荷兰首个全国性威尔姆斯瘤幸存者队列中骨矿物质密度受损和骨折的患病率及决定因素，一项全国性DCCSS-LATER研究
Prevalence and Determinants of Impaired Bone Mineral Density and Fractures in the First National Dutch Wilms Tumor Survivor Cohort, a National DCCSS-LATER Study.
作者信息
Wens Francis S P L, de Winter Demi T C, Janssens Geert O, Litjens Rens, van Atteveld Jenneke E, Nievelstein Rutger A J, Hobbelink Monique G G, de Vries Andrica C H, Loonen Jacqueline J, van Dulmen-den Broeder Eline, van der Pal Helena J H, Pluijm Saskia M F, Kremer Leontien C M, van der Heiden-van der Loo Margriet, Louwerens Marloes, van Santen Hanneke M, Olsson Daniel S, Hoefer Imo, van den Berg Sjoerd A A, van Tinteren Harm, Neggers Sebastian J C M M, van den Heuvel-Eibrink Marry M
机构信息
Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands.
University Medical Center Utrecht, Utrecht, the Netherlands.
出版信息
Cancer Med. 2025 Sep;14(18):e71229. doi: 10.1002/cam4.71229.
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.
背景
肾母细胞瘤（WT）是儿童最常见的肾脏肿瘤，生存率很高（90%）。然而，晚期不良反应值得关注。关于WT幸存者肌肉骨骼后遗症的数据有限。我们旨在评估荷兰全国WT幸存者队列中骨密度（BMD）受损和骨折的患病率及决定因素。
方法
这项横断面研究纳入了1963年至2002年期间接受治疗的WT幸存者，他们于2016年至2020年作为DCCSS-LATER队列的一部分被招募。采用双能X线吸收法（DXA）扫描评估骨密度。低骨密度定义为Z评分≤-1。从诊断后5年起，通过问卷调查评估骨折患病率。采用单变量逻辑回归分析骨密度受损以及骨折与患者特征、治疗、合并症和生活方式相关因素等自变量之间的关联。
结果
在437名受邀的肾脏肿瘤幸存者中，233名WT幸存者参与（中位年龄32.1岁，中位随访27.8年）。分别有173名和221名WT幸存者可获得DXA扫描和骨折数据。26%（n = 46/173）的幸存者在任何部位出现低骨密度，且与包括≥4种药物的治疗显著相关（比值比2.76；95%置信区间 = 1.13 - 6.70）。腹部放疗剂量>30 Gy（比值比4.84；95%置信区间 = 1.06 - 22.2）与腰椎低骨密度显著相关。脆性骨折的患病率为16.3%（n = 36/221）。男性任何首次骨折的标准化发病比（SIR）为2.34，女性为5.38。
结论
接受≥4种药物或腹部放疗（>30 Gy）治疗的WT幸存者似乎骨密度受损风险增加；这可能表明有必要对过去接受这些治疗方案的这部分WT幸存者进行监测。
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