Bisphosphonate treatment in polyostotic fibrous dysplasia of the cranium: case report and literature review.
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.
Mansoori
Lela S
LS
Department of Medicine, University of Colorado at Denver and Health Sciences Center, Aurora, Colorado 80045 , USA. lela.mansoori@ucdenver.edu
Catel
Colin P
CP
Rothman
Micol S
MS
D000328:Adult; D050071:Bone Density Conservation Agents / Q000008:administration & dosage / Q000627:therapeutic use; D004164:Diphosphonates / Q000008:administration & dosage / Q000627:therapeutic use*; D005147:Facial Bones / Q000187:drug effects / Q000473:pathology; D005359:Fibrous Dysplasia, Polyostotic / Q000188:drug therapy*; D006801:Humans; D007093:Imidazoles / Q000008:administration & dosage / Q000627:therapeutic use; D007275:Injections, Intravenous; D008297:Male; D012886:Skull / Q000187:drug effects / Q000473:pathology; D000077211:Zoledronic Acid
D050071:Bone Density Conservation Agents; D004164:Diphosphonates; D007093:Imidazoles; D000077211:Zoledronic Acid