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An 80-year-old white woman presented with an asymptomatic white patch affecting the tongue of 1 month’s duration. She had a 15-year history of oral lichen planus (OLP) managed with betamethasone dipropionate, 0.05%, gel twice daily and clotrimazole troches, 10 mg, 3 times daily as needed for symptomatic OLP flares. Medical history revealed stage 1A mycosis fungoides/cutaneous T-cell lymphoma affecting the right calf and left thigh managed primarily with halobetasol, 0.05%, cream owing to intolerance of narrowband UV-B therapy and mechlorethamine, 0.016%, gel. Immunosuppression or history of infectious diseases, including human immunodeficiency virus (HIV), was not reported. A 1.5 × 1.0-cm nonremovable white, plaquelike lesion was observed on the left lateral tongue (Figure 1A). Biopsy specimens were obtained with a 3-mm punch instrument at 3 different sites, which demonstrated similar microscopic findings (Figure 1B and C).
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C. Oral hairy leukoplakia
Microscopic analysis of all specimens demonstrated hyperkeratosis and acanthosis with a band of cells characterized by lightly stained cytoplasm (balloon cells). The superficial cells contained nuclei with peripheral chromatin margination or nuclear beading typical of Epstein-Barr virus–infected oral keratinocytes and consistent with oral hairy leukoplakia (OHL). The patient was prescribed valacyclovir 1 g 3 times daily, which she discontinued taking after 3 days owing to medication adverse effects. On reexamination, the tongue lesion had completely resolved (Figure 2).
Physicians would consider a clinical diagnosis of oral leukoplakia when they find a white plaque of questionable risk and have excluded other known diseases or disorders that carry no increased risk for cancer.1,2 Oral hairy leukoplakia was first described in 1984 during the AIDS epidemic and named for its white color and corrugated appearance.3 Historically, it has been associated with HIV infection and as a sign of disease progression.3 Recently, several reports describe OHL in non–HIV infected individuals in association with inhaled or topical corticosteroids.4- 6 Epstein-Barr virus is a DNA B-lymphotropic human herpesvirus that infects more than 90% of the world’s population and is associated with the etiopathogenesis of OHL.4,5 Typically, OHLs appear on the lateral borders of the tongue and are asymptomatic. Microscopic analysis of OHLs may demonstrate several histopathologic features: (1) hyperkeratosis and acanthosis, (2) ballooning degeneration of keratinocytes, and (3) little to no inflammatory infiltrate in the lamina propria.5 However, peripheral chromatin margination or nuclear beading is the only essential diagnostic criteria, and in situ hybridization for Epstein-Barr virus detection is often used to confirm the diagnosis.5 Although treatment of OHL is not typically warranted, lesions have responded to antiviral agents such as acyclovir and valacyclovir.3 In the present case, development of OHL may have occurred due to long-term use of topical corticosteroids for OLP management in the absence of any known systemic immunosuppressive conditions. The patient was recommended for further evaluation of possible immunosuppression. It was indeterminate if OHL resolution was attributed to use of valacyclovir or spontaneous resolution.
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Corresponding Author: Eric T. Stoopler, DMD, FDSRCS, FDSRCPS, Department of Oral Medicine, University of Pennsylvania School of Dental Medicine, 240 S 40th St, Philadelphia, PA 19104 (firstname.lastname@example.org).
Published Online: August 8, 2018. doi:10.1001/jamadermatol.2018.1571
Conflict of Interest Disclosures: Dr Stoopler is employed by the University of Pennsylvania, receives stipend for membership on the executive committee of the American Academy of Oral Medicine, receives payment and honoraria for continuing education lectures, received payment from Elsevier Inc for book preparation, received payment from Metropolitan Life Insurance Company for monograph preparation, and received payment for expert legal advice. Dr Sollecito is employed by the University of Pennsylvania, received payment and honoraria for continuing education lectures, received payment from Elsevier Inc for book preparation, received payment from Metropolitan Life Insurance Company for monograph preparation, is a consultant to Dermatreat Corp, and received payment for expert legal advice. Dr Alawi is employed by the University of Pennsylvania, receives payment and honoraria for continuing education lectures, and received payment from Springer for editorship. No other disclosures are reported.
Additional Contributions: We thank the patient for granting permission to publish this information.
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