Original Research Outcome of SIOP patients with low- or intermediate-risk Wilms tumour relapsing after initial vincristine and actinomycin-D therapy only - the SIOP 93-01 and 2001 protocols

被引:9
|
作者
Groenendijk, Alissa [1 ]
van Tinteren, Harm [1 ]
Jiang, Yilin [1 ]
de Krijger, Ronald R. [1 ,2 ]
Vujanic, Gordan M. [3 ]
Godzinski, Jan [4 ,5 ]
Rube, Christian [6 ]
Schenk, Jens-Peter [7 ]
Morosi, Carlo [8 ]
Pritchard-Jones, Kathy [9 ]
Al-Saadi, Reem [9 ,10 ]
Vaidya, Sucheta J. [11 ]
Verschuur, Arnauld C. [12 ]
Ramirez-Villar, Gema L. [13 ]
Graf, Norbert [14 ]
de Camargo, Beatriz [15 ]
Drost, Jarno [1 ,16 ]
Perotti, Daniela [17 ]
Van den Heuvel-Eibrink, Marry M. [1 ]
Brok, Jesper [9 ,18 ]
Spreafico, Filippo [19 ]
Mavinkurve-Groothuis, Annelies M. C. [1 ]
机构
[1] Princess Maxima Ctr Pediat Oncol, Heidelberglaan 25, NL-3584 CS Utrecht, Netherlands
[2] Univ Med Ctr Utrecht, Dept Pathol, Utrecht, Netherlands
[3] Sidra Med, Dept Pathol, Doha, Qatar
[4] Marciniak Hosp, Dept Pediat Surg, Wroclaw, Poland
[5] Med Univ Wroclaw, Dept Pediat Traumatol & Emergency Med, Wroclaw, Poland
[6] Saarland Univ, Med Ctr, Dept Radiat Oncol, Fac Med, Homburg, Germany
[7] Heidelberg Univ Hosp, Dept Diagnost & Intervent Radiol, Div Pediat Radiol, Heidelberg, Germany
[8] Fdn IRCCS Ist Nazl Tumori, Dept Radiol, Milan, Italy
[9] Univ Coll London Great Ormond St Inst Child Hlth, Dev Biol & Canc Res & Teaching Dept, London, England
[10] Great Ormond St Hosp Children NHS Fdn Trust, Dept Histopathol, London, England
[11] Royal Marsden Hosp, Children & Young Peoples Unit, Sutton, Surrey, England
[12] Hop Enfants La Timone, Dept Pediat Oncol, Marseille, France
[13] Hosp Univ Virgen del Rocio, Dept Pediat Oncol, Seville, Spain
[14] Saarland Univ, Med Ctr, Dept Pediat Oncol & Hematol, Fac Med, Homburg, Germany
[15] Inst Nacl Canc, Res Ctr, Pediat Hematol & Oncol Program, Rio De Janeiro, Brazil
[16] Oncode Inst, Utrecht, Netherlands
[17] Fdn IRCCS Ist Nazl Tumori, Dept Res, Mol Bases Genet Risk & Genet Testing Unit, Milan, Italy
[18] Rigshosp, Dept Pediat Oncol & Hematol, Copenhagen, Denmark
[19] Fdn IRCCS Ist Nazl Tumori, Dept Med Oncol & Hematol, Pediat Oncol Unit, Milan, Italy
关键词
Wilms tumour; Recurrence; SIOP protocol; Treatment outcome; DOXORUBICIN; SURVIVAL; CHILDREN; GAIN; 1Q;
D O I
10.1016/j.ejca.2021.12.014
中图分类号
R73 [肿瘤学];
学科分类号
100214 ;
摘要
Purpose: Society of International Pediatric Oncology - Renal Tumor Study Group (SIOP-RTSG) treatment recommendations for relapsed Wilms tumour (WT) are stratified by the intensity of first-line treatment. To explore the evidence for the treatment of patients relapsing after vincristine and actinomycin-D (VA) treatment for primary WT, we retrospectively evaluated rescue treatment and survival of this patient group. Patients and methods: We included 109 patients with relapse after VA therapy (no radiotherapy) for stage I-II primary low-or intermediate-risk WT from the SIOP 93-01 and SIOP 2001 studies. Univariate Cox regression analysis was performed to study the effect of relapse treatment intensity on event-free survival (EFS) and overall survival (OS). Relapse treatment intensity was classified into vincristine, actinomycin-D, and either doxorubicin or epirubicin (VAD), and more intensive therapies (ifosfamide/carboplatin/etoposide [ICE]/> 4 drugs/ high-dose chemotherapy with haematopoietic stem cell transplantation [HD HSCT]). Results: Relapse treatment regimens included either VAD, or cyclophosphamide/carboplatin/ etoposide/doxorubicin (CyCED), or ICE backbones. Radiotherapy was administered in 62 patients and HD HSCT in 15 patients. Overall, 5-year EFS and OS after relapse were 72.3% (95% confidence interval [CI]: 64.0-81.6%) and 79.3% (95% CI: 71.5-88.0%), respectively. Patients treated with VAD did not fare worse when compared with patients treated with more intensive therapies (hazard ratio EFS: 0.611 [95% CI: 0.228-1.638] [p-value = 0.327] and hazard ratio OS: 0.438 [95% CI: 0.126-1.700] [p-value = 0.193]). Conclusion: Patients with relapsed WT after initial VA-only treatment showed no inferior EFS and OS when treated with VAD regimens compared with more intensive rescue regimens. A subset of patients relapsing after VA may benefit from less intensive rescue treatment than ICE/CyCED-based regimens and deserve to be pinpointed by identifying additional (molecular) prognostic factors in future studies. (C) 2021 The Authors. Published by Elsevier Ltd.
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页码:88 / 97
页数:10
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