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Bisphosphonate treatment in polyostotic fibrous dysplasia of the cranium: case report and literature review.
MedLine Citation:
PMID:  20570808     Owner:  NLM     Status:  In-Data-Review    
Abstract/OtherAbstract:
Objective: To report the case of a patient with polyostotic fibrous dysplasia of the cranium who showed dramatic improvement after treatment with intravenous zoledronic acid.Methods: We present the clinical findings, laboratory test results, surgical pathology report, and imaging studies of a man with extensive fibrous dysplasia of the cranium and review the literature regarding the use of bisphosphonates in patients with this debilitating skeletal disorder.Results: A 32-year-old man presented with chronic occipital headache, and computed tomography of the head revealed extensive bony lesions of the middle and posterior cranial fossa. Bone biopsy confirmed the diagnosis of fibrous dysplasia. Laboratory blood test results revealed elevated serum alkaline phosphatase (140 U/L [reference range, 39-117 U/L]) and elevated serum bone-specific alkaline phosphatase (30.6 μg/L [reference range, 6-20 μg/L]). The patient was treated with intravenous pamidronate without improvement, and therapy was switched to zoledronic acid, which resulted in rapid resolution of headache symptoms, decrease in bone-specific alkaline phosphatase levels to 24.9 μg/L, and dramatic radiologic improvement on repeated computed tomography of the head.Conclusions: The treatment options for fibrous dysplasia of the cranium have been limited to conservative follow-up or surgery, and the use of zoledronic acid in this condition has not been previously reported. Intravenous bisphosphonate therapy is suggested to be a useful option in the treatment of cranial fibrous dysplasia.
Authors:
Lela S Mansoori; Colin P Catel; Micol S Rothman
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Endocrine practice : official journal of the American College of Endocrinology and the American Association of Clinical Endocrinologists     Volume:  16     ISSN:  1934-2403     ISO Abbreviation:  Endocr Pract     Publication Date:    2010 Sep-Oct
Date Detail:
Created Date:  2011-03-01     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9607439     Medline TA:  Endocr Pract     Country:  United States    
Other Details:
Languages:  eng     Pagination:  851-4     Citation Subset:  IM    
Affiliation:
Department of Medicine, University of Colorado at Denver and Health Sciences Center, Aurora, Colorado Division of Endocrinology, Metabolism, and Diabetes, University of Colorado at Denver and Health Sciences Center, Aurora, Colorado.
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