A cross-sectional and longitudinal analysis of trabecular bone score in adults with turner syndrome

Abstract

Context: Turner syndrome (TS) is associated with short stature, gonadal failure, and fractures. Spinal trabecular bone score (TBS) is a novel bone imaging modality that has not been evaluated in TS. Objective: To evaluate TBS in TS and its association with bone mineral density (BMD), prevalent fracture, and risk factors. Design and Setting: Longitudinal study of TS from a single tertiary hospital between 2006 and 2017. Patients or Other Participants: Fifty-eight subjects with TS aged 20 to 49 years who underwent dualenergy X-ray absorptiometry (DXA). Main Outcome Measures: TBS, DXA parameters, and prevalent fractures were investigated. Results: Normal, partially degraded, and degraded TBSs were observed in 39 (67%), 15 (26%), and four (7%) subjects, respectively. High rates of prescribed estrogen replacement therapy (ERT) with stable TBS and BMD were observed during follow-up. TBS was positively correlated with spine and femoral neck (FN) BMD and Z-scores (all P<0.05) and negatively correlated with age (20.004 per year; P = 0.014) and delay in ERT initiation inwomenwith primary amenorrhea (20.010 per year; P<0.001). Fractureswere present in 17 (31%) subjects. LowTBS had a significantly higher area under the receiver operator curve for predicting prevalent fracture than low bone mass at either the spine or FN (P < 0.05). Subjects with no history of fracture were more likely to have a normal TBS (P = 0.023). Conclusions: BMD and TBS can be preserved with early initiation and continued use of ERT. TBS may provide additional fracture risk prediction to standard DXA parameters in TS and needs to be validated in larger prospective studies.