Abstract
Lichen Planus is a muco-cutaneous disorder affecting females. The oral lesions precede the skin lesions, with malignant potential rate, ranging from 0.3 to 3%. Malig-nant changes are usually associated with stress and co-carcinogens. Usually, malignant transformation is presented as solitary lesion in the oral cavity, Oral Squa-mous Cell Carcinoma (OSCC), developing at multiple sites, as a very rare finding in patients of Oral Lichen Planus (OLP). A case of OLP without risk factors and strong history of stress, the Grinspan syndrome with field cancerization is presented in this article.
Keywords: lichen planus, field cancerization, emotional stress, carcinogens, Grinspan’s syndrome.
INTRODUCTION
Oral lichen planus (OLP), a chronic inflamma-tory dermatosis of unknown etiology often involving the mucous membranes, is a disease that causes bilateral white striations, papules or plaques on the buccal mucosa, tongue, and gin-giva. [1] Erythema, erosions, and blisters may or
may not be present. Lichen planus is a disorder affecting about 1-2% of a population. Involve-ment of mucous membranes is seen in about 15% of cases. Lesions can be limited to mucous mem-branes only. [1,2]
Literature reviews suggest a malignant trans-formation rate, ranging between 0.3-3%. There is abundant data on Lichen Planus transforming into oral squamous cell carcinoma with strong association of oral carcinogens. [3] Very few
cases, where association of co-carcinogens were absent, and showing features of field canceriza-tion, have been reported.
FIELD CANCERIZATION IN LICHEN PLANUS –
AN UNUSUAL CASE REPORT
Tamgadge Avinash1, Sourab Kumar2, Tamgadge Sandhya3, Sudhir Bhalerao4,
Treville Periera5, Gokul Venkateshwar6
1. Prof & HOD, Dept of Oral & Maxillofacial Pathology and Microbiology, Maharashtra, India 2. PG Student, Dept of Oral & Maxillofacial Pathology and Microbiology, Maharashtra, India 3. Prof & PG Guide, Dept of Oral & Maxillofacial Pathology and Microbiology, Maharashtra, India 4. Prof & PG Guide, Dept of Oral & Maxillofacial Pathology and Microbiology, Maharashtra, India 5. Prof & PG Guide, Dept of Oral & Maxillofacial Pathology and Microbiology, Maharashtra, India 6. Prof & PG Guide, Dept of Oral & Maxillofacial Surgery, Maharashtra, India
Contact person: Sourab Kumar, e-mail: sourab.birla@gmail.com
This article presents a unique case-report of lichen planus undergoing SCC at multiple sites, with strong history of emotional stress and Grinspan’s syndrome with no risk factors.
CASE-REPORT
A 61-year-old widowed woman addressed the Department of Oral and Maxillofacial Pathol-ogy, complaining of white erythematous areas on both buccal mucosa and tongue, with burning sensation for the past six months. (Figs. 1, 2) The patient had a history of diabetes and hyperten-sion of long-standing duration for which she was on medication. Dental history was unremarkable, with extractions of few teeth. General examina-tion showed pruritic and papular erupexamina-tions on extremities (predominantly on lower legs and feet) and back, of long duration. Scalp was not involved. Nails of hands were normal, while those of the feet were showing linear striations with slight changes in color. There was no family history of any skin disorders. The patient denied tobacco abuse or any habits.
differential diagnosis of Erosive Lichen Planus, Pemphigus, Candidiasis was given. Incisional biopsy, performed from Buccal mucosa, tongue, confirmed the diagnosis of well-differentiated SCC. (Figs. 3, 4)
Fig. 1. Erythematous lesion on the left lateral surface of tongue
Fig. 2. Erythematous granular lesions on the right buccal mucosa
Fig. 3. Keratin Pearls are evident,
along with dense inflammatory stroma suggestive of squamous cell carcinoma
Fig. 4. Extra-oral photograph after radical neck dissection
Microscopic examination showed lesional tis-sue consisting of invasive neoplastic stratified squamous epithelium arranged in sheets and islands, with displastic features in the form of hyperchromatic nuclei, altered nuclear – cyto-plasmic ratio, bizarre mitotic figures, pleomor-phism. Many keratin pearls were also seen. Muscle tissue was evident. Stroma shows dense chronic inflammatory cell infiltrate.
All these features suggested well-differenti-ated squamous cell carcinoma. The patient was referred to nearby Oncology Centre for surgery radiotherapy, and radical neck dissection was performed. During the first follow-up, the patient showed deteriorating health. (Figs. 5, 6)
DISCUSSION
It was Erasmus Wilson who coined the term “Lichen Planus” in 1869. The condition is most often seen in middle-aged patients, and affects more women than men. The malignant transfor-mation rate is lower, compared to other Pre-malignant Lesion and conditions. Oral cancer most frequently develops on LP between the sixth and seventh decade, as in our case. With dietary changes, patients with OLP consume less fresh vegetables and fruit, especially citrus fruit, which may increase cancer risk. [4] The mean
interval from the onset of the oral lesions to the development of cancer is nine to twelve years (from 3 months to 40 years). [5] 46% – 54% of the
cancers occur on the buccal mucosa, 30% on the tongue, 16% on the lower lip and 8% in miscel-laneous sites. Any location in the oral cavity may be involved, the most common site being the posterior buccal mucosa. The tongue is consid-ered the preferred site for cancer emergence of cancer. As in our present case, where both tongue and buccal mucosa were involved. An important feature of the presentation and clinical course of carcinoma that arises on OLP is the tendency of multiplicity of lesions. [3] In our case, neoplastic
events were also evident at more than two sites, as both buccal mucosa and the lateral borders of the tongue were involved. With respect to the clinical form of OLP, numerous authors found that atrophic-erosive forms, along with keratotic forms (plaques), were transformed into cancer forms. [4,6] Age, site and type of lichen planus
were in accordance with the ulceration. The etiol-ogy of Lichen Planus is most likely of multifacto-rial origin. OLP has a genetic predisposition and is initiated by a variety of factors, including emo-tional stress and hypersensitivity to drugs, den-tal materials, or spicy food – as in our case. [6]
Emotional stress has been considered a strong contributing factor by many investigators, espe-cially for the exacerbation phases of OLP, which was the strong etiological factor in the presented case, as the patient was a widow, only few years after marriage. [7] As OLP is frequently observed
in patients with cutaneous Lichen Planus, it may be the only finding in approximately 25%. Both oral and cutaneous lesions were evident in our
case. [8] The reticulate clinical presentation
dis-playing the characteristic Wickham’s Striae is most common. [9] However, in our case, it was
predominantly of erosive type, with more malig-nant potential. With respect to the clinical form of OLP, numerous authors found that atrophic-erosive forms predisposed to cancer develop-ment, as in the present case. [10-12] The
interesting finding in our case was the negative history of chronic oral exposure to carcinogens, which made the diagnosis difficult, to which positive history of emotional stress, hyperten-sion and diabetes mellitus (a diagnostic of Grin-span’s syndrome) along with cutaneous lesions were associated. Squamous cell carcinomas can arise without some previous exogenous cause.
[13,14]
CONCLUSIONS
Reports of OLP conversion to SCC have cre-ated a great deal of controversy about the true nature. We have documented the development of SCC in a lesion clinically and histologically diagnosed as OLP, without any history of tobacco, alcohol etc., and associated with strong history of emotional stress, hypertension and diabetes. This case raises many questions on the diagnostic process, illustrating the ongoing dis-cussion on the premalignant potential of OLP and emphasizing the need for a close follow-up.
References
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Table 1
