de Ruiter Ruben Daniel, Botman Esmée, Teunissen Bernd P, Lammertsma Adriaan Anthonius, Boellaard Ronald, Raijmakers Pieter G, Schwarte Lothar A, Nieuwenhuijzen Jakko A, Gonzalez Trotter Dinko, Eekhoff Elisabeth Marelise W, Yaqub Maqsood
Department of Internal Medicine, Endocrinology Section, Amsterdam University Medical Centers, Vrije Universiteit Amsterdam, Amsterdam Movement Sciences, Amsterdam, Netherlands.
Department of Radiology and Nuclear Medicine, Amsterdam University Medical Centers, Vrije Universiteit Amsterdam, Amsterdam Movement Sciences, Amsterdam, Netherlands.
Front Nucl Med. 2024 Jul 22;4:1406947. doi: 10.3389/fnume.2024.1406947. eCollection 2024.
Fibrodysplasia Ossificans Progressiva (FOP) is a rare, genetic disease in which heterotopic bone is formed in muscles, tendons and ligaments throughout the body. Disease progression is variable over time and between individuals. F-fluoride uptake in newly formed bone can be evaluated using [F]NaF (i.e., sodiumfluoride) PET/CT, identifying active areas of bone formation in FOP. The purpose of this study was to assess the performance of various semi-quantitative methods with full kinetic analysis.
Seven patients (age range: 20-31 years) with FOP underwent dynamic [F]NaF scans at baseline and after one year. [F]NaF uptake was measured in aorta descendens, vertebrae, heterotopic bone lesions and metabolically active regions on PET, and quantified using nonlinear regression (NLR) analysis together with standardized uptake value (SUV) and target-to-blood ratio (TBR). SUV was on measured the 40-45 min frame of the dynamic sequence (SUV) and on the subsequent static sweep (SUV). Correlations between and SUV and NLR-derived were comparable when normalized to body weight ( = 0.81, 95% CI 0.64-0.90), lean body mass ( = 0.79, 95% CI 0.61-0.89) and body surface area ( = 0.84, 95% CI 0.70-0.92). Correlation between TBR and NLR-derived ( = 0.92, 95% CI 0.85-0.96) was higher than for SUV. Correlation between TBR and NLR-derived was similar at baseline and after one year ( = 0.93 and 0.94). The change in TBR between baseline measurement and after one year correlated best with the change in NLR-derived in the PET-active lesions ( = 0.87).
The present data supports the use of TBR for assessing fluoride uptake in PET-active lesions in FOP.
Sub-study of the Lumina-1 trial (clinicaltrials.gov, NCT03188666, registered 13-06-2017).
进行性骨化性纤维发育不良（FOP）是一种罕见的遗传性疾病，在全身的肌肉、肌腱和韧带中会形成异位骨。疾病进展随时间和个体而有所不同。新形成骨中的氟摄取情况可通过[F]NaF（即氟化钠）PET/CT进行评估，从而识别FOP中骨形成的活跃区域。本研究的目的是通过完整的动力学分析来评估各种半定量方法的性能。
7例FOP患者（年龄范围：20 - 31岁）在基线期和1年后接受了动态[F]NaF扫描。在PET上测量降主动脉、椎骨、异位骨病变和代谢活跃区域的[F]NaF摄取，并使用非线性回归（NLR）分析以及标准化摄取值（SUV）和靶本比（TBR）进行量化。SUV在动态序列的40 - 45分钟帧（SUV）以及随后的静态扫描（SUV）上进行测量。当以体重（r = 0.81，95%CI 0.64 - 0.90）、去脂体重（r = 0.79，95%CI 0.61 - 0.89）和体表面积（r = 0.84，95%CI 0.70 - 0.92）进行标准化时，SUV与NLR得出的结果之间的相关性具有可比性。TBR与NLR得出的结果之间的相关性（r = 0.92，95%CI 0.85 - 0.96）高于SUV。TBR与NLR得出的结果之间的相关性在基线期和1年后相似（r = 0.93和0.94）。基线测量和1年后TBR的变化与PET活跃病变中NLR得出的结果变化相关性最佳（r = 0.87）。
目前的数据支持使用TBR来评估FOP中PET活跃病变的氟摄取情况。
Lumina - 1试验的子研究（clinicaltrials.gov，NCT03188666，于2017年6月13日注册）。
