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Title: Bisphosphonate therapy for refractory langerhans cell histiocytosis: A case report
Authors: Chayamon Takpradit
Nassawee Vathana
Nattee Narkbunnam
Kleebsabai Sanpakit
Jassada Buaboonnam
Mahidol University
Keywords: Medicine
Issue Date: 1-Nov-2015
Citation: Journal of the Medical Association of Thailand. Vol.98, No.11 (2015), 1145-1149
Abstract: © 2015, Medical Association of Thailand. All rights reserved. Although patients diagnosed as Langerhans cell histiocytosis (LCH) with bone lesion initially respond well to treatment, some may experience relapse or refractory disease. Pamidronate, a potent N-bisphosphonate, has been used in several primary bone diseases, benign bone tumors, and metastatic bone cancers. The mechanism includes an inhibitory effect on osteoclast activity by decreasing development and recruitment of osteoclast progenitors and promoting osteoclast apoptosis. Herein, we introduce a seven-month-old Thai girl who was diagnosed as multiple-relapse LCH with refractory bone lesions and was treated with standard and salvage steroid-based therapies. After receiving two courses of intravenous pamidronate, she had marked clinical and radiographical improvement without any adverse events. She has been in remission for two years after receiving six courses of therapy. This report supports the efficacy of pamidronate in LCH-related bone lesions, but further studies in large cohort are warranted.
ISSN: 01252208
Appears in Collections:Scopus 2011-2015

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