Igor Bartenjev

Ivana Škrinjar

Lovorka Batelja Vuletić

Mirna Bradamante

Suzana Ljubojević Hadžavdić

Vanja Vučičević Boras

English

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195Oral and Vulvar Lichen SclerosusActa Dermatovenerol Croat                     2019;27(3):195-197                            LETTER TO THE EDITORLichen sclerosus (LS) is a chronic, inflammatory, mucocutaneous disorder of genital and extragenital skin (1). Simultaneous involvement of the oral mu-cosa is extremely rare, but it may be the only affected area (2). A 55-year-old woman was referred to the Depart-ment of Oral Medicine, School of Dental Medicine University of Zagreb due to whitish lesions on the right ventrolateral part of the tongue and buccal mu-cosa with desquamative gingivitis (Figure 1, a-c). The lesions were asymptomatic but indurated on palpa-tion. Histology was conclusive for oral lichen sclero-sus (OLS). The lesions on gingiva were successfully treated with betamethasone ointment, three times a day for two weeks.One year earlier, she had been referred to the Department of Dermatology and Venereology with progressive pruritus and dyspareunia, white patches, obliteration of the labia minora, and stenosis of the introitus (Figure 2). Histology was conclusive for vul-var LS (Figure 3, a and b). She was successfully treated for 5 months with clobetasol propionate 0.05% oint-ment. The patient was taking levothyroxine to treat hypothyroidism associated with Hashimoto’s thyroid-itis and was otherwise healthy. Oral LS is clinically characterized by the appear-ance of white macules, papules, or plaques mostly appearing on labial mucosa but also on buccal, pal-ate mucosa and on the lower lip (2,3). On the geni-tals, it typically manifests as atrophic white plaques, which may be accompanied by purpura or fissuring (1). While vulvar LS is often associated with pruritus, dyspareunia, and dysuria, OLS is often asymptomatic, although pain, soreness, pruritus, and tightness when opening the mouth can be present (1,2). Oral mani-festations of LS, as well as association of anogenital and oral LS, are rarely reported in the literature (4-6). Tomo et al. searched the Medline database for papers reporting oral LS cases with histological diagnosis confirmation from 1957 to 2016 and found only 34 cases of oral LS with histopathologic confirmation of the diagnosis (4). Kakko et al. reported 39 histologi-cally proven cases of OLS (2). Attilli et al. (5) reviewed Figure 1a. Whitish lesions on the ventrolateral part of the tongue in patient with lichen sclerosusFigure 1b. Whitish lesions of the buccal mucosa in patient with lichen sclerosusFigure 1c. Desquamative gingivitis in upper jaw in patient with lichen sclerosusACTA DERMATOVENEROLOGICA CROATICA196 ACTA DERMATOVENEROLOGICA CROATICALetter to the editor Acta Dermatovenerol Croat    2019;27(3):195-197the clinical and histologic features of 72 cases of LS with oral/genital involvement. They reported that LS was diagnosed with exclusive genital lesions in 45, exclusive lip involvement in 20, and orogenital in-volvement in only 7 cases (5). Some believe that many cases of clinically diag-nosed lichen planus may actually be LS and that iso-lated oral mucosal LS may not be as rare as is gener-ally thought (2). While vulvar LS can occur at any age with increasing incidence with age, the median age of patients with OLS was 34 years and most of the pa-tients were female (1,2,5). Due to the small number of patients in the litera-ture, treatment recommendations for OLS are not available. In case of symptomatic oral lesions, topi-cal or intralesional corticosteroids are considered to be the first-line treatment (2). First-line treatment for anogenital LS is a potent to very potent topical cor-ticosteroid ointment, and second-line therapies in-clude topical calcineurin inhibitors 1% pimecrolimus and 0.1% and 0.03% tacrolimus (1). For treatment-re-sistant genital LS, oral retinoids, methotrexate, and possibly local steroid injections for single lesions are mainly applicable for women (1). There is limited evi-dence for systemic treatments for both conditions. If it is not treated, genital LS is associated with a greater degree of scarring and an elevated risk of progression to squamous cell cancer; however, ma-lignant transformation of OLS has not been reported (1-6).Due to the very rare presentation in the oral cav-ity, it is important to notice these lesions during a dental exam.References:1. Kirtschig G, Becker K, Günthert A, Jasaitiene D, Cooper S, Chi CC, et al. Evidence-based (S3) Gui-deline on (anogenital) lichen sclerosus. J Eur Acad Dermatol Venereol. 2015;29:e1-43.2. Kakko T, Salo T, Siponen MK. Oral lichen sclerosus: a systematic review of reported cases and two new cases. Int J Dermatol. 2018;57:521-8.3. Bevans SL, Keeley JM, Sami N. Oral lichen sclero-sus-a review of clinical presentation, treatment, and clinical outcomes. Oral Surg Oral Med Oral Pathol Oral Radiol. 2017;124:e243-e248.4. Tomo S, Santos IS, de Queiroz SA, Bernabé DG, Simonato LE, Miyahara GI. Uncommon oral ma-nifestation of lichen sclerosus: critical analysis of cases reported from 1957 to 2016. Med Oral Patol Oral Cir Bucal. 2017;22:e410-e416.Figure 3a and 3b. Hyperkeratotic stratified squamous epi-thelium, partly normal thickness, partly with evidence of thinning, and with loss of the normal rete ridge. The strati-fied squamous epithelium shows hydropic degeneration of basal and parabasal cells in few areas. The subjacent con-nective tissue showed dense linear subepithelial inflamma-tory cell infiltrate. (Hematoxylin and eosin, 10x and 40x)Figure 2. White patches, obliteration of the labia minora and stenosis of the introitus in 55-year old patients with vulvar lichen sclerosus197ACTA DERMATOVENEROLOGICA CROATICA5. Attili VR, Attili SK. Lichen sclerosus of lips: a clinical and histopathologic study of 27 cases. Int J Der-matol. 2010;49:520-5.6. Louvain D, Moura Jacques C, Fernandes Ferreira A, Hoehl Carneiro L, Quintela L, Cuzzi T, et al. Lichen sclerosus in the oral mucosa: a rare form of presen-tation. Acta Dermatovenerol Croat. 2012;20:43-7.Vanja Vučićević Boras 1,2, Ivana Škrinjar2, Lovorka Batelja Vuletić3, Mirna Bradamante4, Igor Bartenjev5, Suzana Ljubojević Hadžavdić41Department of Oral Medicine, School of Dental Medicine, University of Zagreb, Zagreb, Croatia 2Department of Oral Medicine, University Hospital Center Zagreb, Zagreb, Croatia3Department of Pathology, University Hospital Center Zagreb, University of Zagreb School of Medicine, Zagreb, Croatia4Department of Dermatology and Venereology, University Hospital Center Zagreb, University of Zagreb School of Medicine, Zagreb, Croatia5Department of Dermatovenerology, Medical faculty, University of Ljubljana, Ljubljana, SloveniaCorresponding author:Ivana Škrinjar, MDDepartment of Oral Medicine, University Hospital Center ZagrebGundulićeva 510000 ZagrebCroatiaskrinjar.ivana@gmail.comReceived: November 30, 2018Accepted: Augist 7, 2019Letter to the editor Acta Dermatovenerol Croat    2019;27(3):195-197

Oral and vulvar lichen sclerosus

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