A case of osteoarthropathy due to Erdheim-Chester disease with overlapping Langerhans' cell infiltration.
Histiocytosis sometimes involves the joints, and is one of the important differential diagnoses of osteoarthropathy. A 31-year-old man presented with recurrent fever and bilateral knee arthritis two years prior to admission. He also showed the hypopituitary mass lesion and partial hypopituitarism. X-ray studies showed both osteosclerotic and osteolytic lesions near the large joints. Histological findings of bone biopsy revealed foamy macrophage infiltration, which were CD68+CD1a-S100-, and Erdheim-Chester disease was diagnosed. In addition, CD68+CD1a+ Langerhans' cells also aggregated in the same lesions, and we thought this case was a rare variant of Erdheim-Chester disease with overlapping histiocytic invasion.
Naruse
Hiroya
H
Department of Allergy and Rheumatology, Graduate School of Medicine, the University of Tokyo, Tokyo, Japan.
Shoda
Hirofumi
H
Okamoto
Akiko
A
Oka
Teruaki
T
Yamamoto
Kazuhiko
K
D000328:Adult; D031249:Erdheim-Chester Disease / Q000150:complications / Q000175:diagnosis*; D006646:Histiocytosis, Langerhans-Cell / Q000150:complications / Q000175:diagnosis*; D006801:Humans; D007801:Langerhans Cells / Q000473:pathology; D008297:Male; D010005:Osteoarthropathy, Secondary Hypertrophic / Q000150:complications / Q000175:diagnosis*