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NCI CANCERLIT^® Search: Wilms' Tumor - August 2002

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National Cancer Institute^®
Ultima Vez Modificado: 1 de agosto del 2002

Multicentre analysis of anthracycline-induced cardiotoxicity in children following treatment according to the nephroblastoma studies SIOP
Aseptic osteonecrosis in a child with nephroblastoma healed by hyperbaric oxygen therapy.
Genetic changes of two Wilms tumors with anaplasia and a review of the literature suggesting a marker profile for therapy resistance.
Hereditary cleft lip/palate and Wilms tumor: a rare association.
A 3-year old boy with recurrent hematuria.
Expression and prognostic value Of CD44 isoforms in nephroblastoma (Wilms tumor).
Severe intrarenal fibrosis, infundibular stenosis, renal cysts, and persistent perilobar nephrogenic rests in a patient with

Table of Contents

1
UI - 12116074
AU - Marx M; Langer T; Graf N; Hausdorf G; Stohr W; Ludwig R; Beck JD
TI - Multicentre analysis of anthracycline-induced cardiotoxicity in children following treatment according to the nephroblastoma studies SIOP No.9/GPOH and SIOP 93-01/GPOH.
SO - Med Pediatr Oncol 2002 Jul;39(1):18-24
AD - Department of Immunology and Oncology, University Hospital for Children and Adolescents Erlangen-Nuremberg, Germany.
BACKGROUND: To study cardiac function and the incidence of anthracycline-induced cardiotoxicity in children following treatment according to the nephroblastoma studies SIOP No.9/GPOH and SIOP 93-01/GPOH. PROCEDURE: Analysis of clinical status, echocardiography, and ECG findings prior to administration of anthracyclines (median cumulative doxorubicin dose: 250 mg/m(2) [range: 90-411 mg/m(2)] and after a median posttherapeutic interval of 2.9 years [range: 0-10.2 years]. Data on cardiac function before and/or after therapy could be obtained of 186 patients. RESULTS: Posttherapy left ventricular fractional shortening was reduced in 4/157 (2.5%) patients. Out of the 4 children, 2 had clinically reduced tolerance to exercise and received anticongestive therapy. Abnormal ECG findings that were not detectable prior to therapy were found in 7/124 (5.6%) children. CONCLUSIONS: The incidence of abnormal findings is low in our study group in comparison to data from the literature and might be due to the comparably short posttherapeutic interval. Copyright 2002 Wiley-Liss, Inc.

2
UI - 12116080
AU - Bernbeck B; Krauth KA; Scherer A; Engelbrecht V; Gobel U
TI - Aseptic osteonecrosis in a child with nephroblastoma healed by hyperbaric oxygen therapy.
SO - Med Pediatr Oncol 2002 Jul;39(1):47-8
AD - Department of Pediatric Hematology and Oncology, Heinrich-Heine-University, Medical Center, Moorenstr. 5, 40225 Dusseldorf, Germany. bernbeck@med.uni-duesseldorf.de

3
UI - 12127397
AU - Stock C; Ambros IM; Lion T; Zoubek A; Amann G; Gadner H; Ambros PF
TI - Genetic changes of two Wilms tumors with anaplasia and a review of the literature suggesting a marker profile for therapy resistance.
SO - Cancer Genet Cytogenet 2002 Jun;135(2):128-38
AD - Children's Cancer Research Institute (CCRI), St. Anna Children's Hospital, Kinderspitalgasse 6, A-1090 Vienna, Austria. Stock@CCRI.univie.ac.at
Cytogenetic data on Wilms tumors (WT) with anaplasia frequently associated with an unfavorable outcome are scarce. We present cytogenetic changes of two WT with anaplasia (primary tumor material) from nonresponders with a synopsis of the literature. The WT were investigated by cytogenetic analysis, comparative genomic hybridization, fluorescence in situ hybridization, immunofluorescence, and flow cytometric analyses. Both tumors exhibited characteristic genetic changes. One tumor was hypodiploid due to loss of entire chromosome 11; losses of 16p, 16q, 17p, chromosome 19 material, and loss of 22q12-qter. The other tumor was hyperdiploid and triploid, and displayed gain of 1q12-q23 and chromosome 9 material. Moreover, two morphological and genetically distinct cell lines have been established from both tumors, demonstrating underrepresentation of chromosomes 13, 14, 16, and 19. Karyotype descriptions of 120 WT with known clinical data together with data of this report confirm: (1) inter- and intratumor heterogeneity exists; (2) loss or underrepresentation of chromosome material at 11, 13, 14, 16, 17p, 19, and 22q in various combinations presents a new marker profile of resistance to cytotoxic agents regardless of the histological types; and (3) the prognostic impact of gain at 1q12-q23 sequences warrants further validation.

4
UI - 12019017
AU - Yu CC; Wong FH; Lo LJ; Chen YR
TI - Hereditary cleft lip/palate and Wilms tumor: a rare association.
SO - Cleft Palate Craniofac J 2002 May;39(3):376-9
AD - Craniofacial Center, Department of Plastic and Reconstructive Surgery, Chang Gung Memorial Hospital, Taipei, Taiwan.
OBJECTIVE: The association of cleft lip/palate (CLP) with other anomalies is not uncommon, but its association with Wilms tumor (WT) is very rare, especially in a familial pattern. In this report, we present a family in which six members in two generations were affected with CLP, WT, or both. PATIENTS AND RESULTS: A male patient presented with right complete CLP. He had a family history of facial cleft and abdominal tumor. Lip repair was performed at 3 months of age. An abdominal mass was noticed at 12 months of age, which proved to be WT. Surgical excision of the tumor and chemotherapy were conducted. He subsequently underwent palate repair. His father had an unrepaired microform cleft lip. Three of his aunts were known to have similar problems: one had both facial cleft and WT, one had WT only, and the other had facial cleft only. One of his cousins also was affected with WT. CONCLUSIONS: This is a unique family affected with a rare association of CLP and WT. Pedigree study revealed an autosomal dominant hereditary pattern.

5
UI - 12148958
AU - Yang CF; Lin CY
TI - A 3-year old boy with recurrent hematuria.
SO - Acta Paediatr Taiwan 2002 May-Jun;43(3):119-21
AD - Department of Pediatrics, Taipei Veterans General Hospital, Taiwan.

6
UI - 12131349
AU - Ghanem MA; Van Steenbrugge GJ; Van Der Kwast TH; Sudaryo MK; Noordzij
TI - MA; Nijman RJ Expression and prognostic value Of CD44 isoforms in nephroblastoma (Wilms tumor).
SO - J Urol 2002 Aug;168(2):681-6
AD - Department of Pediatric Urology, Josephine Nefkens Institute, The Netherlands Institute for Health Sciences, Erasmus MC, Rotterdam, The Netherlands.
PURPOSE: CD44 is a group of transmembranous glycoproteins formed by alternative splicing of a single messenger RNA. An abnormal pattern of CD44 expression has been demonstrated in several human malignancies. We evaluate the prognostic value of standard CD44 (CD44s) and some of its isoforms in treating clinical Wilms tumor. MATERIALS AND METHODS: The immunohistochemical expression of CD44 isoforms was studied in paraffin material of 61 clinical Wilms tumors. Patients were treated preoperatively with chemotherapy and mean followup was 5.7 years. RESULTS: Generally CD44s, CD44v5 and CD44v10 were expressed in normal kidney tissues and at variable levels in the 3 cell types of Wilms tumor (blastemal, epithelial and stromal). No CD44v6 expression was found neither in normal kidney or in tumor tissue. CD44s, CD44v5 and CD44v10 epithelial expression gradually decreased from stage T1 to T3. By contrast the percentage of CD44 positive cells in the blastemal component significantly increased from T1 to T3. CD44 immunoreactive blastema cells were found in 62%, 44% and 41% for CD44s, CD44v5 and CD44v10, respectively. Blastemal expression of CD44s and CD44v5 statistically significantly correlated with clinicopathological stage. Univariate and multivariate analyses showed that blastemal CD44v5 expression was indicative of poor prognosis. CONCLUSIONS: Increased expression of CD44v5 in the blastemal part of Wilms tumor correlated with tumor stage, clinical progression and tumor related death. Therefore, blastemal CD44v5 expression may be of value in identifying patients with a high propensity for distant metastases. These patients might benefit from adjuvant chemotherapy and/or radiotherapy.

7
UI - 12142789
AU - Kulkarni R; Wolf JS Jr; Padiyar N; Zuckerman L; Gera R; Scott-Emuakpor
TI - AB Severe intrarenal fibrosis, infundibular stenosis, renal cysts, and persistent perilobar nephrogenic rests in a patient with Beckwith-Wiedemann syndrome 27 years after diffuse nephroblastomatosis and Wilms tumor: natural progression or a consequence of treatment?
SO - J Pediatr Hematol Oncol 2002 Jun-Jul;24(5):389-93
AD - Michigan State University, East Lansing, Michigan, USA. Kulkarni@ht.msu.edu
A27-year-old woman presented with back and abdominal pain. She was diagnosed in infancy with Beckwith-Wiedemann syndrome and bilateral multifocal perilobar nephrogenic rests that progressed to diffuse nephroblastomatosis with neoplastic nephroblastomatous rests at 14 months of age and subsequently to a right Wilms tumor at 5 years of age. Computed tomography of the abdomen during the current admission showed multiple obstructed calices. Ureteroscopic inspection of the left kidney revealed severe intrarenal scarring with multiple infundibular stenosis, hydrocalices, and nephrocalcinosis. Renal biopsy showed sclerotic glomeruli with calcification and scarring and persistent subcapsular nodular renal blastema. Electrocautery incision and balloon dilatation provided temporary pain relief. After discharge, the patient has had two or three episodes of recurrent pain associated with new areas of infundibular stenoses and renal cysts. Bilateral nephrectomy and renal transplantation is being considered for management of progressive disease and relief of intractable pain. The potential causes of progressive and severe intrarenal fibrosis, infundibular stenosis and nephrocalcinosis, and renal cysts in this patient may include abnormal renal development secondary to Beckwith-Wiedemann syndrome itself, radiation or chemotherapy damage, or a combination.

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