioned above. The only exception was reaction hyperke-
ratosis due to cheek bite. However, the patient reported no pa-
rafunctional habit, nor was there any evidence found during the
clinical examination. Thus, one was not able to rule out the po-
ssibility of a bite injury in the same region during sleep, since the
patient complained of pain in the TMJ region.
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monitoring are recommended due to high rates of recurrence [8].
The importance of early diagnosis of erythroleukoplakia follows
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from a potential for dysplastic changes and progression to frank
carcinoma. Because of the rate and timing of progression of these
lesions into cancerous ones, the combination of clinical and hi-

stological examination is critical for accurate diagnosis, and the

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As chronic hyperplastic candidiasis was the first diagnostic hy- results from these assessments are used to determine specific tre-
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pothesis, we opted to perform an incisional biopsy. However, atment [20]. Thus, dentists and otolaryngologists need to be alert
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during surgery it was decided to completely remove the lesions during the examination of the mouth to the habit of smoking and
due to their characteristics (small size and injury without a mali- drinking of the patient. Oral biopsy is mandatory to recognize the
gnancy). The histological analysis confirmed that the lesion had presence and the severity of epithelial dysplasia, which is a deci-
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epithelial dysplasia. sive factor for subsequent treatment planning.

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References
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1. Kramer IR, Lucas RB, Pindborg JJ, Sobin LH. Definition of leukoplakia and related lesions: an aid to studies on oral precancer. Oral Surg Oral Med Oral Pa-
pe

thol. 1978; 46(4):518-39.

2. Schwimmer E. Die idiopathischen Schleimhautplaques der Mundhhle (Leukoplakia buccalis) Arch Dermat Syph. 1877;9:570611.
3. Warnakulasuriya S, Johnson NW, van der Waal I. Nomenclature and classification of potentially malignant disorders of the oral mucosa. J Oral Pathol Med.
2007; 36(10):575-80.
for
-

4. Napier SS, Speight PM. Natural history of potentially malignant oral lesions and conditions: an overview of the literature. J Oral Pathol Med. 2008;37:110.
This copy is for personal use only - distribution prohibited.

5. Amagasa T, Yamashiro M, Uzawa N. Oral premalignant lesions: from a clinical perspective. Int J Clin Oncol. 2011; 16(1):5-14.
y is

6. Mortazavi H, Baharvand M, Mehdipour M. Oral potentially malignant disorders: an overview of more than 20 entities. J Dent Res Dent Clin Dent Prospects.
2014; 8(1):6-14.
7. Waldron CA, Shafer WG. Leukoplakia revisited. A clinicopathologic study 3256 oral leukoplakias. Cancer. 1975; 36:138692.
8. Neville BW, Day TA. Oral cancer and precancerous lesions. CA Cancer J Clin. 2002; 52(4):195-215.
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9. Jesper R. Prognosis of oral pre-malignant lesions: significance of clinical, histopathological, and molecular biological characteristics. Crit Rev Oral Biol Med.
2003;14(1):47-62.
is c

10. Abidullah M, Kiran G, Gaddikeri K, Raghoji S, Ravishankar TS. Leuloplakia - review of a potentially malignant disorder. J Clin Diagn Res. 2014; 8(8):ZE01-4.
11. Vedtofte P, Holmstrup P, Hjrting-Hansen E, Pindborg JJ. Surgical treatment of premalignant lesions of the oral mucosa. Int J Oral Maxillofac Surg. 1987;
16(6):656-64.
Th

12. Pinheiro AL, Frame JW. Surgical management of premalignant lesions of the oral cavity with the CO2 laser. Braz Dent J. 1996; 7:1038.
13. M Clin Dent FV, Al-Kheraif AA, Qadri T, Hassan MI, Ahmed A, Warnakulasuriya S, Javed F. Efficacy of photodynamic therapy in the management of oral
premalignant lesions. A systematic review. Photodiagnosis Photodyn Ther. 2014 (In press).
14. Ges C, Weyll B, Sarmento VA, Ramalho LMP. Diagnstico diferencial e manejo da leucoplasia bucal Caso clnico: acompanhamento de 4 anos. RGO.
-

POLSKI PRZEGLD OTORYNOLARYNGOLOGICZNY, TOM 4, NR 1 (2015), s. 20-24 23

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ed
ibit
2007; 55(1):95-100.

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15. Shafer WG, Waldron CA. Erythroplakia of the oral cavity. Cancer 1975;36:10218.
16. Lockhart PB, Norris CM Jr, Pulliam C. Dental factors in the genesis of squamous cell carcinoma of the oral cavity. Eur J Cancer B Oral Oncol. 1998; 34:1339.
17. Silverman S Jr, Gorsky M, Lozada F. Oral leukoplakia and malignant transformation. A follow-up study of 257 patients. Cancer. 1984; 53(3):563-8.

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18. Feller L, Altini M, Slabbert H. Pre-malignant lesions of the oral mucosa in a South African sample - a clinicopathological study. J Dent Assoc S Afr. 1991;
46(5):261-5.
19. Sankaranarayanan R, Mathew B, Varghese C, Sudhakaran PR, Menon V, Jayadeep A, Nair MK, Mathews C, Mahalingam TR, Balaram P, Nair PP. Chemopre-

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vention of oral leukoplakia with vitamin A and beta carotene: an assessment. Oral Oncol. 1997; 33(4):231-6.
This copy is for personal use only - distribution prohibited.

20. Jaber MA, Porter SR, Gilthorpe MS, Bedi R, Scully C. Risk factors for oral dysplasiathe role of smoking and alcohol. Eur J Cancer B Oral Oncol. 1999; 35:1516.

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Word count: 1779 Tables: Figures: 4 References: 20

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Access the article online: DOI: 10.5604/20845308.1132406 Full-text PDF: www.otorhinolaryngologypl.com/fulltxt.php?ICID=1132406

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Corresponding author: Antonio Adilson Soares de Lima, Department of Stomatology, School of Dentistry - Universidade Federal do Paran
(UFPR), Rua Prefeito Lothrio Meissner 632, Jardim Botnico, 80170-210 Curitiba - PR Brazil, Telephone: + 55 41 33604050, e-mail: aas.lima@ufpr.br
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Copyright 2015 Polish Society of Otorhinolaryngologists Head and Neck Surgeons. Published by Index Copernicus Sp. z o.o. All rights reserved
Funding: Study was developed through funding by the researchers themselves
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Competing interests: The authors declare that they have no competing interests.
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Cite this article as: de Barros Vieira Guilgen N.G., Kang S., Martins Tommasi M.H., Vieira I, Naval Machado M. A., Soares de Lima A.A.: Oral erythroleukoplakia a
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potentially malignant disorder. Pol Otorhino Rev 2015; 4(1): 20-24

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