Prognostic markers in nephroblastoma (Wilms’ tumor)

Ghanem, Mazen A.; Steenbrugge, G.J. van; Nijman, Rien J.M.; Kwast, Theodorus H. van der

doi:10.1016/j.urology.2004.12.005

Cited by 26 publications

(23 citation statements)

References 36 publications

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“…Therefore, it could be questioned whether all bilateral Wilms tumors in nonsyndromic patients should be screened on germline mutations of WT1, as we did in our patient. Our patient revealed a germline splice site mutation in [2,7,24,26]. However, Schumacher et al [26] found that an extensive rhabdomyomatous diVerentiation and the presence of WT1 mutations correlate with a tumor subtype that responds poorly to chemotherapy.…”

Section: Discussionmentioning

confidence: 64%

“…Future studies in larger cohorts of patients are necessary to reveal whether the novel WT1 gene mutation in our patient is associated with histological subtype and response. More information is available on the association of somatic WT1 gene mutations, which are found in approximately in 10% of all WT cases, and response, although conXicting results have been published [7,18,33]. Loss of heterozygosity (LOH) analysis revealed correlations between allelic loss or gene ampliWcation and outcome in Wilms tumor patients [8,12,16,31,32].…”

Section: Discussionmentioning

confidence: 99%

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Topotecan distribution in an anephric infant with therapy resistant bilateral Wilms tumor with a novel germline WT1 gene mutation

Lugtenberg

Cransberg

Loos

et al. 2008

Cancer Chemother Pharmacol

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The therapeutic strategy for bilateral Wilms tumor (WT) remains a challenge. Especially in cases with chemotherapy resistant disease, bilateral nephrectomy is sometimes inevitable. For optimal cure rates stage V WT patients beneWt from adjuvant treatment; however, there are limited data available on chemotherapy pharmacokinetics in anephric children. In this report, we describe a 10-month old girl with bilateral Wilms tumor and a novel germline WT1 gene mutation. This patient hardly showed any response on preoperative chemotherapy, and ultimately, underwent sequential bilateral tumor-nephrectomy. Subsequently, during peritoneal dialysis, she received topotecan as adjuvant chemotherapy based on plasma levels, indicating that this is a reasonable option as adjuvant treatment in therapy-resistant Wilms tumor patients after bilateral nephrectomy. This case showed a novel germline WT1 gene mutation of which the correlation with resistant phenotype has to be conWrmed in larger cohorts of WT patients.

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Section: Discussionmentioning

confidence: 64%

Section: Discussionmentioning

confidence: 99%

Topotecan distribution in an anephric infant with therapy resistant bilateral Wilms tumor with a novel germline WT1 gene mutation

Lugtenberg

Cransberg

Loos

et al. 2008

Cancer Chemother Pharmacol

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“…The associations of various growth factors, cell adhesion, and extracellular matrix proteins are valuable prognostic markers in WT [132]. In addition to these recognized factors, angiogenetic regulation plays an important role in the formation of new blood vessels involved in the growth and metastatic spread of solid tumors, but the clinical significance of using serum levels of angiogenic growth factors in WT remains to be understood.…”

Section: Serum Biomarkers In Thoracic and Abdominal Tumorsmentioning

confidence: 99%

Clinical Significance of Serum Biomarkers in Pediatric Solid Mediastinal and Abdominal Tumors

Sandoval

Malkas

Hickey

2012

IJMS

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Childhood cancer is the leading cause of death by disease among U.S. children between infancy and age 15. Despite successes in treating solid tumors such as Wilms tumor, disappointments in the outcomes of high-risk solid tumors like neuroblastoma have precipitated efforts towards the early and accurate detection of these malignancies. This review summarizes available solid tumor serum biomarkers with a special focus on mediastinal and abdominal cancers in children.

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“…9 There have been no recent systematic reviews assessing the effectiveness of WT biomarkers at predicting prognostic factors such as relapse and mortality. The objectives of this review were to examine the accumulated literature on the use of biomarkers among children with WT, to quantify the prognostic value of biomarkers that have been adequately tested in appropriately designed studies, and to provide a useful summary for the practicing urologist.…”

Section: Introductionmentioning

confidence: 99%

Biomarkers for Wilms Tumor: A Systematic Review

et al. 2016

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Purpose Wilms tumor is the most common childhood renal malignancy and the fourth most common childhood cancer. Many biomarkers have been studied but there has been no comprehensive summary. We systematically reviewed the literature on biomarkers in Wilms Tumor with the objective of quantifying the prognostic implication of the presence of individual tumor markers. Methods We searched for English language studies from 1980–2015 performed on children with Wilms Tumor under 18 years old with prognostic data. The protocol was conducted as per PRISMA guidelines. Two reviewers abstracted data in duplicate using a standard evaluation form. We performed descriptive statistics, then calculated relative risks and 95% confidence intervals for markers appearing in multiple level 2 or 3 studies. Results 40 studies were included examining 32 biomarkers in 7381 Wilms patients. Studies had a median of 61 patients with 24 biomarker positive patients per study, and a median follow-up of 68.4 months. Median percent of patients in Stage 1, 2, 3, 4, and 5 were 28.5%, 26.4%, 24.5%, 14.1%, and 1.7%, with 10.2% anaplasia. The strongest negative prognostic association was loss of heterozygosity on 11p15, with a risk of recurrence of 5.00, although loss of heterozygosity on 1p and gain of function on 1q were also strongly linked to increased recurrence (2.93 and 2.86 respectively). Conclusions Several tumor markers are associated with an increased risk of recurrence or a decreased risk of overall survival in Wilms Tumor. These data suggest targets for development of diagnostic tests and potential therapies.

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Prognostic markers in nephroblastoma (Wilms’ tumor)

Cited by 26 publications

References 36 publications

Topotecan distribution in an anephric infant with therapy resistant bilateral Wilms tumor with a novel germline WT1 gene mutation

Topotecan distribution in an anephric infant with therapy resistant bilateral Wilms tumor with a novel germline WT1 gene mutation

Clinical Significance of Serum Biomarkers in Pediatric Solid Mediastinal and Abdominal Tumors

Biomarkers for Wilms Tumor: A Systematic Review

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