ERYTHROLEUKOPLAKIA CASE REPORT

Introduction: It is a non-homogenous form of leukoplakia in which part of lesion is white & the rest appears reddened. It is a highly dysplastic lesion which has a high potential to turn malignant. We detected such a case in our dental OPD. CASE HISTORY: I. Patient Particulars • • • • • • Name Age Sex C.R. No. Residence Occupation : : : : : : Bhupinder Singh 50 years Male 819512 Indra Colony, Manimajra, Labourer

II Chief Complaint • • Patient wants to get missing teeth replaced. Discolouration of the left cheek.

III History of present illness • • • • Patient lost his teeth in a motor accident a year ago. Discolouration was first noticede on left cheek 6 months ago. Patient has no discomfort or pain. Patient has been smoking one pack of bidis per day for the past 10 years (10 pack years). Before that he used to smoke rarely. • • Patient also take alchohol thrice a week about 2-3 pegs in one sitting. Patient says that he takes tobacco in the chewable form as gutka which he places in his left vestibule and is right handed. III Past dental history • He had suffered from a para-symphysial fracture on his mandible in a motor accident a year ago. • • He was first operated by a plastic surgery which had failed. He was then referred to the department of oral and maxillofacial surgery at PGI where wiring was done on his mandible. • He had also lost his teeth in the same accident.

IV Past medical history • • • No history of any systemic disease. Patient says he has no allergy to any medicine. Patient says that he has not been taking a balanced diet since the accident.

V Family history None as elicited by the patient. EXAMINATION 1. Extra oral examination • • • • Facial symmetry - normal Presence of any lump or swelling - negative. TMJ - slight clicking on right side. Lymph nodes - not palpable.

2. Intra – Oral Examination a) Soft tissues  Lips - dark in colour  Tongue – normal papillary structure  Buccal mucosa – A lesion on the left cheek 4.5 cm in size with red and white patches which can not be scraped off. No ulceration, induration, nodularity present. o Present in anterior part of cheek.  Floor of mouth – normal  Palate – normal  Vestibule – deep lower labial vestibule b) Hard tissues  Generalised staining & calculus  Poor oral hygiene  Missing teeth – 36, 37, 38  Carious – 26

TREATMENT PLAN 1. Patient education : Advise the patient to completely stop the use of tobacco products & alchohol. 2. Surgical Excision.

ERYTHROLEUKOPLAKIA / SPECKLED LEUKOPLAKIA / SPECKLED ERTHROPLAKIA (new term). “Oral leukoplakia is a predominantly white lesion of the oral mucosa that cannot be characterized as any other definable lesion, some oral leukoplakia will transform into cancer.” – (Axell T, 1996). Erythroleukoplakia is a form of leikoplakia in which part of the lesion is white and rest appears reddened. DEFINITION: Term ‘erythroplakia’ is used analogously to leukoplaia to designate lesions of the oral mucosa that present as bright red velvety plaques which cannot be characterized clinically or pathologically as due to any other condition.

EPIDEMIOLOGY:

The prevelance of leukoplakic & erythroplakic lesions in India is seen almost exclusively among tobacco users. The annual age adjusted rate was 2.1 per thousand among men and 1.3 per thousand among women. The highest incident (6 per thousand) was among men who both smoked & chewed tobacco. The prevelance of this lesion is ernakulam district (Kerala, India) was 17 per 1000 and was highest (61 per thousand) among people with mixed habits. In an adult Swedish population a 3.6% prevelance rate was recorded. A definite dose – response relationship was noticed between leukoplakia & various forms of tobacco used. Dose – response relationship was stronger for smoking habit than for chewing habit that remained significant after taking account of age, gender & type of tobacco habit.

CLINICAL CLASSIFICATION: It is desirable to record separately the various forms of leukoplakia and for this purpose subdivisions are recommended (WHO, 1980). Forms of leukoplakia: a) Homogenous - lesion are uniformly white. b) Non homogenous – lesions in which part of the lesion is white and rest appears reddened. A more elaborate subdivision may be used such as: 1. Homogenous : a) Smooth b) Furrowed (fissured) c) Ulcerated 2. Nonhomogenous nodulospeckled : Well demarcated raised white areas, interspersed with reddened areas. When recording leukplakia, space has been allowed in the recording form for 3 different subdivisions. 1. Homogenous – smooth & fissured 2. Homogenous – ulcerated 3. Nonhomogenous – nodulospeckled
Adapted from community dentistry and oral epidemiology: 1980, 8: 1-26

A modified classification and staging system for oral leukoplakia : A proposal for modified classification and staging system for oral leukoplakia (OLEP) has been presented by Vander Wall, et al, 2000 in which the size of the leukoplaia and the presence or absence of epithelial dysplasia are taken into account. L1 – size of leukplakia < 2cm L2 – size of leukplakia 2-4cm L3 – size of leukplakia > 4cm Lx – size not specified P – Pathology P0 – no epithelial dysplasia P1 – distinct epithelial dysplasia Px – dysplasia not specified in the pathology report.

OLEP staging system Stage I – L1P0 Stage II – L2P0 Stage III – L3P0 & L1 L2 P1 Stage IV – L3P1 It has yet to be shown that whether such a staging system may also be helpful in providing guidelines for the management of oral leukoplakias. ETIOPATHOGENESIS

Etiology: 1. Leukoplakia occurs more frequently in smokers of tobacco than in nonsmokers. i. There is a dose response relationship between tobacco use and the prevalence of oral leukoplakia. ii. Reducing or cessation of tobacco use may result in the regression or disappearance of oral leukoplakia. iii. On the other hand disappearance of oral leukoplakia has occasionally been reported in patients who continue to smoke (Silverman and Rozen, 1968). iv. Tobacco was most often chewed as an ingredient in betwl quid (smokeless tobacco or paan) in India. 2. Whether the use of alchohol by itself is an independent etiological factor in the development of oral leukoplaia, is still questionable. Its effect, at best, may be synergistic to other well-known etiological factors (physical irritants). 3. Candida albicans: The role of Candida as a possible etiological factor in leukoplakia and its possible role in malignant transformation is still unclear. i. About 10% of oral leukoplakias satisfy the clinical and histological criteria for chronic hyperplastic candidiasis. ii. Epithelial dysplasia is reported to occur four to five times more frequently in Candida leukoplakia than in general. However, this change is more common in the speckled variant than in homogeneous leukoplakia and carcinomatous change is more a characteristic of the speckled lesion.

iii. Various kinds of evidence have been presented to justify an etiologic role for candida in neoplastic transformation, which includes, among others, the catalytic transformation in vitro of the carcinogenic nitrosamine, N-nitrosobenzyl-methylamine, by strains of C. albicans demonstrated to be selectively associated with leukoplakia. 4. Viral agents : The possible contribution of viral agents (human papilloma virus strains 16, 18) in the pathogenesis of oral precancerous lesions has been discussed. 5. Dietary Factors: In a study from India, serum levels of vitamin A, B12, C, betacarotene and folic acid were significantly decreased in patients with oral leukoplakia compared to controls, whereas, serum vitamin E was not (Ramaswamy G,et al, 1996). CLINICAL FEATURES : The term erythroplasia was originally used by Queyrat to describe aprecancerous redlesion that develops on the penis. Oral erythroplakia is clinically and histopathologically similar to the genital process. Whereas red lesions of the oral mucosa have been noted for many years, the use of the term erythoplakia in this context was rare. This blatant underreporting probably reflects the fact that leukoplakias are more likely to be biopsied and emphasizes the lack of appreciation of the significance of erythrplakia clinically. • • Many of these lesions are irregular in outline. Some contain islands or normal mucosa within areas of erythoplakia. This has been attributed to coalescence of a number of precancerious foci. • The high rate of premalignant and malignant changes noticed of erythroplakia is true for all clinical varieties of this lesion and not solely a feature of speckled erythroplakia.

Different studies have demonstrated that 80-90 per cent of erythroplakias are histopathologically either severe epithelial dysplasia, carcinoma-in-situ, or invasive carcinoma.

Erythroplakia has no apparent sex predilection most cases reported have occurred in the sixth and seventh decades.

HISTOPATHOLOGIC FEATURES : • The epithelium shows a lack of keratin production and is often atrophic, but it may be hyperplastic. • This lack of keratinization, especially when combined with epithelial thinness, allows the underlying microvasculature to show through, thereby causing the red color. • • The underlying connective tissue often demonstrates chronic inflammation. Differentiation of erythroplakia with malignant change and other early squamous cell carcinomas from benign inflammatory lesions of the oral mucosa can be ehanced by the use of 1 per cent toluidine blue solution applied topically with a swab or as an oral rinse. This technique gives excellent results in detecting epithelial dysplasia with false-negative and false positive rates of well below 10 per cent.

Clinical presentation of Bhupinder Singh with speckled erythroplakia on the left cheek.

Mild dysplasia

Severe Dysplasia

DIFFERENTIAL DIAGNOSES There are many oral lesions that appear as red ones • • • Dermatoses Inflammatory conditions due to local infection More general subacute or chronic stomatitis associated with the presence of dentures. • • Tuberculosis Fungal infections

DIAGNOSES 1. Elimination of Possible Cause(s) : The clinician should first try to rule out any of the definable white lesions before accepting a definitive clinical diagnosis of leukoplakia. 2. • Biopsy : In nonhomogenous leukoplakia, biopsy should be taken at the site of symptoms, if present, and/or at a site of redness or induration. • If treatment consists of CO2-laser evaporatin, it is mandatory to have a biopsy taken prior to such treatment. 3. Diagnostic methods other than histological examination, such as the use of toluidine blue staining or Lugol’s iodine and exfoliative cytology are of limited value when dealing with leukoplakia.

TREATMENT MODALITIES 1. Surgical excision is the most common. 2. Other modalities available are : • • • • Cryosurgery CO2-laser surgery Retinoids and other drugs Photodynamic therapy is the most recent one

PROGNOSIS : Early detection and treatment gives the most favourable prognosis. Surgical excision gives excellent results and a recurrences rate of less than 5 per cent is reported. Sometimes cessation of the predisposing factors leads to regression.

SUMMARY : This overview demonstrates that tobacco smoking and betel-quid chewing are detrimental to oral health, as they are strongly associated with oral leukoplakia, oral cancer and other mucosal pathologies. In view of these findings, specific studies for primary and secondary prevention of these lesions are warranted. Prevention was found to be feasible and effective (Gupta et al., 1980) by timely diagnosis and management of oral precancerous lesions like leukoplakia and by measures like habit alleviation. Effort in this direction will take us a long way in to the overall control and management of potentially malignant oral mucosal lesions.

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