American Journal of Pharmacology and Pharmacotherapeutics

Case Report Oral Leukoerythroplakia- A Case Report Dr. Sheeba Ali *, Dr. Puja Bansal and Dr. Deepak Bhargava

Department of Oral & Microbiology, School of Dental Sciences, Sharda University, Greater Noida, U.P., India

*Corresponding author e-mail: [email protected]

A B S T R A C T Objective: The objective of this study was to report a case of oral leukoerythroplakia, which is a potentially malignant disorder and has a high malignant transformation rate. Method: A 58 year old male patient reported with the chief complaint of burning sensation on his right inner cheek region. On clinical examination he was diagnosed as a case of oral leukoerythroplakia and excisional was performed. Results: Excisional biopsy revealed a highly dysplastic atrophic parakeratinized with dense inflammatory infiltrate, confirming the clinical diagnosis of oral leukoerythroplakia. Conclusion: All mixed red should be examined carefully since many of these could turn out to be oral leukoerythroplakia.

Keywords: , Leukoerythroplakia, Speckled, Pre-malignant .

INTRODUCTION The term oral erythroplakia is used abusers, 80% of these red patches may to describe a red plaque or macular in already contain focal areas of microinvasive the for which a specific clinical cancer at the time of initial biopsy. Its usual diagnosis cannot be established. Lesions are microscopic counterpart, in situ, named erythroleukoplakia, leukoery- has been shown to recur and transform into throplakia or speckled when invasive carcinoma in approximately 25% of white patches are present over the red treated cases. 3 The objective of this study plaque. 1 Erythroplakia is a clinical term; it was to report a case of oral does not indicate a particular microscopic leukoerythroplakia, which is a potentially diagnosis, although after a biopsy most are malignant disorder and has a high malignant found to be severe or carcinoma. 2 transformation rate. In very high risk cases, such as oral floor lesions in heavy smokers and alcohol

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CASE REPORT which describes it as, ‘‘any lesion of the that presents as bright red velvety A 58 year old male patient reported plaques which cannot be characterized to our department with the chief complaint clinically or pathologically as any other of burning sensation on his right inner cheek recognizable condition’’. 5 An updated region. The patient had been aware of a red definition for erythroplakia was proposed by patch on his right inner cheek for about 2 Bouquot as “a chronic red mucosal macule years; however, he had not sought any which cannot be given another specific treatment as the lesion was not painful. diagnostic name and cannot be attributed to Medical and dental histories were non- traumatic, vascular, or inflammatory contributory. Patient had the habit of causes”. Erythroplakia patches may be smoking of bidi approx 25bidis/day and located near, or associated with, oral occasional alcohol consumption. Intraoral leukoplakias. Bouquot and Whitaker examination showed 2cm X 1.5cm bright suggested that erythroplakia may occur with red patch with white specks on its surface on leukoplakia in the stage called the right buccal mucosa (Fig 1). The lesion erythroleukoplakia. 6 was sharply demarcated from the The prevalence rate of these lesions surrounding normal mucosa. No has been reported between 0.01%- 0.21%. surrounding induration was present. Clinical The incidence is not known, but the average diagnosis of leukoerythroplakia was made. annual incidence for microscopically proven Complete surgical excision of the lesion was oral , which represents the advised (Fig 2). Histopathological great majority of erythroplakias, has been examination of the biopsy showed highly estimated to be 1.2 per 100,000 population dysplastic, atrophic parakeratinized stratified (2.0 in males and 0.5 in females) in the squamous epithelium with dysplastic United States. 7 features like nuclear hyperchromatism, It predominately occurs in the floor increased nuclear cytoplasmic ratio, nuclear of the mouth, buccal mucosa, soft , and cellular pleomorphism and few mitotic ventral and tonsillar fauces. 8 In a figures. Underlying connective tissue stroma study on 58 cases of erythroplakia, the showed dense inflammatory cell infiltrate, disease was found to be more common chiefly lymphocyte. Based on histo- among people in their 50s and 60s. The risk pathological features, a diagnosis of severe factors for such as chewing dysplasia was made (Fig 3). tobacco, smoking, and alcohol drinking are assumed to be associated with erythroplakia. DISCUSSION In a recent case-series study, erythroplakia was associated with a high prevalence In 1911, Queyrat described a sharply of TP53 mutations. TP53 mutations may be defined, bright red, glistening velvety associated with tobacco exposure for oral precancerous lesion of the glans penis, cancer, which would possibly indicate that which was termed ‘erythroplasie’. Although tobacco exposure may play an important red lesions of the oral mucosa have been role in the development of erythroplakia. 6 noted for many years, the use of the term The differential diagnosis includes: “erythroplakia” in this context has been 4 erythematous candidiasis, early SCC, local common for only about 25 years. irritation, mucositis, , lupus Over the years several definitions for erythematosous, drug reaction and median oral erythroplakia have been suggested. rhomboid . 8 Most accepted is the one given by WHO,

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Because localized areas of redness situ lesions because of their superficial are not uncommon in the oral cavity, areas nature and the fact that dysplastic cells of erythroplakia are likely to be disregarded usually extend beyond the clinically evident by the examiner, and they are often falsely lesion. However, since recurrence and determined to be a transient inflammatory multifocal involvement is common, long- response to local irritation. Differentiation of term follow-up is mandatory. 2,9 erythroplakia from benign inflammatory lesions of the oral mucosa can be enhanced CONCLUSION by the use of a 1% solution of toluidine blue, applied topically with a swab or as an oral Erythroplakia has been called "the rinse. 9 dangerous oral mucosa" because it typically Histopathologically, epithelium presents as carcinoma in situ, severe shows lack of keratin production and is or superficially invasive often atrophic, but it may be hyperplastic. carcinoma under the microscope. There is This lack of keratinization and epithelial currently no unique reliable parameter to thinness allows the underlying identify these lesions predictive of microvasculature to show through, thereby malignant transformation. Risk assessment causing the red color. 4,7 Epithelium shows is usually based on clinical, pathological and dysplastic features like hyperchromatism, more recently on bio-molecular evaluations. pleomorphism and increase in number of Few data are available on oral erythroplakia mitotic figures. 6 In a sister study, to their and there is an urgent need for randomized large series of leukoplakia cases, Shafer and controlled trials. Waldron also analyzed their biopsy experience with 65 cases of REFERENCES erythroplakia. All the erythroplakia cases 1. Hosni ES, Salum FG, Cherubini K, Yurgel showed some degree of epithelial dysplasia; LS, Figueiredo MAZ. Oral erythroplakia 51% showed invasive squamous cell and speckled leukoplakia: retrospective carcinoma, 40% were carcinoma in situ or analysis of 13 cases. Brazilian Journal of severe epithelial dysplasia, and the Otorhinolaryngology . 2009;75(2):295-9. remaining 9% demonstrated mild-to- 2. Regezi JA, Sciubba JJ, Jordan RCK. Oral moderate dysplasia. Therefore, true clinical Pathology: Clinical Pathologic Correlations. th erythroplakia is a much more worrisome 4 edition. Red-Blue lesion. Saunders An lesion than leukoplakia. 10 imprint of Elsevier Science . USA. 2003. p- Erythroplakia has been considered 118. the most severe form among all of the oral 3. Bouquot JE, Paul M. Speight PM, Paula M. Farthing PM. Epithelial dysplasia of the oral premalignant lesions because of its high 6 mucosa—Diagnostic problems and malignant potential. Generally, prognostic features. Current Diagnostic transformation rates, including those with Pathology. 2006;12:11–21. invasive carcinoma already at biopsy, vary 4. Sahfer, Hine, Levy. Benign and malignant from 14% to 50%. 5 Table 1 shows the tumors of the oral cavity. Textbook of Oral malignant transformation of various Pathology. 7 th ed. Elsevier India Private premalignant lesions. 3 Limited. Gurgaon. 2012.p.94. The treatment of choice for 5. Reichart PA, Philipsen HP. Oral erythroplakia is surgical excision. It is erythroplakia—a review . Oral Oncology . generally more important to excise widely 2005;41:551–561. 6. Hashibe M, Mathew B, Kuruvilla B, than to excise deeply in dysplastic and in Thomas G, Sankaranarayanan R, Parkin

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DM, et al. Chewing Tobacco, Alcohol, and implication for clinicians. Australian Dental the Risk of Erythroplakia. Cancer Epidemiol Journal. 2011;56(3):253-256. Biomarkers Prev. 2000;9:639. 9. Greenberg MS, Glick M. Burket’s Oral 7. Neville BW, Damm DD, Allen CM, Medicine. Diagnosis and Treatment. 10 th ed. Bouquot. Epithelial Pathology. Oral and Ch-5. Red and White Lesions of the Oral Maxillofacial Pathology. 3rd ed. Reed Mucosa. BC Decker Inc. Hamilton,Ontario Elsevier India Private Limited. Noida. 2009. 2003. p-85. p.345. 10. Nair DR, Pruthy R, Pawar U, Chaturvedi P. 8. Villa A, Villa C, Abati S. Oral cancer and Oral cancer: Premalignant conditions and oral erythroplakia: an update and screening - an update. J Can Res Ther. 2012;8:57-66..

Table 1: Potentially malignant lesions of the oral, pharyngeal and laryngeal mucosa, clinical terms only. 3

Disease name Malignant transformation potential

Proliferative verrucous leukoplakia ****** Erythroplakia ****** Nicotine palatinus in reverse smokers ***** **** Speckled, granular (non-homogeneous) leukoplakia **** Laryngeal keratosis/ leukoplakia *** Actinic *** Smooth, thick (homogeneous) leukoplakia ** Smokeless tobacco keratosis * Lichen planus * (*) indicates the severity of malignant transformation.

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Figure No. 1: Photograph showing a red patch with white nodules on the buccal mucosa.

Figure No. 2: Complete excised lesion .

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Figure No. 3: Photomicrograph showing atrophic parakeratized stratified squamous epithelium with severe dysplastic features. Dense inflammatory cell infiltrate is also seen. (H&E 10X)

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