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

Year 2023, Volume: 25 Issue: 1, 85 - 88, 30.04.2023

Ayat Gamal-abdelnaser

https://doi.org/10.18678/dtfd.1182644

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

Abstract

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.

Keywords

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

References

• Cascone M, Celentano A, Adamo D, Leuci S, Ruoppo E, Mignogna MD. Oral lichen planus in childhood: a case series. Int J Dermatol. 2017;56(6):641-52.
• George S, John SA, Anandaraj S, Issac JS, Harris A, Reshmi J. Childhood oral lichen planus: report of two cases. J Dent (Tehran). 2015;12(5):374-8.
• Jahan AJ, Vandana S, Rangdhol V, Palla S. Juvenile oral erosive lichen planus - A rare case report. Int J Med Dent Case Reports. 2017;4(1):1-3.
• Chatterjee K, Bhattacharya S, Mukherjee CG, Mazumdar A. A retrospective study of oral lichen planus in paediatric population. J Oral Maxillofac Pathol. 2012;16(3):363-7.
• Chaitra TR, Telgi RL, Kishor A, Kulkarni AU. Juvenile oral lichen planus: a clinical rarity. BMJ Case Rep. 2012;2012: bcr2012006192.
• Hugar SM, Reddy R, Gokhale N, Hugar SS. Oral lichen planus in childhood: A rare case report. J Adv Clin Res Insights. 2015;2(5):226-8.
• Wang F, Tan YQ, Zhang J, Zhou G. Familial oral lichen planus in a 3-year-old boy: a case report with eight years of follow-up. BMC Oral Health. 2020;20(1):341.
• Sharma G, Sardana D, Vohra P, Rehani S, Nagpal A. Oral lichen planus in a pediatric patient: a novel therapeutic approach. J Dent (Tehran). 2017;14(2):109-14.
• Chinnasamy NK, Venugopal DC, Sankarapandian S, Narasimhan M. Oral lichen planus in a 7-year-old child: A rare case report. Int J Clin Pediatr Dent. 2020;13(1):91-3.
• Woo VL, Manchanda-Gera A, Park DS, Yoon AJ, Zegarelli DJ. Juvenile oral lichen planus: A report of 2 cases. Pediatr Dent. 2007;29(6):525-30.
• Alam F, Hamburger J. Oral mucosal lichen planus in children. Int J Paediatr Dent. 2001;11(3):209-14.
• Laeijendecker R, Van Joost T, Tank B, Oranje AP, Neumann HAM. Oral lichen planus in childhood. Pediatr Dermatol. 2005;22(4):299-304.
• Georgaki M, Andreou A, Tziveleka S, Nikitakis NG. Oral erosive/ulcerative lichen planus in a 3 years old child : a case report. Int J Res Reports Dent. 2022;5(2):1-6.
• Padmini C, Bai KY, Chaitanya V, Reddy MS. Ulcerative lichen planus in childhood. Case Rep Dent. 2013;2013:874895.
• Shikha, Gupta S, Mahajan A, Ambika, Garg R, Ghosh S. Childhood oral lichen planus: a case series with review of literature. Eur Arch Paediatr Dent. 2022;23(2):341-53.
• Larsson A, Warfvinge G. Immunohistochemistry of 'tertiary lymphoid follicles' in oral amalgam-associated lichenoid lesions. Oral Dis. 1998;4(3):187-93.
• Yiannias JA, el-Azhary RA, Hand JH, Pakzad SY, Rogers RS 3rd. Relevant contact sensitivities in patients with the diagnosis of oral lichen planus. J Am Acad Dermatol. 2000;42(2 Pt 1):177-82.