A recalcitrant case of erosive oral lichen planus

INTRODUCTION

Lichen planus is a mucocutaneous disease of inflammatory origin affecting around 1-2% of the general population. It can occur in single or multiple cutaneous and non-cutaneous (mucosal) areas within the body. Consequently the disease can affect either skin alone, the oral cavity alone, or occur in both the areas simultaneously; it may also affect other sites namely nails, scalp, oesophagus, and the genital areas.^1,2Although the aetiology is yet elusive, the various factors attributed include genetics, immunologic factors, medications, and hepatitis C infection. The literature shows six distinct types of OLP, namely reticular, atrophic, erosive, plaque-type, popular, and bullous type;(3)one or more forms can coexist in a single patient. The atrophic and erosive types are reported to carry an increased risk for malignant transformation. OLP is usually found in a bilateral fashion but need not be symmetrical. The common sites for OLP are buccal mucosa, dorsal surface of the tongue, and gingiva, at times occurring on other rare sites as well.⁴ The reticular OLP is the most common form which is asymptomatic and is usually diagnosed on routine oral examination. The erosive OLP is the most noteworthy of all the types having characteristic symptoms and consisting of the lesions in the form of a network formed by radiating keratinized striations;⁵ the other types with symptoms and difficult to treat include the atrophic and the bullous types. The erosive OLP becomes multifocal with time and patients complain of varying amounts of pain and interference with the normal mastication process. Topical or systemic corticosteroids remain the mainstay of treatment for OLP; however, it is advised to use further adjuvants to enhance the process of healing and assist in modulation of the immune process in the OLP microenvironment. We report a case of erosive OLP effectively treated with topical clobetasol propionate and systemic prednisone.

CASE REPORT

A 60-year-old male patient with no underlying systemic anamnesis approached our department of oral medicine, for a therapeutic opinion for a painful ulcer of 6 months duration on the left buccal mucosa. The patient had no history of smoking or tobacco chewing. The ulcer on the left buccal mucosa had increased in size in the last 6 months. In addition, the patient complained of pain and tenderness during phonetics and while having food. The patient was using Tess buccal paste (Triamcinolone acetonide 0.1%); advised by a local Dentist he had visited around 15 days back, but without any significant relief. On examination patient had an irregular shallow ulcer around 3cm x 2cm on the posterior region of the left buccal mucosa; the right buccal mucosa had white striations which the patient was not aware, possibly due to lack of any symptoms. (Figure 1a & 1b) The hard tissue examination revealed metal crowns on teeth numbers 36 and 46, and amalgam restoration with respect to tooth number 37. The approximation of amalgam restorations and the metal crowns prompted us for a provisional diagnosis of oral lichenoid reaction (OLL), and a differential diagnosis of OLP Incisional biopsy of the lesion on the left buccal mucosa was carried out and showed features of erosive lichen planus without any dysplastic features. Stratified squamous parakeratinised epithelium was noticed with saw tooth rete ridges in a few areas. (Figure 2a) Areas of ulceration surrounded by hyalinization and a thick band of inflammatory infiltrate can be appreciated below the dense mixed inflammatory infiltrate; chiefly neutrophils. (Figure 2b) Most of the areas have subepithelial epithelium and the deep connective tissue has muscle fibers, adipose tissue, and extravasated RBCs. The patient was advised topical clobetasol propionate 0.05% and systemic prednisone in the form of 20 mg/day oral tablets for 1week. The patient was evaluated after 1 week with marked improvement noticed on both the right and the more severe left side. (Figure 3a & 3b) The patient was advised to continue topical clobetasol propionate 0.05% and systemic prednisone was tapered for another week. The patient showed remission in lesions bilaterally in 3^rd week of treatment, and the erosive lesion on the left buccal mucosa was completely resolved. (Figure 4) The amalgam restorations and the metal crowns were replaced with ceramic restorations and the patient was kept on follow-up. The patient reported back with relapse even after the replacement of the restorations, (Figures 5a & 5b) supporting our clinicopathological diagnosis of OLP The patient was retreated with local and systemic corticosteroids and showed signs of healing during the second course of treatment. (Figure 5 c & 5d) The patient did not report for further evaluation.

Figure 1

A) Reticular lichen planus on the right buccal mucosa. B) Erosive lichen planus on the left posterior buccal mucosa.

Figure 2

A) Stratifed squamous parakeratised epithelium with saw tooth rete ridges and dense subepithelial lymphocytic infiltration. B)Ulceration surrounded by hyalinization and thick band of inflammatory infiltrate

Figure 3

A) Resolution of lesion on the right buccal mucosa at 7 days of treatment B) Marked improvement on the left buccal mucosa after 7 days of treatment

Figure 4

Complete resolution of erosive lichen planus after initial course of corticosteroids, on the left buccal mucosa after 3 weeks of treatment.

Figure 5

A,B) ​Recurrence after replacement of metallic restorations. C,D) Improvement during second course of treatment.

DISCUSSION

The etiology of lichen planus is still not clear. Much of the data yielded through years of research, attributes immunologic mechanisms as the mainstay for the pathogenesis of the lichen planus.⁶ The role of Langerhans cells, the mast cells, and their interactivity with the abundant T-cells assembled in the underlying connective tissue has been primarily focused on in the literature. The evidence gathered over the years suggests lichen planus is an autoimmune disorder mediated by T-cells in which apoptosis of basal keratinocytes occurs as a result of cluster of differentiation (CD8) + T cells. OLP is a non-infectious chronic inflammatory disorder of stratified squamous epithelium of oral mucosa and the underlying lamina propria with or without its cutaneous form. It is estimated that 30% to 50% of cases with OLP have cutaneous manifestations, and such skin lesions can aid in the diagnosis of this disorder. Lichen planus occurs on cutaneous sites only, in 35% of cases, and 25% of cases have only a mucosal presentation. OLP usually occurs in the middle-aged and elderly with a female to male ratio of 1.5: 1. OLP has significantly higher chronicity than cutaneous lichen planus. Although this inconsistency in chronicity between OLP and its cutaneous counterpart is not clear, the role of T-cell RANTES (regulated upon activation, normal T-cell expressed and secreted) and mast cell degranulation, that releases tumor necrosis factor α and a cyclical process, where interleukin 4 and interferon may forecast the chronic nature of the disease in certain cases. (Figure 6)¹

Figure 6

The disease chronicity of oral lichen planus promoted by cyclic mechanism

Clinical characteristics of OLP are dictated by the severity of the disease and present in the range of clinical appearances. The four types of OLP lesions most often described and discussed are reticular, erosive, plaque-like, and bullous. (Table 1) Topical and systemic `corticosteroids are the most widely used agents for OLP with the aim to modulate a patient’s immune response. Topical corticosteroids constitute the mainstay and depending upon the degree of severity, the agents commonly used in increasing order of their potency are triamcinolone acetonide, fluocinonide, or fluocinolone acetonide (fluorinated steroids), betamethasone phosphate, and halogenated clobetasol propionate.⁷ Topical corticosteroid resistant and recalcitrant OLP can alternatively be managed by other topical agents, like tacrolimus or cyclosporine, both calcineurin inhibitors, and sometimes by retinoids.⁸ Systemic corticosteroids have a decent role in recalcitrant, erosive, or when topical agents are ineffective. A number of studies have established systemic corticosteroids as the most effective modality of treatment for OLP⁹ As corticosteroids dosage has a wide range, and further due to variable patient responses to systemic corticosteroids, a number of dosing options exist. The oral dose of prednisone ranges from 10–20 mg/day for moderate OLP to as high as 35 mg/day for extreme cases.⁷ It is challenging to treat and diagnose symptomatic OLP in a definite manner due to the lack of a uniform method and also due to variable individual factors.¹⁰

CONCLUSION

The present case with a history of 6 months duration and ineffective results with topical triamcinolone acetonide was managed with a combination of a super potent topical agent (clobetasol propionate) and a systemic (oral) corticosteroid. The patient had a relapse during the course of the treatment. There is a need for large group studies of clinical trials to unravel potential medications for the effective treatment of OLP

DECLARATION OF PATIENT CONSENT

Duly signed informed consent was taken from the patient for publication purposes. It was conveyed to the patient that his name and initials shall be concealed, and all efforts would be taken to hide his identity although anonymity cannot be guaranteed.

CORRESPONDING AUTHOR

Dr Sajad Ahmad Buch
MDS Oral Medicine & Radiology
School of Dentistry, IMU University
no. 126, Jalan Jalil Perkasa 19
Bukit Jalil-57000, Kuala Lumpur-Malaysia
email: buchh.sajad@gmail.com

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