Successful treatment of multicentric reticulohistiocytosis with alendronate: evidence for a direct effect of bisphosphonate on histiocytes.
We describe the case of a 44-year-old Japanese woman with severe nodular erythematous skin lesions and arthritis mutilans who was admitted for further treatment of multicentric reticulohistiocytosis. Skin and synovial biopsies showed heavy infiltration with tartrate-resistant acid phosphatase-positive histiocytes and multinucleated giant cells. Immunohistochemical analysis showed that some of the mononuclear cells in the skin were positive for RANKL. After 1 month of Alendronate, an aminobisphosphonate, given at a dosage of 10 mg once a week intravenously for the first 6 weeks and then once a month thereafter, the arthritis and skin nodules improved, and the remission has continued for more than 2 years. The findings in this patient suggest that osteoclast-like multinucleated giant cells differentiate locally in the skin from infiltrating histiocytes with the help of RANKL-positive stromal cells and that alendronate acts directly on cells of monocyte/macrophage lineage in humans. Thus, alendronate should be added to the list of drugs for the treatment of multicentric reticulohistiocytosis.
Goto
Hitoshi
H
Osaka City University, Graduate School of Medicine, Osaka, Japan. goto@msic.med.osaka-cu.ac.jp
Inaba
Masaaki
M
Kobayashi
Keisuke
K
Imanishi
Yasuo
Y
Kumeda
Yasuro
Y
Inui
Kentaro
K
Okada
Fumihiko
F
Nishizawa
Yoshiki
Y
D000328:Adult; D019386:Alendronate / Q000627:therapeutic use*; D001168:Arthritis / Q000188:drug therapy / Q000473:pathology; D005260:Female; D006644:Histiocytes / Q000187:drug effects* / Q000473:pathology; D015616:Histiocytosis, Non-Langerhans-Cell / Q000188:drug therapy* / Q000473:pathology*; D006801:Humans; D012871:Skin Diseases / Q000188:drug therapy / Q000473:pathology
D019386:Alendronate