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

doi:10.1111/j.1600-0714.2005.00320.x

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 journal › Article

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 language	English
Pages (from-to)	420-422
Number of pages	3
Journal	Journal of Oral Pathology and Medicine
Volume	34
Issue number	7
DOIs	https://doi.org/10.1111/j.1600-0714.2005.00320.x
Publication status	Published - Aug 2005

Keywords

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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Nagatsuka, H., Han, P. P., Taguchi, K., Tsujigiwa, H., Gunduz, M., Fukunaga, J., Sugahara, T., Asaumi, J., & Nagai, N. (2005). Erdheim-Chester disease in a child presenting with multiple jaw lesions. Journal of Oral Pathology and Medicine, 34(7), 420-422. https://doi.org/10.1111/j.1600-0714.2005.00320.x

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title = "Erdheim-Chester disease in a child presenting with multiple jaw lesions",

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.",

keywords = "Bilateral symmetrical bony lesions, Erdheim-Chester disease, Jaws, Lipid-storing histiocytes, Touton-type giant cells",

author = "Hitoshi Nagatsuka and Han, {Phuu Pwint} and Koji Taguchi and Hidetsugu Tsujigiwa and Mehmet Gunduz and Joji Fukunaga and Toshio Sugahara and Junichi Asaumi and Noriyuki Nagai",

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AU - Nagatsuka, Hitoshi

AU - Han, Phuu Pwint

AU - Taguchi, Koji

AU - Tsujigiwa, Hidetsugu

AU - Gunduz, Mehmet

AU - Fukunaga, Joji

AU - Sugahara, Toshio

AU - Asaumi, Junichi

AU - Nagai, Noriyuki

PY - 2005/8

Y1 - 2005/8

N2 - 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.

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.

KW - Bilateral symmetrical bony lesions

KW - Erdheim-Chester disease

KW - Jaws

KW - Lipid-storing histiocytes

KW - Touton-type giant cells

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