Abstract
BACKGROUND: Wilms tumors (WT) are the most common kidney tumors in children, with excellent survival rates (90%). However, late adverse effects warrant attention. Limited data exist on musculoskeletal sequelae in WT survivors. We aimed to assess the prevalence and determinants of impaired bone mineral density (BMD) and fractures in a national cohort of Dutch WT survivors. METHOD: This cross-sectional study includes WT survivors treated between 1963 and 2002, recruited as part of the DCCSS-LATER cohort between 2016 and 2020. Dual-energy X-ray absorptiometry (DXA) scans were used to assess BMD. Low BMD was defined as a Z-score ≤ 1. From 5 years after diagnosis, fracture prevalence was assessed by questionnaires. Univariable logistic regression was used to analyze associations between impaired BMD as well as fractures with independent variables like patient characteristics, treatments, comorbidities, and lifestyle-related factors. RESULTS: Of 437 invited kidney tumor survivors, 233 WT survivors participated (median age 32.1 years, median follow-up 27.8 years). DXA scans and fracture data were available for 173 and 221 WT survivors, respectively. Low BMD at any site was observed in 26% (n = 46/173) of survivors and was significantly associated with treatment including ≥ 4 drugs (OR 2.76; 95% CI = 1.13-6.70). Abdominal radiotherapy doses > 30 Gy (OR 4.84; 95% CI = 1.06-22.2) were significantly associated with low lumbar spine BMD. The prevalence of fragility fractures was 16.3% (n = 36/221). The standardized incidence ratio (SIR) of any first fracture was 2.34 for males and 5.38 for females. CONCLUSION: Wilms tumor survivors treated with ≥ 4 drugs or abdominal radiotherapy (> 30 Gy) seem to be at increased risk of impaired BMD; this could indicate the need for surveillance for this subset of Wilms tumor survivors exposed to these treatment regimens in the past.