Loss of heterozygosity analysis at different chromosome regions in wilms tumor confirms 1p allelic loss as a marker of worse prognosis

A study from the Italian association of pediatric hematology and oncology

Filippo Spreafico, Beatrice Gamba, Luigi Mariani, Paola Collini, Paolo D'Angelo, Andrea Pession, Andrea Di Cataldo, Paolo Indolfi, Marilina Nantron, Monica Terenziani, Carlo Morosi, Paolo Radice, Daniela Perotti

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Abstract

Purpose: The specific aims of the AIEOP-TW-2003 protocol included prospectively investigating a possible association of tumor loss of heterozygosity with outcomes in children treated for Wilms tumor. Materials and Methods: We analyzed 125 unilateral favorable histology Wilms tumors registered between 2003 and 2008 in the Italian cooperative protocol for microsatellite markers mapped to chromosomes 1p, 7p, 11q, 16q and 22q. Results: The 3-year disease-free survival and overall survival probabilities were 0.87 (95% CI 0.81-0.93) and 0.98 (95% CI 0.96-1.0), respectively. Loss of heterozygosity at 1p was significantly associated with a worse disease-free survival (probability 0.67 for patients with and 0.92 for those without 1p loss of heterozygosity, p = 0.0009), as confirmed also by multivariate analysis adjusting for tumor stage and patient age at diagnosis. There was no difference in disease-free survival probability among children with loss of heterozygosity in the other chromosomal regions tested. The worse outlook for children older than 2 years at diagnosis did not seem to be influenced by the loss of heterozygosity patterns considered. Conclusions: Chromosome 1p loss of heterozygosity seems to be a risk factor for nonanaplastic Wilms tumor, possibly regardless of other clinical factors. Our findings were uninformative regarding loss of heterozygosity in the other chromosomal regions tested.

Original languageEnglish
Pages (from-to)260-266
Number of pages7
JournalJournal of Urology
Volume189
Issue number1
DOIs
Publication statusPublished - Jan 2013

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Wilms Tumor
Loss of Heterozygosity
Hematology
Chromosomes
Pediatrics
Disease-Free Survival
Microsatellite Repeats
Neoplasms
Histology
Multivariate Analysis
Survival

Keywords

  • kidney neoplasms
  • loss of heterozygosity
  • pediatrics
  • Wilms tumor

ASJC Scopus subject areas

  • Urology

Cite this

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title = "Loss of heterozygosity analysis at different chromosome regions in wilms tumor confirms 1p allelic loss as a marker of worse prognosis: A study from the Italian association of pediatric hematology and oncology",
abstract = "Purpose: The specific aims of the AIEOP-TW-2003 protocol included prospectively investigating a possible association of tumor loss of heterozygosity with outcomes in children treated for Wilms tumor. Materials and Methods: We analyzed 125 unilateral favorable histology Wilms tumors registered between 2003 and 2008 in the Italian cooperative protocol for microsatellite markers mapped to chromosomes 1p, 7p, 11q, 16q and 22q. Results: The 3-year disease-free survival and overall survival probabilities were 0.87 (95{\%} CI 0.81-0.93) and 0.98 (95{\%} CI 0.96-1.0), respectively. Loss of heterozygosity at 1p was significantly associated with a worse disease-free survival (probability 0.67 for patients with and 0.92 for those without 1p loss of heterozygosity, p = 0.0009), as confirmed also by multivariate analysis adjusting for tumor stage and patient age at diagnosis. There was no difference in disease-free survival probability among children with loss of heterozygosity in the other chromosomal regions tested. The worse outlook for children older than 2 years at diagnosis did not seem to be influenced by the loss of heterozygosity patterns considered. Conclusions: Chromosome 1p loss of heterozygosity seems to be a risk factor for nonanaplastic Wilms tumor, possibly regardless of other clinical factors. Our findings were uninformative regarding loss of heterozygosity in the other chromosomal regions tested.",
keywords = "kidney neoplasms, loss of heterozygosity, pediatrics, Wilms tumor",
author = "Filippo Spreafico and Beatrice Gamba and Luigi Mariani and Paola Collini and Paolo D'Angelo and Andrea Pession and {Di Cataldo}, Andrea and Paolo Indolfi and Marilina Nantron and Monica Terenziani and Carlo Morosi and Paolo Radice and Daniela Perotti",
year = "2013",
month = "1",
doi = "10.1016/j.juro.2012.09.009",
language = "English",
volume = "189",
pages = "260--266",
journal = "Journal of Urology",
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T1 - Loss of heterozygosity analysis at different chromosome regions in wilms tumor confirms 1p allelic loss as a marker of worse prognosis

T2 - A study from the Italian association of pediatric hematology and oncology

AU - Spreafico, Filippo

AU - Gamba, Beatrice

AU - Mariani, Luigi

AU - Collini, Paola

AU - D'Angelo, Paolo

AU - Pession, Andrea

AU - Di Cataldo, Andrea

AU - Indolfi, Paolo

AU - Nantron, Marilina

AU - Terenziani, Monica

AU - Morosi, Carlo

AU - Radice, Paolo

AU - Perotti, Daniela

PY - 2013/1

Y1 - 2013/1

N2 - Purpose: The specific aims of the AIEOP-TW-2003 protocol included prospectively investigating a possible association of tumor loss of heterozygosity with outcomes in children treated for Wilms tumor. Materials and Methods: We analyzed 125 unilateral favorable histology Wilms tumors registered between 2003 and 2008 in the Italian cooperative protocol for microsatellite markers mapped to chromosomes 1p, 7p, 11q, 16q and 22q. Results: The 3-year disease-free survival and overall survival probabilities were 0.87 (95% CI 0.81-0.93) and 0.98 (95% CI 0.96-1.0), respectively. Loss of heterozygosity at 1p was significantly associated with a worse disease-free survival (probability 0.67 for patients with and 0.92 for those without 1p loss of heterozygosity, p = 0.0009), as confirmed also by multivariate analysis adjusting for tumor stage and patient age at diagnosis. There was no difference in disease-free survival probability among children with loss of heterozygosity in the other chromosomal regions tested. The worse outlook for children older than 2 years at diagnosis did not seem to be influenced by the loss of heterozygosity patterns considered. Conclusions: Chromosome 1p loss of heterozygosity seems to be a risk factor for nonanaplastic Wilms tumor, possibly regardless of other clinical factors. Our findings were uninformative regarding loss of heterozygosity in the other chromosomal regions tested.

AB - Purpose: The specific aims of the AIEOP-TW-2003 protocol included prospectively investigating a possible association of tumor loss of heterozygosity with outcomes in children treated for Wilms tumor. Materials and Methods: We analyzed 125 unilateral favorable histology Wilms tumors registered between 2003 and 2008 in the Italian cooperative protocol for microsatellite markers mapped to chromosomes 1p, 7p, 11q, 16q and 22q. Results: The 3-year disease-free survival and overall survival probabilities were 0.87 (95% CI 0.81-0.93) and 0.98 (95% CI 0.96-1.0), respectively. Loss of heterozygosity at 1p was significantly associated with a worse disease-free survival (probability 0.67 for patients with and 0.92 for those without 1p loss of heterozygosity, p = 0.0009), as confirmed also by multivariate analysis adjusting for tumor stage and patient age at diagnosis. There was no difference in disease-free survival probability among children with loss of heterozygosity in the other chromosomal regions tested. The worse outlook for children older than 2 years at diagnosis did not seem to be influenced by the loss of heterozygosity patterns considered. Conclusions: Chromosome 1p loss of heterozygosity seems to be a risk factor for nonanaplastic Wilms tumor, possibly regardless of other clinical factors. Our findings were uninformative regarding loss of heterozygosity in the other chromosomal regions tested.

KW - kidney neoplasms

KW - loss of heterozygosity

KW - pediatrics

KW - Wilms tumor

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