Abstract
Fibrous dysplasia (FD) is a mosaic skeletal disorder resulting in fractures, deformity, and functional impairment. Clinical evaluation has been limited by a lack of surrogate endpoints capable of quantitating disease activity. The purpose of this study was to investigate the utility of 18F-NaF PET/CT imaging in quantifying disease activity in patients with FD. Fifteen consecutively evaluated subjects underwent whole-body 18F-NaF PET/CT scans, and FD burden was assessed by quantifying FD-related 18F-NaF activity. 18F-NaF PET/CT parameters obtained included (i) SUVmax (standardized uptake value [SUV] of the FD lesion with the highest uptake); (ii) SUVmean (average SUV of all 18F-NaF–positive FD lesions); (iii) total volume of all 18F-NaF–positive FD lesions (TV); and (iv) total FD lesion activity determined as the product of TV multiplied by SUVmean (TA = TV × SUVmean) (TA). Skeletal outcomes, functional outcomes, and bone turnover markers were correlated with 18F-NaF PET/CT parameters. TV and TA of extracranial FD lesions correlated strongly with skeletal outcomes including fractures and surgeries (p values ≤ 0.003). Subjects with impaired ambulation and scoliosis had significantly higher TV and TA values (P < 0.05), obtained from extracranial and spinal lesions, respectively. Craniofacial surgeries correlated with TV and TA of skull FD lesions (P < 0.001). Bone turnover markers, including alkaline phosphatase, N-telopeptides, and osteocalcin, were strongly correlated with TV and TA (P < 0.05) extracted from FD lesions in the entire skeleton. No associations were identified with SUVmax or SUVmean. Bone pain and age did not correlate with 18F-NaF PET/CT parameters. FD burden evaluated by 18F-NaF-PET/CT facilitates accurate assessment of FD activity, and correlates quantitatively with clinically-relevant skeletal outcomes.
Original language | English (US) |
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Pages (from-to) | 1619-1631 |
Number of pages | 13 |
Journal | Journal of Bone and Mineral Research |
Volume | 34 |
Issue number | 9 |
DOIs | |
State | Published - Sep 1 2019 |
Funding
This study was funded by the Intramural Research Program of the National Institute of Dental and Craniofacial Research, National Institutes of Health. Authors' roles: Study design: GZP, MTC, and AMB. Study conduct: GZP, MTC, and AMB. Data collection: GZP and AMB. Data analysis: GZP, GCM, and AMB. Data interpretation: GZP, GCM, PF, AHK, UB, KM, and AMB. Drafting manuscript: GZP, GCM, and AMB. Revising manuscript content: GZP, GCM, and AMB. Approving final version of manuscript: GZP, GCM, PF, AHK, UB, KM, MTC, and AMB. AMB takes responsibility for the integrity of the data analysis.
Keywords
- ANALYSIS/QUANTITATION OF BONE (OTHER)
- BIOCHEMICAL MARKERS OF BONE TURNOVER
- DISEASES AND DISORDERS OF/RELATED TO BONE (OTHER)
ASJC Scopus subject areas
- Endocrinology, Diabetes and Metabolism
- Orthopedics and Sports Medicine