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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 8  |  Issue : 2  |  Page : 109-115

Multivariant lichen planus of lips and gingiva: Report of a case and a review


1 Department of Periodontics, V S Dental College, Bangalore, Karnataka, India
2 Department of Periodontology, V S Dental College, Bangalore, Karnataka, India
3 Dermatologist and Cosmetologist, Cuti Care Clinic, Chamrajpet, Bangalore, Karnataka, India

Date of Web Publication18-Dec-2018

Correspondence Address:
Apeksha Basawant Annigeri
Department of Periodontology, V S Dental College, Bengaluru - 560 004, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohs.ijohs_18_18

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  Abstract 


Oral lichen planus (OLP) is a relatively common inflammatory mucocutaneous disorder that frequently involves the oral mucosa. There are many etiological factors, and some among them are stress, diabetes mellitus, and hypothyroidism. An important complication of OLP is the development of oral squamous cell carcinoma, which led the World Health Organization to classify OLP as a potentially malignant disorder. This article reports a multivariant (reticular, erosive, and papular) case of lichen planus that has affected a 40-year-old medically compromised female who was managed successfully and still under follow-up.

Keywords: Corticosteroids, desquamative gingivitis, diabetes mellitus, hypothyroidism, lichen planus


How to cite this article:
Rekha S M, Annigeri AB, Galgali SR, Jagadish P. Multivariant lichen planus of lips and gingiva: Report of a case and a review. Int J Oral Health Sci 2018;8:109-15

How to cite this URL:
Rekha S M, Annigeri AB, Galgali SR, Jagadish P. Multivariant lichen planus of lips and gingiva: Report of a case and a review. Int J Oral Health Sci [serial online] 2018 [cited 2019 May 20];8:109-15. Available from: http://www.ijohsjournal.org/text.asp?2018/8/2/109/247799




  Introduction Top


Oral lichen planus (OLP) is a common chronic immunological inflammatory mucocutaneous disorder that affects the stratified squamous epithelium that varies in the appearance from keratotic (reticular or plaque-like) to erythematous and ulcerative. It is derived from the Greek word “lichen” means “tree moss” and Latin word “planus” means “flat.”[1],[2]


  Case Report Top


A 40-year-old female patient reported to the Department of Periodontics, Vokkaligara Sangha Dental College, Bangalore, with a chief complaint of burning sensation in the right and left posterior buccal mucosa for 15 days, and the sensation was insidious in onset and moderate in intensity, aggravated on consuming spicy food and relieved on intake of sugar. Burning sensation was assessed pre and posttreatment using numeric analog scale, and the patient rated pretreatment burning sensation with a score of 8. The patient gave a history of hypothyroidism for 18 years and is on medication (thyroxine) and diabetes mellitus Type II for 2 years and is on medication (glimepiride, metformin, voglibose, empagliflozin, and sitagliptin). The patient also had a history of increased cholesterol and is on rosuvastatin. Personal history revealed that she is on mixed diet and has no deleterious habits and under chronic stress as she has two daughters and is worried about their future. Stress was assessed using perceived stress scale, and the total score was 29 which indicated high perceived stress.

The patient had approached a dermatologist for her oral and lip lesions for which she was prescribed levocetirizine, topical triamcinolone, hydralipz, and multivitamins for lip lesions. On extraoral examination, multiple brownish black pigmented papules were noticed on the lips [Figure 1]. On intraoral examination, bilateral irregular erythematous, erosive, and reticular patches measuring 1.5 cm × 2.0 cm on the right posterior buccal mucosa [Figure 2] and 2.0 cm × 1.5 cm on the left posterior buccal mucosa were seen. A small reticular lesion was also present anterior to the lesion in the left buccal mucosa [Figure 3]. All these lesions exhibited faint white striae with mild melanin pigmentation. On palpation, the lesions were tender, smooth, and nonscrapable. Gingiva was observed with bleeding on probing and recession. Attached gingiva with respect to 17, 16, 13, 16, 17, 31, 32, 33, 34, 35, 46, 47, and 48 showed typical white striae with melanin pigmentation and was tender on palpation [Figure 4]. Systemic involvement was not noticed. Considering the history and clinical features of reticular, erosive, and papular lesions, lichen planus was considered as a provisional diagnosis and a differential diagnosis being lichenoid drug reaction.
Figure 1: Multiple pigmented papules on the lips

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Figure 2: Irregular erythematous erosive and reticular patches measuring about 1.5 cm × 2.0 cm on the right posterior buccal mucosa

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Figure 3: Irregular erythematous erosive and reticular patches measuring about 2.0 cm × 1.5 cm on the left posterior buccal mucosa along with a small reticular lesion

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Figure 4: Gingiva showing typical white striae with melanin pigmentation

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After taking patient's consent, incisional biopsy of the left buccal mucosa was done along with routine blood investigations. The patient's random blood sugar was 157 mg/dl, HbA1c was 7.1%, and thyroid-stimulating hormone level was 3.85. Subsequent histopathological examination of biopsy specimen showed stratified squamous epithelium with hyperkeratosis [Figure 5]. Focal areas of epithelium showed atrophy and hyperplasia with irregular rete ridges along with basal cell degeneration and focal basilar hyperplasia [Figure 6]. Focal area also showed inflammatory atypia with subepithelial melanosis [Figure 7].
Figure 5: Histopathological examination of biopsy specimen showing stratified squamous epithelium with hyperkeratosis

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Figure 6: Focal areas of epithelium showing atrophy and hyperplasia with irregular rete ridges along with basal cell degeneration and focal basilar hyperplasia

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Figure 7: Focal area showing inflammatory atypia with subepithelial melanosis

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The patient was treated with systemic corticosteroid tablet prednisolone in three doses as 40 mg, 20 mg, and 10 mg once daily for 5 days each and on antioxidant capsule lycored for 2 months once daily and on hexidine mouthwash in 1:1 dilution. The patient was asked to continue using hydralipz cream for the lesions on the lips which was prescribed by the dermatologist and discontinue levocetirizine, topical triamcinolone, and oral prophylaxis was also done.

During the recall visit after 15 days, there was considerable remission of the erythematous lesion, and the symptom of burning sensation was also subsided with a score of 0 from 8. The patient is still under follow-up [Figure 8], [Figure 9], [Figure 10], [Figure 11].
Figure 8: Blackish pigmentation representing healing of lip lesions

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Figure 9: Irregular erythematous reticular patches measuring about 1 cm × 1.5 cm on the right posterior buccal mucosa

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Figure 10: Irregular erythematous reticular patches measuring about 1.5 cm × 1.0 cm on the left posterior buccal mucosa

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Figure 11: Gingiva showing remission of white striae

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  Discussion Top


OLP is a common chronic immunological inflammatory mucocutaneous disorder that varies in the appearance from keratotic (reticular or plaque) to erythematous and ulcerative. The disease is most often reported in middle-aged patients of 30–60 years of age with a female: male ratio of 1.4:1. The prevalence of lichen planus varies from 0.5% to 2.2% and about 1%–2% of the world population suffer from lichen planus and 1.5% of Indians suffer from this disorder.[1],[3],[4]

Cutaneous lichen planus

Cutaneous lichen planus is characterized by polygonal flat-topped, violaceous papules, and plaques, which in some cases can be intensely itchy. The lesions may result in long-standing residual hyperpigmentation, especially in dark-skinned patients. Lichen planus lesions are generally symmetric in distribution and can affect any area of the body, but lichen planus tends to favor flexural surfaces of the forearms, wrists, ankles, dorsal surface of the hands, shins, trunk, and sacral region.[5],[6]

Etiology

The different etiological factors are genetic background, dental materials, drugs, infectious agents, autoimmunity, immunodeficiency, food allergy, stress, habits, trauma, diabetes mellitus, hypothyroidism, hypertension, malignant neoplasm, and bowel disease. In spite of extensive research, exact etiology is still unknown. The most accepted and current data suggest that OLP is T-cell-mediated disorder in which there is production of cytokines which leads to apoptosis. Autocytotoxic CD8 and T-cell trigger apoptosis of oral epithelial cells. Immune system is triggered due to the interaction between and among genetic environmental and lifestyle factors. Other theories include genetic background where the weak association between HLA antigen and lichen planus was found. Dental materials and infectious agents such as Gram-negative aerobic bacillus, spirochetes, and increased prevalence of Candida species were also suggested. Some reported the strong association of psychological factors such as higher level of anxiety, greater depression, and psychic disorders in patients with erosive lichen planus.[4],[7]

Clinical presentation

OLP has six presentations as follows:

  1. Reticular: the most common clinical form presents with fine, asymptomatic intertwined radiant lines such as pattern described as Wickham striae in a bilateral symmetrical form and involves the posterior mucosa of the cheek
  2. Papular: appearance of white papules usually coalesces forming a network of lines that may intersect or crisscross each other forming various patterns. Louis Frederick Wickham described the presence of fine white or gray lines or dots seen on the top of pruritis papular rashes on the skin in lichen planus
  3. Plaque-like: homogeneous well-demarcated white plaque but often not always surrounded by striae
  4. Erosive: the second most common form characterized by oral ulcers presenting with persistent, irregular areas of redness, ulcerations, and erosions covered with a yellowish slough. In 25% of people with erosive lichen planus, gums are involved described as desquamative gingivitis
  5. Atrophic: characterized by homogeneous red area, striae are frequently seen at the periphery
  6. Bullous: in 1892, Kaposi first described a distinctive clinical variant of disease with blisters, this form is rarer than other forms of lichen planus. It appears as a small bullae or vesicle ranging from 4 mm to 2 cm and rupture leaving erythematous zone.[4],[7],[8],[9],[10]


Diabetes mellitus and lichen planus

Diabetes mellitus refers to a group of disorders characterized by elevated levels of blood glucose and leads to variety of abnormalities of carbohydrates, fat, and protein metabolism. The oral mucosa is normally protected by saliva when it is adequate in amount and quality. However, salivary gland function and immune function are negatively affected by diabetes. Diabetes patients are at increased risk for mucosal lesions and other disorders. OLP is found to be one of the most frequent oral manifestations observed in diabetic patients. There are numerous reports assuming an association between diabetes mellitus and OLP, based on immunological changes as well. The prevalence of manifestations of diabetes mellitus among OLP patients varies between 1.6% and 37.7% according to different investigators. Lichen planus showed the highest occurrence in the 2nd year of established diabetes, and their prevalence was higher among insulin-treated diabetes. According to the majority of reports, OLP seems to show a higher prevalence in diabetes mellitus. However, it must be taken into consideration that some antidiabetic drug might also produce lesions resembling lichen planus.[11],[12],[13]

Hypothyroidism and lichen planus

Some external and/or internal antigens have been suggested to trigger OLP although the true mechanism is not understood yet. Based on the immunological pathogenesis of OLP, research has shown the presence of serum antibodies to thyroglobulin and thyroid microsomal antibodies in OLP patients. The concept of possible association between OLP and thyroid diseases particularly hypothyroidism has originated from several reports of patients who were affected by both lichen planus and thyroid diseases, and the first case report was identified in 1994, but the association is controversial. Patients with any autoimmune disease are more susceptible to other autoimmune diseases. In the systemic review by Li et al., the odds ratio for the association between OLP and thyroid disease varied from 1.71 to 4.16, demonstrating a statistically significant difference in the prevalence of thyroid disease between OLP patients and controls. From these studies, we can assume that thyroid disease may be involved in the pathogenesis of OLP or that OLP is a clinical manifestation of thyroid disease.[14],[15],[16]

Lichen planus with systemic illness

Carrozzo et al. (2003), Assieta et al. (2000), and Jubert et al. (1984) found the frequent association of hepatitis C virus and OLP and suggested that hepatitis C virus occasionally replicates in OLP tissue contributing to the pathogenesis of mucosal damage. Lichen planus is often associated with immune-mediated diseases such as alopecia areata, dermatomyositis, lichen sclerosus et atrophicus, morphea, myasthenia gravis, ulcerative colitis, and primary biliary sclerosis.[4]

Syndromes associated with oral lichen planus

Grinspan syndrome is association of OLP with diabetes mellitus and hypertension. Graham-Little syndrome and vulvovaginal syndrome are other syndromes associated with OLP, in which there is mucosal involvement of gingival and genital region usually of erosive type.[1],[4]

Differential diagnosis

Differential diagnosis of the reticular type of OLP includes lichenoid drug reaction, electrogalvanic white lesions, frictional keratosis, and leukoplakia.

Keratotic form of lichen planus can be differentiated from leukoplakic plaque as the former is usually associated with burning sensation and association with etiologic factors in the later.

Erosive lichen planus resembles lupus erythematosus and has to be differentiated from the lichenoid reaction, graft versus host disease, discoid lupus erythematosus, and speckled leukoplakia.

Desquamative gingivitis of gingival erosive lichen planus should be differentiated from other forms such as pemphigus, pemphigoid, and linear IgA disease.[1],[3],[4]

Histopathology

Areas of hyperparakeratosis or hyperorthokeratosis often with the thickening of granular cell layer and a saw-tooth appearance to the rete pegs, liquefaction degeneration or necrosis of basal cell layer, and an eosinophilic band may be seen just beneath the basement membrane and contain fibrin covering the lamina propria. A dense subepithelial band-shaped infiltrate of lymphocytes and macrophages is also seen. Degenerating basal keratinocytes that form colloid (Civatte, hyaline, and cytoid) bodies appear as homogeneous eosinophilic globules. This degeneration and disruption of the anchoring elements of the epithelial basement membrane and basal keratinocytes weaken the epithelial-connective tissue interface. As a result, histological clefts (Max-Joseph spaces) may form, and blisters on the oral mucosa (bullous lichen planus) may be seen.[1],[3]

Malignant transformation

The malignant potential of lichen planus is controversial; there seems to be a slightly higher incidence of oral squamous cell carcinoma in patients with OLP than in the general population. The actual overall frequency of malignant transformation is low varying between 0.3% and 3%. The forms that more commonly undergo malignant transformation are the erosive and atrophic forms.[9]

Management

The main aim of current OLP therapy is to resolve the painful symptoms of oral mucosal lesions to reduce the risk of oral cancer and to maintain good oral hygiene and to eliminate the local exacerbating factors as preventive measures. In the present case, after thorough oral prophylaxis patient was treated with systemic steroid prednisolone in gradually tapering doses due to its anti-inflammatory and immunosuppressive actions. In addition, lycopene was advised as it acts as a potent antioxidant and a free radical scavenger thus reducing the oxidative stress which has a role in disease pathogenesis. The patient showed a favorable response with systemic steroids in combination with an antioxidant.

The various treatment modalities include psychotherapy as exacerbation of OLP has been often linked to periods of psychological stress and anxiety, and the patient should be promptly referred for psychological counseling. Relaxation, meditation, and hypnosis have positive impact as they help to calm and rebalance the inflammatory response which can ameliorate inflammatory skin or mucosal disorders. Systemic and topical corticosteroids are the mainstay of treatment for OLP lesions, and they function by modulating the patient's immune response.[4],[9],[12]

Erythematous OLP constitutes a therapeutic challenge. To be successful, it is critical to remove both subgingival and supragingival plaque and calculus. If the microbial plaque-induced gingivitis is present, it seems to work in concert with gingival lichen planus and make it more resistant to pharmacological treatment. Hence, oral hygiene should be optimized before the beginning of steroid treatment. Once the oral hygiene treatment is complete, some patients experience a decrease or even elimination of symptoms, and steroid treatment is no longer justified. If symptoms persist steroid gels in prefabricated plastic trays may be used for 30 min at each application to increase the concentration of steroids in gingival tissue.[1]

Drugs used systemically are dapsone, azathioprine, levamisole, thalidomide, metronidazole, griseofulvin, hydroxychloroquine, retinoids, and corticosteroids. Systemic steroid therapy should be reserved for patients who are recalcitrant to topical steroid management. When systemic periods of corticosteroids last greater than 2 weeks, dosages of steroids must be gradually tapered to avoid precipitating adrenal crisis.[1],[2],[3],[4],[7],[8]

Topical application of cyclosporine, tacrolimus, and retinoids has been suggested as a second-line therapy for OLP. Topical corticosteroids are mainly implemented for treating mild to moderately symptomatic lesions. The common topical formulations used are 0.04% clobetasol propionate gel, 0.1% or 0.05% betamethasone valerate gel, 0.05% fluocinonide gel, 0.05% clobetasone butyrate ointment or cream, 0.1% triamcinolone acetonide ointment and 0.1% tacrolimus or pimecrolimus ointment, hydrocortisone hemisuccinate aqueous solution, fluticasone propionate spray, and betamethasone sodium phosphate mouthrinse. Mometasone furoate microemulsion, clobetasol propionate 0.05% in orabase, ointment, or aqueous solution has shown to be effective. In lesions recalcitrant to topical therapy, intralesional corticosteroids can be affective, often triamcinolone acetonide 5 mg/ml combined with local anesthesia to inject 0.1 ml/cm. Retinoids are effective and usually used in combination with topical steroids as adjuvant therapy.[1],[2],[4],[7],[8],[10]

Other treatment modalities include amlexanox 5% paste, topical anti-inflammatory and immunomodulatory drug. Aloe vera which is also an anti-inflammatory, antibacterial, antiviral, and antifungal has a hypoglycemic effect. Curcuminoids, a main component of turmeric, has also shown to be effective. Curcumin acts through suppression of inflammatory response and is effective. Lycopene a red-colored carotenoid is helpful in reducing OLP symptoms. Photochemotherapy a new method in which clinician uses ultraviolet A with wavelengths ranging from 320 nm to 400 nm after the injection psoralen is used. Surgical excision, cryotherapy, carbon dioxide laser, and ND:YAG laser have all been used in the treatment of OLP.[1],[4],[7],[8],[9]


  Conclusion Top


OLP is a heterogeneous group of disease afflicted with mucosal involvement, identifying and eliminating multifactorial agents associated with the disease such as psychological stress, and various systemic illnesses such as diabetes mellitus, hypothyroidism, or drugs for the treatment of these systemic illnesses are essential. Palliative treatment can be achieved with topical steroids alone or in combination with other immunomodulatory agents. Patients should be kept under long-term follow-up due to lack of complete resolution and the malignant tendency of lichen planus.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Acknowledgment

We would like to acknowledge the Department of Oral Medicine and Radiology and Department of Oral Pathology, V S Dental College, Bangalore.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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Li D, Li J, Li C, Chen Q, Hua H. The association of thyroid disease and oral lichen planus: A literature review and meta-analysis. Front Endocrinol (Lausanne) 2017;8:310.  Back to cited text no. 14
    
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]



 

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