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Oral Hairy Leukoplakia Related to Orodispersible Budesonide Use

McNeice, Andrew MRCP1; Loughrey, Maurice B. BSc, MRCP, FRCPath, MD2; Mainie, Inder MD, FRCP1

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doi: 10.14309/crj.0000000000000502
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CASE REPORT

A 46-year-old nonsmoking man with a history of eosinophilic esophagitis (EoE), for which he was taking orodispersible budesonide 1 mg twice daily, presented with a painless, white plaque on his tongue (Figure 1). He had been taking orodispersible budesonide for 7 months. A clinical diagnosis of oral hairy leukoplakia was made, and this was supported by biopsy findings. Histology showed surface hyperkeratosis and an underlying band of cells with clear cytoplasm and koilocyte-like nuclear changes (Figure 2). The latter cells stained positively for Epstein-Barr virus (EBV) RNA in situ hybridization (Figure 3). The patient's immune screen, including human immunodeficiency virus (HIV) testing, revealed no immunodeficiency. Medication was stopped for 5 days and recommenced once daily, with ongoing improvement of the lesion and EoE control maintained.

Figure 1.
Figure 1.:
White plaque on patient's tongue.
Figure 2.
Figure 2.:
Surface hyperkeratosis and an underlying band of cells with clear cytoplasm and koilocyte-like nuclear changes.
Figure 3.
Figure 3.:
In situ hybridization on underlying band of cells demonstrating positive staining for Epstein-Barr virus RNA.

We report the first case of oral hairy leukoplakia in a patient taking orodispersible budesonide. Improvement on drug discontinuation supports causation. Oral hairy leukoplakia is typically associated with HIV infection and likely due to secondary EBV infection. This patient was immunocompetent and HIV-negative, but the lesion demonstrated evidence of local EBV infection. As orodispersible budesonide is taken orally and allowed to disperse on the tongue before swallowing, it is likely that the budesonide reduced the local immunity on the tongue's surface, allowing for opportunistic EBV infection and leading to oral hairy leukoplakia. The patient stated that he slept on his right side, which explains the lesion being on this side of his tongue. Removing the nocte dose of orodispersible budesonide resulted in ongoing improvement of the oral hairy leukoplakia, while still being sufficient to maintain EoE control. Oral hairy leukoplakia has been previously reported in a small series in patients lacking immunosuppression but with lesional EBV infection.1 The patient remains under clinical review.

DISCLOSURES

Author contributions: All authors contributed equally to this manuscript. A. McNeice is the article guarantor.

Financial disclosure: None to report.

Informed consent was obtained for this case report.

REFERENCE

1. Darling MR, Alkhasawneh M, Mascarenhas W, Chirila A, Copete M. Oral hairy leukoplakia in patients with no evidence of immunosuppression: A case series and review of the literature. J Can Dent Assoc 2018;84:i4.
© 2020 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of The American College of Gastroenterology.