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, Jun-Ichi Asaumi, Noriyuki Nagai

Research output: Contribution to journalArticle

10 Citations (Scopus)

Abstract

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
Volume34
Issue number7
DOIs
Publication statusPublished - Aug 2005

Fingerprint

Erdheim-Chester Disease
Jaw
Histiocytes
Bone and Bones
Juvenile Xanthogranuloma
Giant Cells
Rare Diseases
Lower Extremity
Lipids
Brain

Keywords

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

ASJC Scopus subject areas

  • Cancer Research
  • Pathology and Forensic Medicine
  • Dentistry(all)

Cite this

Erdheim-Chester disease in a child presenting with multiple jaw lesions. / Nagatsuka, Hitoshi; Han, Phuu Pwint; Taguchi, Koji; Tsujigiwa, Hidetsugu; Gunduz, Mehmet; Fukunaga, Joji; Sugahara, Toshio; Asaumi, Jun-Ichi; Nagai, Noriyuki.

In: Journal of Oral Pathology and Medicine, Vol. 34, No. 7, 08.2005, p. 420-422.

Research output: Contribution to journalArticle

Nagatsuka, H, Han, PP, Taguchi, K, Tsujigiwa, H, Gunduz, M, Fukunaga, J, Sugahara, T, Asaumi, J-I & Nagai, N 2005, 'Erdheim-Chester disease in a child presenting with multiple jaw lesions', Journal of Oral Pathology and Medicine, vol. 34, no. 7, pp. 420-422. https://doi.org/10.1111/j.1600-0714.2005.00320.x
Nagatsuka, Hitoshi ; Han, Phuu Pwint ; Taguchi, Koji ; Tsujigiwa, Hidetsugu ; Gunduz, Mehmet ; Fukunaga, Joji ; Sugahara, Toshio ; Asaumi, Jun-Ichi ; Nagai, Noriyuki. / Erdheim-Chester disease in a child presenting with multiple jaw lesions. In: Journal of Oral Pathology and Medicine. 2005 ; Vol. 34, No. 7. pp. 420-422.
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AB - 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.

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