Abstract

IntroductionJuvenile xanthogranuloma is a rare non-Langerhans cell histiocytosis that usually occurs during infancy and early childhood. The presence of single or multiple raised cutaneous lesions characterize this self-healing disorder. Extracutaneous sites are rare.Case presentationWe present a rare case of oral juvenile xanthogranuloma in a 49-year-old Caucasian woman. The histopathologic diagnosis of the lingual neoformation was histiocitary proliferation with the presence of giant cells, Touton type, compatible with juvenile xanthogranuloma.ConclusionTo establish an accurate diagnosis, microscopic evaluation and immunohistochemical staining are necessary. Dentists, dermatologists and general practitioners may be the first to recognize this rare condition during the inspection of the oral cavity.

Highlights

  • Juvenile xanthogranuloma is an uncommon nonLangerhans cell histiocytosis that usually occurs during infancy and early childhood

  • Juvenile xanthogranuloma is a benign disorder which characteristically develops as a well-demarcated mass in the dermis of a child

  • Kawashiri S, Kumagai S, Nakagawa K, Yamamoto E, Imai K: Juvenile xanthogranuloma occurring in the oral cavity: case report and histopathological findings

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Summary

Introduction

Juvenile xanthogranuloma is an uncommon nonLangerhans cell histiocytosis that usually occurs during infancy and early childhood. This lesion was first reported by Adamson in 1905 [1], who used the term congenital xanthoma multiplex. Case presentation Approximately five years ago, a 49-year-old Caucasian woman came to our hospital for diagnosis, evaluation and treatment of a lingual lesion. She complained of constant dysphagia and dysgeusia. These symptoms had worsened in the past three months because of growth of the lingual mass (Figure 1) Her medical history was uneventful, except for breast cancer several years earlier, and included no history of smoking or drinking. A hyperchromic area and a mild atrophy remain, without any evidence of local recurrence after follow-up of more than four years (Figure 4)

Discussion
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