Oral Lichen Planus in Childhood with Unique Histological Finding: A Case Report

Abstract

Oral lichen planus is a chronic inflammatory disease that targets adults. Over the years, few reports documented solitary cases of children being affected by oral lichen planus. In this report, a case of an 8-year-old, medically uncompromised boy who complained of a severe burning sensation in the tongue with a history of one year of remission and exacerbation was presented. When examined, the tongue showed a large irregular superficial non-indurated ulcer occupying the whole anterior 2/3 of the tongue sparing the tip and was surrounded by white radiating lines. The ulcerative lesion showed a peculiar butterfly pattern. Histopathological examination of the incisional biopsy showed intraepithelial civatte bodies and dense subepithelial lymphocytic infiltrate arranged in a pseudo-lymphomatous shape. Although the clinical and histological pictures suggest lichen planus, the atypical arrangement of lymphocytes brought up a question mark. This report demonstrates a peculiar clinical picture of a rare disease combined with a unique histopathologic finding.

Keywords

• Civatte bodies
• juvenile oral lichen planus
• oral lichenoid lesions
• tertiary lymphoid follicle

Çocukluk Çağında Benzersiz Histolojik Bulgusu Olan Oral Liken Planus: Olgu Sunumu

Öz

Oral liken planus yetişkinleri hedef alan kronik inflamatuar bir hastalıktır. Yıllar boyunca, oral liken planustan etkilenen tekil çocuk vakaları az sayıda raporla belgelenmiştir. Bu raporda, 8 yaşında, medikal açıdan herhangi bir sorunu olmayan, dilde şiddetli yanma şikayeti ile başvuran, bir yıldır remisyon ve alevlenme öyküsü olan bir erkek çocuk olgusunu sunulmuştur. Dil muayenesinde dil ucu hariç anterior 2/3'lük bölümün tamamını kaplayan, etrafı beyaz yayılan çizgilerle çevrili, büyük, düzensiz, yüzeyel, sertleşmemiş ülser saptandı. Ülseratif lezyon tuhaf bir kelebek paterni gösterdi. İnsizyonel biyopsinin histopatolojik incelemesinde intraepitelyal civatte cisimcikleri ve psödo-lenfomatöz bir şekilde düzenlenmiş yoğun subepitelyal lenfositik infiltrat görüldü. Klinik ve histolojik tablo liken planusu düşündürse de, lenfositlerin atipik dizilimi bir soru işareti yaratmıştır. Bu rapor, benzersiz bir histopatolojik bulguya sahip nadir bir hastalığın kendine özgü klinik tablosunu göstermektedir.

Anahtar Kelimeler

• Civatte cisimcikleri
• Juvenil oral liken planus
• Oral likenoid lezyonlar
• Üçüncül lenfoid folikül

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