TY - JOUR
T1 - Erdheim-Chester disease in a child presenting with multiple jaw lesions
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
N1 - Copyright:
Copyright 2008 Elsevier B.V., All rights reserved.
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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U2 - 10.1111/j.1600-0714.2005.00320.x
DO - 10.1111/j.1600-0714.2005.00320.x
M3 - Article
C2 - 16011611
AN - SCOPUS:22344450958
VL - 34
SP - 420
EP - 422
JO - Journal of Oral Pathology and Medicine
JF - Journal of Oral Pathology and Medicine
SN - 0904-2512
IS - 7
ER -
