Treatment of relapsed Wilms tumour (WT) patients: Experience with topotecan. A report from the SIOP Renal Tumour Study Group (RTSG)

Mavinkurve‐Groothuis, Annelies M. C.; Heuvel‐Eibrink, Marry M. van den; Tytgat, Godelieve A.M.; Vujanić, Gordan; Pritchard‐Jones, Kathy; Howell, Lisa; Graf, Norbert; Bergeron, Christophe; Acha, T.; Catania, Serena; Spreafico, Filippo

doi:10.1002/pbc.25357

Cited by 13 publications

(13 citation statements)

References 31 publications

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“…Despite thorough Bayesian analysis of published literature there is insufficient evidence for efficacy of high-dose chemotherapy with autologous stem cell rescue (ASCR) (66). Topoisomerase inhibitors have shown promising results, especially in WT patients with diffuse anaplasia, but further evidence is required (59,(67)(68)(69).…”

Section: Management Of Relapsementioning

confidence: 99%

Pharmacotherapeutic Management of Wilms Tumor: An Update

Oostveen

Pritchard‐Jones

2019

Pediatr Drugs

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 Collaboration in international clinical trials, improvement of multimodal therapy and risk stratification strategies have led to improved outcome for children with Wilms tumour (WT).  In children with WT where cure rates are high it is critical that the balance of risk and benefit of treatment continues to be optimised through further fine tuning of risk stratification strategies that determine which children need more intensive therapy and those that can do with less.  Work in the field of genetics and epigenetics is pointing towards common signalling pathways, dysregulation of the epigenome and the identification of gene expression profile subsets in children with WT that will direct future research into targeted therapies and enable better predictions of tumour sensitivity to therapy and recurrence.

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Section: Management Of Relapsementioning

confidence: 99%

Pharmacotherapeutic Management of Wilms Tumor: An Update

Oostveen

Pritchard‐Jones

2019

Pediatr Drugs

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“…Unlike 'first-line' treatment, 'second-line' chemotherapy is not based on evidence from randomised trials, but solely on prospective single arm studies and case series. Alternative chemotherapy (e.g., irinotecan, topotecan and temozolamide) or a further increase in dose intensity, like those reached in myeloid-ablative regimens, have demonstrated limited evidence for improvement in survival rates [17][18][19][20].…”

Section: Introductionmentioning

confidence: 99%

Review of phase I and II trials for Wilms' tumour – Can we optimise the search for novel agents?

Brok

Pritchard‐Jones

Geller

et al. 2017

European Journal of Cancer

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Survival rates for patients with Wilms' tumour (WT) approximate 90% with refined use of currently available interventions. However, a subgroup of patients, with initial high-risk histopathology or relapsing disease, have a poor prognosis, and it is a challenge to identify and prioritise the development of new innovative approaches for these subgroups. We conducted a systematic literature search for published phase I and II clinical trials that registered patients with WTs and characterised the early phase trial activity, quantified response rates and highlighted avenues for further development. We identified 63 trials (48 phase I, three phase I/II, and 12 phase II trials) enrolling 214 patients with WTs, alongside other malignancies. The number of annually recruited WTs did not change significantly and was less than 20% of the potential candidates. The vast majority of the trials were conducted in North America, and 56 different interventions were investigated, including conventional chemotherapy and biologically targeted therapies. Overall, 33 WTs revealed some degree of tumour control. Of these, five patients demonstrated complete remission (2%), 15 patients partial response (7%) and 13 patients stable disease (6%). None of the included novel biologically targeted therapies emerged as promising interventions, and only conventional chemotherapy was able to induce a complete and partial response. We conclude that early phase trial recruitment of WTs is below expected levels, and the clinical outcome of the included patients is dismal. Improvement of the availability and recruitment to early phase trials for WTs, especially in Europe, is needed.

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“…Similarly, Mavinkurve et al. observed more responses to topotecan in patients with intermediate‐risk (IR) histology compared to those with high‐risk (HR) histology . So far, limited information on irinotecan for relapsed WT is available from preclinical and phase I/II studies, showing some antitumor activity, but no randomized studies have been performed .…”

Section: Introductionmentioning

confidence: 99%

“…[1][2][3] Irinotecan is a camptothecin compound that interferes with DNA replication and cell division by a mechanism similar to that of topotecan. 4,5 For topotecan, a large phase II trial by Metzger et al…”

Section: Introductionmentioning

confidence: 99%

“…6 Similarly, Mavinkurve et al observed more responses to topotecan in patients with intermediate-risk (IR) histology compared to those with high-risk (HR) histology. 5 So far, limited information on irinotecan for relapsed WT is available from preclinical and phase I/II studies, showing some antitumor activity, but no randomized studies have been performed. [7][8][9][10][11][12][13][14][15][16][17][18][19][20][21] Researchers from the Children's Oncology Group are evaluating irinotecan combined with other chemotherapeutic agents in upfront treatment for metastatic diffuse anaplastic WT, a diagnosis with a poor prognosis.…”

Section: Introductionmentioning

confidence: 99%

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Irinotecan for relapsed Wilms tumor in pediatric patients: SIOP experience and review of the literature—A report from the SIOP Renal Tumor Study Group

Hol

Heuvel‐Eibrink

Graf

et al. 2017

Pediatric Blood & Cancer

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While irinotecan has been studied in various pediatric solid tumors, its potential role in Wilms tumor (WT) is less clear. We evaluated response and outcome of irinotecan-containing regimens in relapsed WT and compared our results to the available literature. Among 14 evaluable patients, one complete response (CR) and two partial responses (PRs) were observed in patients with initial intermediate-risk (CR and PR) and blastemal-type histologies (PR). Two patients were alive at last follow-up showing no evidence of disease. Our results and the reviewed literature suggest some effectiveness of irinotecan in the setting of relapsed WT.

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Treatment of relapsed Wilms tumour (WT) patients: Experience with topotecan. A report from the SIOP Renal Tumour Study Group (RTSG)

Abstract: Topotecan does not seem to show effectiveness in the treatment of relapsed WT patients with initial high-risk histology. In patients with intermediate-risk histology, the role of topotecan might deserve further attention, to prove its efficacy.

Cited by 13 publications

References 31 publications

Pharmacotherapeutic Management of Wilms Tumor: An Update

Pharmacotherapeutic Management of Wilms Tumor: An Update

Review of phase I and II trials for Wilms' tumour – Can we optimise the search for novel agents?

Irinotecan for relapsed Wilms tumor in pediatric patients: SIOP experience and review of the literature—A report from the SIOP Renal Tumor Study Group

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