Successful treatment of multicentric reticulohistiocytosis with alendronate: Evidence for a direct effect of bisphosphonate on histiocytes

✍ Scribed by Hitoshi Goto; Masaaki Inaba; Keisuke Kobayashi; Yasuo Imanishi; Yasuro Kumeda; Kentaro Inui; Fumihiko Okada; Yoshiki Nishizawa

• Publisher: John Wiley and Sons
• Year: 2003
• Tongue: English
• Weight: 175 KB
• Volume: 48
• Category: Article
• ISSN: 0004-3591
• DOI: 10.1002/art.11362

✦ Synopsis

Abstract

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.