Prevalence and Determinants of Impaired Bone Mineral Density and Fractures in the First National Dutch Wilms Tumor Survivor Cohort, a National DCCSS-LATER Study

IF 3.1 2区 医学 Q2 ONCOLOGY
Cancer Medicine Pub Date : 2025-09-16 DOI:10.1002/cam4.71229
Francis S. P. L. Wens, Demi T. C. de Winter, Geert O. Janssens, Rens Litjens, Jenneke E. van Atteveld, Rutger A. J. Nievelstein, Monique G. G. Hobbelink, Andrica C. H. de Vries, Jacqueline J. Loonen, Eline van Dulmen-den Broeder, Helena J. H. van der Pal, Saskia M. F. Pluijm, Leontien C. M. Kremer, Margriet van der Heiden-van der Loo, Marloes Louwerens, Hanneke M. van Santen, Daniel S. Olsson, Imo Hoefer, Sjoerd A. A. van den Berg, Harm van Tinteren, Sebastian J. C. M. M. Neggers, Marry M. van den Heuvel-Eibrink
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引用次数: 0

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.

Abstract Image

一项国家DCCSS-LATER研究:荷兰首例Wilms肿瘤幸存者队列中骨密度受损和骨折的患病率和决定因素。
背景:肾母细胞瘤(Wilms tumor, WT)是儿童最常见的肾脏肿瘤,生存率极高(90%)。然而,后期的不良反应值得注意。关于WT幸存者肌肉骨骼后遗症的资料有限。我们的目的是评估荷兰WT幸存者的国家队列中骨密度受损和骨折的患病率和决定因素。方法:本横断面研究包括1963年至2002年期间治疗的WT幸存者,作为2016年至2020年期间DCCSS-LATER队列的一部分招募。采用双能x线吸收仪(DXA)扫描评估骨密度。低BMD定义为Z-score≤1。从诊断后5年开始,通过问卷调查评估骨折患病率。采用单变量logistic回归分析骨密度受损和骨折与患者特征、治疗、合并症和生活方式相关因素等自变量之间的关系。结果:在437名受邀的肾肿瘤幸存者中,233名WT幸存者参与了研究(中位年龄32.1岁,中位随访时间27.8年)。分别有173例和221例WT幸存者获得DXA扫描和骨折数据。26% (n = 46/173)的幸存者在任何部位均出现低骨密度,并且与包括≥4种药物的治疗显著相关(OR 2.76; 95% CI = 1.13-6.70)。腹部放疗剂量> ~ 30gy (OR 4.84; 95% CI = 1.06 ~ 22.2)与低腰椎骨密度显著相关。脆性骨折发生率为16.3% (n = 36/221)。男性首次骨折的标准化发生率为2.34,女性为5.38。结论:接受≥4种药物治疗或腹部放射治疗(bbb30 Gy)的Wilms肿瘤幸存者BMD受损的风险增加;这可能表明需要对过去暴露于这些治疗方案的Wilms肿瘤幸存者进行监测。
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来源期刊
Cancer Medicine
Cancer Medicine ONCOLOGY-
CiteScore
5.50
自引率
2.50%
发文量
907
审稿时长
19 weeks
期刊介绍: Cancer Medicine is a peer-reviewed, open access, interdisciplinary journal providing rapid publication of research from global biomedical researchers across the cancer sciences. The journal will consider submissions from all oncologic specialties, including, but not limited to, the following areas: Clinical Cancer Research Translational research ∙ clinical trials ∙ chemotherapy ∙ radiation therapy ∙ surgical therapy ∙ clinical observations ∙ clinical guidelines ∙ genetic consultation ∙ ethical considerations Cancer Biology: Molecular biology ∙ cellular biology ∙ molecular genetics ∙ genomics ∙ immunology ∙ epigenetics ∙ metabolic studies ∙ proteomics ∙ cytopathology ∙ carcinogenesis ∙ drug discovery and delivery. Cancer Prevention: Behavioral science ∙ psychosocial studies ∙ screening ∙ nutrition ∙ epidemiology and prevention ∙ community outreach. Bioinformatics: Gene expressions profiles ∙ gene regulation networks ∙ genome bioinformatics ∙ pathwayanalysis ∙ prognostic biomarkers. Cancer Medicine publishes original research articles, systematic reviews, meta-analyses, and research methods papers, along with invited editorials and commentaries. Original research papers must report well-conducted research with conclusions supported by the data presented in the paper.
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