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Oral juvenile xanthogranuloma: An unusual presentation in an adult patient and a systematic analysis of published cases

  • Gioele Gioco
    Affiliations
    Head and Neck Department, Fondazione Policlinico Universitario A Gemelli IRCCS, Roma, Università Cattolica del Sacro Cuore, Rome, Italy
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  • Cosimo Rupe
    Correspondence
    Reprint requests: Cosimo Rupe, DDS.Head and Neck DepartmentFondazione Policlinico Universitario A. Gemelli IRCCSUniversità Cattolica del Sacro Cuore Largo A. Gemelli 8, 00168RomeItaly.
    Affiliations
    Head and Neck Department, Fondazione Policlinico Universitario A Gemelli IRCCS, Roma, Università Cattolica del Sacro Cuore, Rome, Italy
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  • Alessia Basco
    Affiliations
    Head and Neck Department, Fondazione Policlinico Universitario A Gemelli IRCCS, Roma, Università Cattolica del Sacro Cuore, Rome, Italy
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  • Maria Contaldo
    Affiliations
    University of Campania-Luigi Vanvitelli, Multidisciplinary Department of Medical-Surgical and Odontostomatological Specialties, Naples, Italy
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  • Patrizia Gallenzi
    Affiliations
    Head and Neck Department, Fondazione Policlinico Universitario A Gemelli IRCCS, Roma, Università Cattolica del Sacro Cuore, Rome, Italy
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  • Carlo Lajolo
    Affiliations
    Head and Neck Department, Fondazione Policlinico Universitario A Gemelli IRCCS, Roma, Università Cattolica del Sacro Cuore, Rome, Italy
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Published:October 07, 2021DOI:https://doi.org/10.1016/j.oooo.2021.09.015
      Objective. The purpose of this study was to describe an unusual case of intraoral juvenile xanthogranuloma (JXG) and to perform a systematic review to investigate the available literature regarding oral JXGs.
      Study design. We present a new case of oral JXG arising in a 36-year-old Italian woman and conducted a systematic literature review in PubMed, Web of Science, and Scopus, according to the PRISMA guidelines.
      Results. Our review of the English-language literature yielded 34 cases of oral JXG, which included our case report.
      Conclusions. JXG is a non-Langerhans cell histiocytosis. Oral JXG has been reported, but it is a rare manifestation. Because of the rarity of oral lesions and possible variations in the clinical and histologic presentation, the correct diagnosis can be challenging, requiring a careful clinical and histopathologic evaluation with adjuvant immunohistochemical studies.
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