Oral Lichen Planus: A Report and Review of an Autoimmune-Mediated Condition in Gingiva
Gowri Pendyala, MDS; Saurabh Joshi, MDS; Jithendra Kalburge, MDS; Manjiri Joshi, MDS; and Avneesh Tejnani, MDS
Oral lichen planus (OLP) is a chronic autoimmune, mucocutaneous disease that affects the oral mucosa as well as the skin, genital mucosa, scalp, and nails. It is one of the most common dermatological diseases presenting in the oral cavity. An immune-mediated pathogenesis is recognized in lichen planus, although the exact etiology is unknown. The disease most commonly affects middle-aged females. It is infrequently found in children, with a prevalence of about 0.03%, and reports of this are scarce in the literature. The erosive and atrophic forms of OLP are less common, yet they are more likely to cause symptoms. OLP is the target of much controversy, especially in relation to its potential for malignancy. Thus, it is important for clinicians to maintain a high index of suspicion for all intraoral lichenoid lesions. Periodic follow-up of all patients with OLP is recommended. In view of the above, the authors highlight a case of gingival erosive lichen planus affecting a 17-year-old adolescent without concomitant cutaneous lesions, with special emphasis on clinical and microscopic characteristics of the condition and management with retinoids and steroid therapy.
Oral lichen planus (OLP) is a rather common disease in the middle-aged and elderly populations. It has a prevalence of about 0.5% to 2%, with a female-to-male ratio of approximately 2:1.1,2 The reported prevalence rates in the Indian population is 2.6%.3 In contrast, oral lichen planus in childhood (OLPc) is rare, and only a few reports are available in the literature.3-8
OLP is classified into reticular, erosive, atrophic, and bullous types.9 The erosive and reticular forms are the most common types and present as papules and plaques with interlacing white keratotic lines (Wickham striae) with an erythematous border.10 The higher frequency of erosive OLP compared to the reticular and atrophic forms, as previously observed by Silverman,11 is probably the result of the symptomatic nature of this lesion, which often prompts an evaluation visit. The occurrence of erosive OLP confined to the gingival mucosa is characterized by the presence of diffuse erythematous areas that may or may not be interspersed with desquamative and ulcerated foci. The lesions may occur following the gingival outline, and hyperkeratotic radiating straie can be found at the periphery of the erosive region. When erosive OLP involves the attached gingival tissue, it is called desquamative gingivitis. The lesions of erosive OLP migrate over time and tend to be multifocal.12
These variants may occur together in one patient or may transform one to another. The lesions are found (in diminishing frequency) on the buccal mucosa (often symmetrical), lateral margins of the tongue, gingiva, lips, and palatum durum.2,3,9 Whereas cutaneous lichen planus is self-limiting, OLP is chronic, rarely undergoes spontaneous remission, and may be a potential source of significant morbidity.3 Family history of lichen planus is more commonly positive in patients with lichen planus in childhood than in adulthood.9 The etiology of lichen planus remains uncertain, but many factors have been implicated. Such factors include genetic predisposition, infective agents, systemic diseases, graft-versus-host disease (GVHD), drug reactions, vitamin deficiencies, and hypersensitivity to dental materials.13 Lichen planus has been associated with several auto-immune diseases, including lupus erythematosus, pemphigus, Sjögren’s syndrome, and autoimmune liver disease.2,4 The pathogenesis of lichen planus is not completely understood, but a T lymphocyte infiltrate suggests cell-mediated immunological damage to the epithelium.14 Modified Langerhans’ cells and keratinocytes may trigger an immune response and the recruitment of T lymphocytes caused by expression of cell-surface adhesion molecules.4,13 Both CD4 (helper) and CD8 (cytotoxic) cells are present, but increasing numbers and activation of the CD8 cells are thought to contribute to the characteristic damage to the basal epithelium.13,14
It is important for all clinicians to be aware of its clinical presentations and management, because OLP is one of the most common mucosal conditions affecting the oral cavity. The most widely accepted treatment for OLP is topical corticosteroids.12 Alternative treatments include retinoids, cyclosporine, tacrolimus, surgery, and carbon dioxide (CO2) laser.12 This report highlights the use of topical and systemic corticosteroids and retinoids for the management of erosive gingival lichen planus in an adolescent; it also includes a review of current literature on this condition.
A 17-year-old male presented with a chief complaint of a burning sensation in his gums leading to difficulty in eating and swallowing for 6 months. The patient reported aggravation of the discomfort and sensitivity in the gingiva on consumption of acidic or spicy food and drinks. Local deposits were inconsistent with the corresponding lesion. No cutaneous lesions were visible.
The patient’s past medical, dental, and family histories were not significant. The patient was otherwise fit and healthy and was not taking any medication. He was a non-smoker who did not consume alcohol.
Intraoral examination revealed peeling of the underlying gingival epithelium, where red erosion of the connective tissue involving the entire maxillary and mandibular buccal and lingual/palatal surfaces was observed. The peeled epithelium could be separated from the underlying connective tissue with a periodontal probe (Figure 1 and Figure 2).
Routine hematological investigations and biochemistry yielded negative results. An incisional biopsy was performed, and a wedge of tissue including both the epithelium and connective tissue was obtained from the most prominent area, which was the lingual side of the mandibular anteriors. It is important to obtain an elliptical wedge of mucosa extending beyond the affected area to avoid stripping the superficial epithelial layer from the underlying connective tissue. However, as the lesion was generalized—involving both the arches—performing a biopsy was challenging, with the epithelium stripping away from the underlying connective tissue.
Histopathological findings revealed that the epithelium was completely separated from the underlying connective tissue. The epithelium was hyperplastic, with parakeratinized stratified squamous epithelium showing signs of hypeparakeratosis, acanthosis, and hydropic degeneration of the basal layer. A dense band-like lymphocytic infiltration was found at the epithelial-connective tissue interface, and there were areas of atrophic epithelium with saw-tooth rete ridges (Figure 3 and Figure 4).
Based on the clinical and histopathological findings, a final diagnosis of erosive lichen planus of gingiva was made.
Various treatment regimens have been designed to improve management of symptomatic OLP, but a permanent cure remains to be found. Treatment in this case was aimed primarily at reducing the length and severity of symptomatic outbreaks. The therapeutic regimen for the first 7 days included triamcinolone acetonide 0.1% (Tess Buccal Paste, Troikaa Pharmaceuticals, www.troikaa.com) for topical application. The patient was then instructed to apply a thin layer of the ointment directly on the lesion three times a day (after meals and at bedtime) and not to take anything orally for 1 hour after its application, after which he was instructed to rinse with diclofenac 0.074% mouthrinse (Disoral Mouthwash, Elder Pharmaceuticals Pvt. Ltd., www.elderindia.com) twice daily to avoid desiccation and irritation of the oral tissues. The patient was put on systemic therapy of antioxidants—capsule Lycostar (Mankind Pharma Pvt. Ltd, www.mankindpharma.com) twice daily.
The patient was advised to avoid spicy food, comply with a healthy diet rich in fresh fruit and vegetables, and visit the office regularly for ongoing preventive care. The patient was educated and motivated regarding a proper plaque control regimen, which included brushing twice daily with a soft brush and toothpaste and rinsing twice daily with alcohol-free chlorhexidine.
At the 1-week recall visit, the patient reported improvement in the burning sensation, but the erythematous areas persisted (Figure 5). Oral hygiene instructions were reinforced, and the patient was asked to continue the same medication and to return in 15 days (Figure 6).
On re-evaluation after 15 days, the erythematous desquamated area and burning sensation were reduced. Also there was no secondary growth of Candida organisms. The patient was advised to discontinue the oral rinse and taper the dose of topical steroid ointment. He was also asked to continue with Lycostar capsules for 3 months.
Lichen planus was first described in the literature by Eramus Wilson in 18692 as predominately a disease of the middle-aged or older. There is limited literature available reporting the occurrences of oral lichen planus in children.4,5,7,8,15-20 Childhood lichen planus has been documented as a complication of hepatitis B virus (HPV) vaccination, where the recombinant proteins of the HBV vaccine—specifically the viral S epitope—may trigger a cell-mediated autoimmune response targeted at keratinocytes, giving rise to a lichenoid reaction.21,22 It is also found in association with predisposing conditions such as graft-versus-host disease (GVHD) and chronic active hepatitis C. Cottoni20 reported oral lichen planus involvement in one patient affected by autoimmune chronic active hepatitis, and Agrawal22 reported oral lesions in one patient after HBV vaccination.
Familial lichen planus has been reported as being uncommon.5,18,19,23 Milligan23 reported a family history present in 1% to 2% of cases. Childhood familial lichen planus is said to occur at an early age5 and with greater severity.23 Mahood18 reported that 12% of his patients with familial lichen planus presented before the age of 10 years.
Reports in the literature describing children with lichen planus have tended to demonstrate the majority as having cutaneous manifestations and a low incidence of oral involvement. Kanwar7 and Kumar8 reported one out of 25 (4%) and one out of 17 (6%) cases, respectively, showing mucosal involvement. Recent studies have revealed a greater incidence of children developing oral lichen planus. Nanda15 reported nine out of 23 (39%) of their childhood cases presented with mucosal lesions, and Sharma16 described 15 out of 50 cases (30%) with mucosal lichen planus involvement. The most recent study of 87 cases of childhood lichen planus reported 12 of the patients (13.8%) to have oral mucosal involvement.17
It has been documented that childhood lichen planus is more common in the tropics8 and that children of Asian origin may be more prone to the condition. The lichen planus adolescent presented in this case is also of Asian origin.
Some authors20,23 have suggested that there is a difference between childhood and adult lichen planus in that the presentation is often of prolonged atypical skin lesions, which are sometimes recalcitrant to topical therapy. Also, among those cases reported, a significant proportion had a family history of lichen planus. Handa,17 in a study of 87 cases, concluded that the natural history of lichen planus in children was essentially similar to that in adults. The majority of children affected with oral lichen planus, however, have been asymptomatic or they have complained of slight dryness of the mouth along with the burning sensation, as was the case with the patient presented.
Erosive oral lichen planus tends to affect approximately 40% of adult patients4; it is rarely reported in children. Alam and Hamburger4 reported one case of erosive lichen planus from their study of six cases. Howard and Tsuchiya,24 reviewing pediatric cutaneous disorders, reported one 16-year-old with erosive oral lichen planus; this report also presented a case of gingival erosive lichen planus without any cutaneous lesions in an adolescent.
The diagnosis may be made using clinical features alone or may require clinicopathologic correlation to rule out malignant conditions or atypical presentations. The differential diagnosis depends on the age of the patient, clinical variant of OLP, and the severity and persistence of lesions.4 The clinical differential diagnoses include: lichenoid drug reaction (LR), leukoplakia, lupus erythematous, and GVHD.
Lichenoid reactions to dental amalgam and drugs25 have been reported to exhibit histopathologic features similar to OLP. A detailed history and absence of amalgam restorations excluded the possible diagnosis of lichen planus [lichenoid reactions] in the present case. The age of the patient, pathognomonic appearance of reticular OLP (Wickham striae) on the buccal mucosa, and the histopathologic findings excluded the possibility of lupus erythematous, leukoplakia, and GVHD. Various studies have reported possible association or non-association between hepatitis C virus (HCV) infection and lichen planus and have related this to variations in genetic, environmental, and geographic factors.21,26,27 However, no such association was found in the present case.
Children affected with OLP are often asymptomatic or minimally symptomatic. The aims of current oral lichen planus therapy are to eliminate mucosal erythema and ulceration, alleviate symptoms, ensure maintenance of oral hygiene, and reduce the risk of oral cancer in oral lichen planus patients. In this context, medical treatment is restricted currently to atrophic, erosive, bullous, or symptomatic oral lichen planus lesions.12 Corticosteroids remain the mainstay of oral lichen planus therapy because of their activity in dampening cell-mediated immune activity, and they can be administered topically, intralesionally, or systemically. Topical corticosteroids (in order of decreasing potency) such as 0.05% betamethasone valerate gel, 0.05% fluocinonide gel, and 0.1% triamcinolone acetonide can be used.28 In the presented case, the authors used 0.1 % triamcinolone acetonide topically, which was of lowest potency and can be used in mild to moderate cases of discomfort. This drug was available over the counter and was useful in the treatment of OLP.
Retinoids, metabolites of vitamin A known to have anti-keratinizing and immunomodulating effects, form the second line of treatment of OLP. Lower concentrations of retinoids combined with other antioxidant agents like lycopene and α-tocoferol (vitamin E) are known to have synergistic effect in the remission of the lichen planus lesions.29
Lycopene, a carotenoid, is a most efficient antioxidizing and immunomodulating agent, and is known to modify intercellular exchange junctions. It is the most promising candidate in reducing hyperkeratosis of oral mucosa and can be used effectively in management of oral lichen planus lesions.29
Alpha-tocoferol (vitamin E) was also an effective antioxidant at high levels of oxygen, protecting cellular membranes from lipidic peroxidation. It can be used to reduce the size of lichen planus lesions.29
Capsule Lycostar contains a combination of lycopene, vitamin E, and retinoids. It was found to be very effective in the current case to reduce the gingival lesions, along with topical corticosteroids. Capsule Lycostar is an FDA-approved drug. Regarding the teratogenicity of retinoid supplementation, a systematic review and meta-analysis of 23 trials demonstrated that retinoid and carotenoids supplementation during pregnancy had no significant effect on maternal, fetal, neonatal, and early infant health outcome.30
Diclofenac mouthrinses (0.074%) were used to reduce the oral discomfort. Plaque control is also crucial because OLP is an inflammatory condition, and local irritants, such as plaque and calculus, consistently trigger exacerbations. An alcohol-free rinse should be prescribed to avoid desiccation and irritation of oral tissues. Furthermore, prophylactic use of 0.12% chlorhexidine gluconate rinse reduces the incidence of candidiasis during the corticosteroid therapy.31
Conclusion and Clinical Significance
Although oral lichen planus is considered rare in childhood, the presence of often-asymptomatic oral lesions should alert the clinician to such a diagnosis. The case described highlights the importance of considering lichen planus in the differential diagnosis of hyperkeratotic and erosive lesions of the oral mucosa in childhood and their management. Topical corticosteroid therapy, retinoid therapy, and a plaque control regimen in children with symptomatic erosive oral lichen planus have shown favorable responses. However, although not yet reported in children, periodic follow-up visits should be emphasized to monitor for possible malignant transformation.
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About the Authors
Gowri Pendyala, MDS
Department of Periodontics
Rural Dental College
Saurabh Joshi, MDS
Department of Pedodontics
Rural Dental College
Jithendra Kalburge, MDS
Department of Oral Pathology
Rural Dental College
Manjiri Joshi, MDS
Department of Oral Medicine and Radiology
Rural Dental College
Avneesh Tejnani, MDS
Post Graduate Student
Department of Periodontics Rural Dental College