Erdheim-Chester disease in a child presenting with multiple jaw lesions

Hitoshi Nagatsuka, Phuu Pwint Han, Koji Taguchi, Hidetsugu Tsujigiwa, Mehmet Gunduz, Joji Fukunaga, Toshio Sugahara, Junichi Asaumi, Noriyuki Nagai

Research output: Contribution to journalArticlepeer-review

17 Citations (Scopus)


BACKGROUND: Erdheim-Chester disease is a rare histiocytic disease entity related to juvenile xanthogranuloma. It is a systemic condition, usually occurs in adult, characterized by infiltration of foamy histiocytes within the bone and soft tissues. METHODS AND RESULTS: We report a case of 13-year-old female patient who first presented with multiple osteolytic lesions of the jaws followed by bilateral symmetrical bone lesions affecting the lower extremities, as well as brain and abdominal involvement. Histological findings of the jaw lesions showed lipid-storing CD68 (+), CDIa (-) histiocytes with Touton type giant cells. CONCLUSION: To the best of our knowledge, this is the first case of Erdheim-Chester disease with jaw bone lesions occurring as initial presenting symptom.

Original languageEnglish
Pages (from-to)420-422
Number of pages3
JournalJournal of Oral Pathology and Medicine
Issue number7
Publication statusPublished - Aug 2005


  • Bilateral symmetrical bony lesions
  • Erdheim-Chester disease
  • Jaws
  • Lipid-storing histiocytes
  • Touton-type giant cells

ASJC Scopus subject areas

  • Pathology and Forensic Medicine
  • Oral Surgery
  • Otorhinolaryngology
  • Cancer Research
  • Periodontics


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