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Xanthogranuloma juvenile: a case report.

T Mortensen, K Weismann, T Kobayasi

    Acta Dermato-Venereologica
    |January 1, 1983
    PubMed
    Summary
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    Juvenile xanthogranuloma (JXG) in newborns presents as red-brown lesions that spontaneously resolve within a year. Histiocytes and Touton giant cells characterize the dermal infiltrate in this rare condition.

    Area of Science:

    • Dermatology
    • Pediatric Pathology
    • Neonatology

    Background:

    • Juvenile xanthogranuloma (JXG) is a rare, benign, non-Langerhans cell histiocytosis typically affecting infants and young children.
    • While commonly presenting in the skin, JXG can involve extracutaneous sites, posing diagnostic challenges.
    • Understanding the histopathological features of congenital JXG is crucial for accurate diagnosis and management.

    Observation:

    • A case report details a female newborn infant presenting with congenital JXG.
    • The infant exhibited multiple red-brown, flat, well-demarcated cutaneous lesions on the scalp, face, and trunk at birth.
    • These lesions demonstrated slow, spontaneous regression over a one-year observation period.

    Findings:

    • Histopathological examination revealed a dermal infiltrate predominantly composed of histiocytes and Touton giant cells.

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  • Electron microscopy of Touton cells showed characteristic features including bizarre invaginated nuclei, lysosomes, granular endoplasmic reticulum, and intracytoplasmic cholesterol clefts.
  • Notably, no intracellular fat droplets were observed within the Touton cells.

    • Implications:

    • This case highlights the presentation and natural history of congenital juvenile xanthogranuloma.
    • The detailed ultrastructural findings of Touton giant cells contribute to the understanding of JXG histiogenesis.
    • The spontaneous regression observed underscores the generally benign prognosis of cutaneous JXG in neonates.