Full-text - Polski Przegląd Otorynolaryngologiczny

kazuistyka / case report
Oral erythroleukoplakia – a potentially
malignant disorder
Erytroleukoplakia jamy ustnej – schorzenie
o charakterze potencjalnie złośliwym
Natalhia Gabriela de Barros Vieira Guilgen 1, Sylvia Kang1, Maria Helena Martins Tommasi1, Iran Vieira1,
Maria Ângela Naval Machado1, Antonio Adilson Soares de Lima1
1
Department of Stomatology, School of Dentistry, Universidade Federal do Paraná – UFPR, Curitiba/PR, Brazil.
Article history: Received: 30.10.2014 Accepted: 18.11.2014 Published: 28.02.2015
ABSTRACTErythroleukoplakia or speckled leukoplakia is a painless lesion characterized by an association of white areas interspersed 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, alcoholism, 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 collected by the excisional biopsy. Otolaryngologists and dentists performing an oral examination should be alert to the
patient’s habit of smoking and drinking. Oral biopsy is mandatory to recognize the presence and the severity of epithelial dysplasia, which is a decisive factor for subsequent treatment planning.
KEYWORDS: Erythroleukoplakia, Leukoplakia, Smoking, Oral Mucosa.
STRESZCZENIEErythroleukoplakia or speckled leukoplakia is a painless lesion characterized by an association of white areas interspersed 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, alcoholism, 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 collected by the excisional biopsy. Otolaryngologists and dentists performing an oral examination should be alert to the
patient’s habit of smoking and drinking. Oral biopsy is mandatory to recognize the presence and the severity of epithelial dysplasia, which is a decisive factor for subsequent treatment planning.
SŁOWA KLUCZOWE: Erytroleukoplakia, leukoplakia, nikotynizm, błona śluzowa jamy ustnej.
INTRODUCTION
Leukoplakia (leukos meaning white; plakia meaning patch) is a
disease characterized by the presence of a white plate which constitutes an expression of alteration in the keratinization process
[1]. In 1877, Schwimmer was the first author that used this term
to describe a white plaque on the tongue [2]. However, it can also
20
DOI: 10.5604/20845308.1132406
develop in the oral mucosa, penis or vulva. According to the World
Health Organization, leukoplakia is described as a white lesion
that cannot be removed by scraping. It is caused by local irritating
factors (smoking and alcohol) which stimulate the formation of
hyperkeratosis. The incisional biopsy is essential for diagnosis [1].
The natural history of oral leukoplakia remains poorly understood [4]. Oral leukoplakia can develop into a carcinoma. Malignant
WWW.OTORHINOLARYNGOLOGYPL.COM
kazuistyka / case report
transformation rates of oral leukoplakia range from 0.13 to 17.5%.
The risk factors of malignant transformation in the buccal mucosa and labial commissure are male gender with chewing tobacco
or smoking in some countries such as India, or older age and/or
being a non-smoking female in other countries [5].
According to the clinical appearance, leukoplakia can be classified into four types: early or thin, homogenous or thick, granular
or verruciform and speckled or erythroleukoplakia. Each subdivision has a different malignant transformation potential. For
example, thin leukoplakia often becomes malignant without clinical changes. Thick leukoplakia undergoes malignant transformation in 1–7% of cases [6].
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 demonstrated that 19.9% of leukoplakia cases had some degrees of dysplasia,
3.1% were frank carcinoma, 4.6% showed severe dysplasia or carcinoma in situ, and 12.2% showed mild to moderate dysplasia [7].
Erythroplakia is another oral lesion considered as a potentially
malignant disorder which precedes the development of cancer.
Erythroplakia is a red lesion characterized by patches, plaques or
erosions of various dimensions that affect regions of the hard and
soft palate, floor of the mouth, tongue and retromolar region. It
can also be caused by local irritating factors, such as tobacco and
alcohol [8]. Erythroplakia, when associated with leukoplakia, has
a higher malignant potential and shall be called erythroleukoplakia [3]. In order to establish the diagnosis, it is also necessary to
perform a biopsy and this should be done in the perilesional region. The histopathological features of erythroleukoplakia include
epithelial atrophy with varying degrees of atypia [9].
The treatment of choice for oral leukoplakia and erythroplakia is
surgery. There exists a possibility of recurrence so monitoring of
the patient should be permanent. The first step in the treatment
of oral leukoplakia and eritroplasias is advising the patient to stop
his/her habits (alcohol and tobacco). The treatment of these lesions
may be conservative or surgical [10]. Oral precancerous lesions
can be eradicated by surgical excision [11], laser surgery [12], or
photodynamic therapy [13]. The aim of this paper was to report
on a case of erythroleukoplakia that developed bilaterally in the
buccal mucosa of an alcoholic and smoker.
CASE REPORT
Male, 37 years old, presented to the clinic of the School of Dentistry (Universidade Federal do Paraná, Curitiba, Brazil) complaining of pain in the posterior teeth and difficulty in chewing.
POLSKI PRZEGLĄD OTORYNOLARYNGOLOGICZNY, TOM 4, NR 1 (2015), s. 20-24
Figure 1. Erythroleukoplakia - White plaque with red areas in the buccal mucosa
During the anamnesis, the patient reported tenderness in the posterior teeth and a temporo-mandibular joint pain. Physical examination 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
smoker’s melanosis were observed during oral examination. In
addition, white lesions interspersed with the reddish ones were
located bilaterally in the retrocomissural region. The lesions had
a triangular shape and were painless. Clinically, the lesion on the
right side was more apparent during the examination (Figure 1).
Clinically, the lesion was a plate with approximate size of 20 mm,
and white color interspersed with red areas. It had a slightly rough consistency with sessile insertion and absence of symptoms.
The diagnosis of chronic hyperplasic candidiasis was considered initially because of the clinical appearance of the lesions, and
smoking and drinking habits. The patient was not aware of the
existence of lesions. Therefore, it was not possible to establish the
time of lesion development. During medical interview, the patient
reported that he had drunk alcohol every day. In addition, he had
smoked on average two packs of cigarettes a day for over 20 years.
Before dental treatment, the patient underwent an incisional biopsy of the lesion. However, it was decided to completely remove the
lesions because of their location and size. The diagnostic hypotheses were chronic hyperplasic candidiasis or erythroleukoplakia.
The patient was medicated with 200 mg of Ibuprofen in a therapeutic regimen of doses administered every 12 hours for 3 days.
Seven days later, he returned for suture removal. No change was
observed during the repair process.
The lesions were sent for histopathological examination. The material was processed in the laboratory and stained by two stains:
21
kazuistyka / case report
hematoxylin and eosin and Grocott. The hematoxylin and eosin
revealed a fragment of oral mucosa lined by parakeratinized epithelial tissue with areas of hyperplasia, acanthosis, and exocytosis.
Some cells of the basal layer exhibited enlarged nuclei with condensed chromatin, nuclear atypia and mitotic figures (Figures 2
and 3). The underlying connective tissue showed chronic inflammatory infiltrate with a predominance of lymphocytes. No fungal
structures were identified with Grocott staining (Figure 4). That
finding excluded the possibility of fungal infection of the lesion.
According to those histological findings, the diagnosis of mild
epithelial dysplasia was established.
Figure 2. Epithelial tissue with hyperplasia, acanthosis, and exocytosis (HE x100)
The patient was advised to stop or reduce the consumption of cigarettes and alcohol because of the diagnosis of epithelial dysplasia and nicotine stomatitis. Periodic examinations were planned
but the patient did not return for the follow-up.
DISCUSSION
Oral leukoplakia is defined as a white plate which cannot be clinically or histologically characterized as any other disease [3]. Thus,
leukoplakia is essentially a clinical entity. Its diagnosis is based on
exclusion of other injuries, because this entity has no specific histopathological 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
the tongue are the most affected areas. Erythroplakia is defined
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].
Figure 3. Nuclear changes and cromatin condensation in epithelial cells of the
basal stratum (HE x400).
When the lesion presents with red and white mucosal alterations
concomitantly, the term erythroleukoplakia is used. However, in
erythroleukoplakia lesions, the red or erythroplakia areas have
been shown to be most likely to demonstrate dysplastic changes
compared to the white hyperkeratotic areas [16].
It is believed that the etiology of this lesion is related to habits such
as smoking and when associated with alcohol consumption, the
probability of malignant transformation is increased. They are
generally considered idiopathic lesions and without apparent origin. The erythroleukoplakias are more prevalent in men, due to
an increased association with smoking [17].
Figure 4. No fungal structure in epithelial tissue (Grocott x100).
22
A study by Feller et al. [18] revealed that the prevalence of erythroleukoplakias is approximately 11.2%. The same study observed
that it mainly affects males and individuals in their seventh decade of life. In our case report, the patient was male, 37 years of age,
smoked cigarettes (40 cigarettes a day) and drank alcohol (daily).
The anatomical regions most affected by potentially malignant
WWW.OTORHINOLARYNGOLOGYPL.COM
kazuistyka / case report
disorders are: buccal mucosa (28.8%), floor of the mouth (18.3%),
alveolar ridge and gums (17.3%), and tongue (12.0%). The patient
reported here had a lesion within the buccal mucosa.
The initial diagnosis of this case was chronic hyperplasic candidiasis due to the clinical aspect of the lesion and patient’s habits of
cigarette smoking and alcohol drinking. Some white lesions need
to be considered in the differential diagnosis of oral potentially
malignant disorders with white coloration, i.e.: pseudomembranous and chronic hyperplasic candidiasis, linea alba, lichen planus,
leukoedema, reactive hyperkeratosis due to oral mordiscamento
and spongy white nevus [8]. Initially, pseudomembranous candidiasis was excluded because it was not possible to remove the
lesion with scraping. In addition, the lesions of the patient had no
clinical features consistent with the diagnosis of other possibilities mentioned above. The only exception was reaction hyperkeratosis due to cheek bite. However, the patient reported no parafunctional habit, nor was there any evidence found during the
clinical examination. Thus, one was not able to rule out the possibility of a bite injury in the same region during sleep, since the
patient complained of pain in the TMJ region.
As chronic hyperplastic candidiasis was the first diagnostic hypothesis, we opted to perform an incisional biopsy. However,
during surgery it was decided to completely remove the lesions
due to their characteristics (small size and injury without a malignancy). The histological analysis confirmed that the lesion had
epithelial dysplasia.
The histopathological findings that characterize leukoplakia are
hyperkeratosis, dysplasia, chronic inflammatory infiltrate, acanthosis, and atrophy. According to the classification of dysplasia
(mild, moderate and severe), two lesions with severe dysplasia are
already considered carcinoma in situ [8]. However, histopathological examination revealed that this lesion presented hyperparakeratosis, acanthosis, atrophy, mild dysplasia, and chronic inflammatory infiltrate. In addition, staining by Grocott ruled out
the possibility of chronic hyperplastic candidiasis.
Erythroleukoplakia can be treated by surgical removal at the time
of the biopsy, or surgical laser, photodynamic therapy, and chemopreventive agents [11,12,13,19]. Thus, it is necessary that the diagnosis is made in advance and safely. In cases where moderate to
severe dysplasia is evident, complete surgical removal and periodic
monitoring are recommended due to high rates of recurrence [8].
The importance of early diagnosis of erythroleukoplakia follows
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 histological examination is critical for accurate diagnosis, and the
results from these assessments are used to determine specific treatment [20]. Thus, dentists and otolaryngologists need to be alert
during the examination of the mouth to the habit of smoking and
drinking of the patient. Oral biopsy is mandatory to recognize the
presence and the severity of epithelial dysplasia, which is a decisive factor for subsequent treatment planning.
References
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 Pathol. 1978; 46(4):518-39.
2.
Schwimmer E. Die idiopathischen Schleimhautplaques der Mundhöhle (Leukoplakia buccalis) Arch Dermat Syph. 1877;9:570–611.
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.
4.
Napier SS, Speight PM. Natural history of potentially malignant oral lesions and conditions: an overview of the literature. J Oral Pathol Med. 2008;37:1–10.
5.
Amagasa T, Yamashiro M, Uzawa N. Oral premalignant lesions: from a clinical perspective. Int J Clin Oncol. 2011; 16(1):5-14.
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; 36:1386–92.
8.
Neville BW, Day TA. Oral cancer and precancerous lesions. CA Cancer J Clin. 2002; 52(4):195-215.
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.
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, Hjørting-Hansen E, Pindborg JJ. Surgical treatment of premalignant lesions of the oral mucosa. Int J Oral Maxillofac Surg. 1987;
16(6):656-64.
12. Pinheiro AL, Frame JW. Surgical management of premalignant lesions of the oral cavity with the CO2 laser. Braz Dent J. 1996; 7:103–8.
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. Góes C, Weyll B, Sarmento VA, Ramalho LMP. Diagnóstico diferencial e manejo da leucoplasia bucal – Caso clínico: acompanhamento de 4 anos. RGO.
POLSKI PRZEGLĄD OTORYNOLARYNGOLOGICZNY, TOM 4, NR 1 (2015), s. 20-24
23
kazuistyka / case report
2007; 55(1):95-100.
15. Shafer WG, Waldron CA. Erythroplakia of the oral cavity. Cancer 1975;36:1021–8.
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; 34:133–9.
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.
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. Chemoprevention of oral leukoplakia with vitamin A and beta carotene: an assessment. Oral Oncol. 1997; 33(4):231-6.
20. Jaber MA, Porter SR, Gilthorpe MS, Bedi R, Scully C. Risk factors for oral dysplasia—the role of smoking and alcohol. Eur J Cancer B Oral Oncol. 1999; 35:151–6.
Word count: 1779 Tables: – Figures: 4 References: 20
Access the article online: DOI: 10.5604/20845308.1132406 Full-text PDF: www.otorhinolaryngologypl.com/fulltxt.php?ICID=1132406
Corresponding author: Antonio Adilson Soares de Lima, Department of Stomatology, School of Dentistry - Universidade Federal do Paraná
(UFPR), Rua Prefeito Lothário Meissner 632, Jardim Botânico, 80170-210 Curitiba - PR Brazil, Telephone: + 55 41 33604050, e-mail: [email protected]
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 Competing interests: The authors declare that they have no competing interests.
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
potentially malignant disorder. Pol Otorhino Rev 2015; 4(1): 20-24
24
WWW.OTORHINOLARYNGOLOGYPL.COM