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
{"title":"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。","authors":"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","doi":"10.1002/cam4.71229","DOIUrl":null,"url":null,"abstract":"<div>\n \n \n <section>\n \n <h3> Background</h3>\n \n <p>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.</p>\n </section>\n \n <section>\n \n <h3> Method</h3>\n \n <p>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 <i>Z</i>-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.</p>\n </section>\n \n <section>\n \n <h3> Results</h3>\n \n <p>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% (<i>n</i> = 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% (<i>n</i> = 36/221). The standardized incidence ratio (SIR) of any first fracture was 2.34 for males and 5.38 for females.</p>\n </section>\n \n <section>\n \n <h3> Conclusion</h3>\n \n <p>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.</p>\n </section>\n </div>","PeriodicalId":139,"journal":{"name":"Cancer Medicine","volume":"14 18","pages":""},"PeriodicalIF":3.1000,"publicationDate":"2025-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12438959/pdf/","citationCount":"0","resultStr":"{\"title\":\"Prevalence and Determinants of Impaired Bone Mineral Density and Fractures in the First National Dutch Wilms Tumor Survivor Cohort, a National DCCSS-LATER Study\",\"authors\":\"Francis S. P. L. 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We aimed to assess the prevalence and determinants of impaired bone mineral density (BMD) and fractures in a national cohort of Dutch WT survivors.</p>\\n </section>\\n \\n <section>\\n \\n <h3> Method</h3>\\n \\n <p>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 <i>Z</i>-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.</p>\\n </section>\\n \\n <section>\\n \\n <h3> Results</h3>\\n \\n <p>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% (<i>n</i> = 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% (<i>n</i> = 36/221). The standardized incidence ratio (SIR) of any first fracture was 2.34 for males and 5.38 for females.</p>\\n </section>\\n \\n <section>\\n \\n <h3> Conclusion</h3>\\n \\n <p>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.</p>\\n </section>\\n </div>\",\"PeriodicalId\":139,\"journal\":{\"name\":\"Cancer Medicine\",\"volume\":\"14 18\",\"pages\":\"\"},\"PeriodicalIF\":3.1000,\"publicationDate\":\"2025-09-16\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12438959/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Cancer Medicine\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://onlinelibrary.wiley.com/doi/10.1002/cam4.71229\",\"RegionNum\":2,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"ONCOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Cancer Medicine","FirstCategoryId":"3","ListUrlMain":"https://onlinelibrary.wiley.com/doi/10.1002/cam4.71229","RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"ONCOLOGY","Score":null,"Total":0}
Prevalence and Determinants of Impaired Bone Mineral Density and Fractures in the First National Dutch Wilms Tumor Survivor Cohort, a National DCCSS-LATER Study
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.
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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