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Treatment of Langerhans cell histiocytosis bone lesions with zoledronic acid: a case series.
MedLine Citation:
PMID:  21519845     Owner:  NLM     Status:  Publisher    
Abstract/OtherAbstract:
Langerhans cell histiocytosis (LCH) is a rare disease caused by a clonal proliferation of specialized dendritic (Langerhans) cells. Although uncommon, it is potentially fatal and carries significant morbidity. Bone involvement is particularly destructive and to date, no standard of care exists for management of both the disease and the significant bone pain as many of these patients experience. In the literature, 12 patients who had previously been heavily pretreated for their disease had their bone pain treated with a bisphosphonate as extrapolated from the cancer literature. Interestingly, these patients had a complete or near complete resolution of their pain, return of functional status and in 75% of cases radiographic evidence of reduction or regression of disease. Only 6 of these patients were treated with a newer generation bisphosphonate, zoledronic acid. In this paper, we report a case series of 2 patients with LCH bone involvement who received 4 mg of intravenous zoledronic acid monthly for 1 year with complete resolution in their bone pain. In addition, both patients demonstrated reduction in tumor burden after bisphosphonate treatment. Uniquely, our first case is the only reported case in the literature using a bisphosphonate as first line therapy in the treatment of LCH. This case demonstrates the potential role of zoledronic acid therapy in the first line setting for disease stabilization and symptomatic control in patients unable to receive conventional therapy.
Authors:
Shanthi Sivendran; Harold Harvey; Allan Lipton; Joseph Drabick
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Publication Detail:
Type:  JOURNAL ARTICLE     Date:  2011-4-26
Journal Detail:
Title:  International journal of hematology     Volume:  -     ISSN:  1865-3774     ISO Abbreviation:  -     Publication Date:  2011 Apr 
Date Detail:
Created Date:  2011-4-26     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9111627     Medline TA:  Int J Hematol     Country:  -    
Other Details:
Languages:  ENG     Pagination:  -     Citation Subset:  -    
Affiliation:
Penn State Milton S. Hershey Medical Center, The Department of Medicine, Division of Hematology-Oncology, H046, 500 University Drive, PO Box 850, Hershey, PA, 17033-0850, USA, shanthi.sivendran@mssm.edu.
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