Genomic complexity in pediatric synovial sarcomas (Synobio study): the European pediatric soft tissue sarcoma group (EpSSG) experience

被引:22
作者
Orbach, Daniel [1 ]
Mosseri, Veronique [2 ]
Pissaloux, Daniel [3 ]
Pierron, Gaelle [4 ]
Brennan, Bernadette [5 ]
Ferrari, Andrea [6 ]
Chibon, Frederic [7 ]
Bisogno, Gianni [8 ]
De Salvo, Gian Luca [9 ]
Chakiba, Camille [7 ]
Corradini, Nadege [10 ]
Minard-Colin, Veronique [11 ]
Kelsey, Anna [12 ]
Ranchere-Vince, Dominique [3 ]
机构
[1] PSL Univ, Inst Curie, SIREDO Oncol Ctr Care Innovat & Res Children Adol, Paris, France
[2] Inst Curie, Dept Biostat, Paris, France
[3] Ctr Leon Berard, Inst Hematol & Oncol Pediat, Biopathol Dept, Lyon, France
[4] Inst Curie, Somat Genet Unit, Paris, France
[5] Royal Manchester Childrens Hosp, Dept Paediat Oncol, Manchester, Lancs, England
[6] Fdn IRCCS Ist Nazl Tumori, Pediat Oncol Unit, Milan, Italy
[7] Inst Bergonie, Dept Biopathol, Bordeaux, France
[8] Padova Univ, Pediat Hematol & Oncol Div, Padua, Italy
[9] IRCCS Ist Oncol Veneto, Clin Trials & Biostat Unit, Padua, Italy
[10] Ctr Leon Berard, Inst Hematol & Oncol Pediat, Lyon, France
[11] Gustave Roussy, Dept Paediat & Adolescent Oncol, Villejuif, France
[12] Royal Manchester Childrens Hosp, Dept Diagnost Paediat Histopathol, Manchester, Lancs, England
关键词
Adolescent; comparative genomic hybridization; EpSSG; genomic index; synovial sarcoma; SSX FUSION TYPE; CHROMOSOME INSTABILITY; RETROSPECTIVE ANALYSIS; PROGNOSTIC-FACTOR; ADOLESCENTS; CHILDREN; CHEMOTHERAPY; SURVIVAL; CANCER; TUMORS;
D O I
10.1002/cam4.1415
中图分类号
R73 [肿瘤学];
学科分类号
100214 ;
摘要
A genomic index (GI) tool using array comparative genomic hybridization (aCGH) on tumor cells has emerged as independent prognostic factor associated with the risk of metastatic relapse in synovial sarcoma (SS). The aim was to assess GI in pediatric patients with SS, to determine its value as a prognostic factor. All pediatric/adolescent/young adults' (<25 years) with localized SS prospectively included in the European EpSSG-NRSTS05 protocol with a contributive aCGH were selected. Definition of GI was A(2)/C, where A is the total number of alterations (segmental gains and losses) and C is the number of involved chromosomes on aCGH results. GI(1) group corresponds to cases with no copy number alterations (flat profile, GI=0) and GI(2) group cases with at least one or more copy number alterations (rearranged profile; GI(1)). Samples were available from 61 patients. The median age of the cohort was 13 years (range: 4-24). Overall, 55.7% were GI(1) group, and 44.3% GI(2). After a median follow-up of 62 months (range: 0.1-112), 10 tumor events occurred and five patients died. Respectively, for GI(1) versus GI(2) groups, five-year event-free survival (EFS)was 93.8 +/- 4.2% versus 64.9 +/- 10.1% (P < 0.006) and five-year Metastatic-Free Survival (MFS) 93.8 +/- 4.2% versus 72.9 +/- 9.5% (P < 0.04). In multivariate analysis, GI status as adjusted for IRS group, patient age, site, and tumor size remain independent prognostic for EFS with a relative risk (RR) of 6.4 [1.3-31.9] (P < 0.01) and RR for MFS is 4.8 [0.9-25.7] (P < 0.05). Genomic complexity evaluated through GI may explain the metastatic behavior of pediatric SS.
引用
收藏
页码:1384 / 1393
页数:10
相关论文
共 28 条
[1]   Genomic Grade Index predicts postoperative clinical outcome of GIST [J].
Bertucci, F. ;
Finetti, P. ;
Ostrowski, J. ;
Kim, W. K. ;
Kim, H. ;
Pantaleo, M. A. ;
Astolfi, A. ;
Polkowski, M. ;
Birnbaum, D. .
BRITISH JOURNAL OF CANCER, 2012, 107 (08) :1433-1441
[2]   Synovial Sarcoma in Childhood and Adolescence: A Retrospective Series of 77 Patients Registered by the Children's Cancer and Leukaemia Group Between 1991 and 2006 [J].
Brennan, Bernadette ;
Stevens, Michael ;
Kelsey, Anna ;
Stiller, Charles A. .
PEDIATRIC BLOOD & CANCER, 2010, 55 (01) :85-90
[3]   Response to chemotherapy is not related to chromosome instability in synovial sarcoma [J].
Chakiba, C. ;
Lagarde, P. ;
Pissaloux, D. ;
Neuville, A. ;
Brulard, C. ;
Perot, G. ;
Coindre, J. M. ;
Terrier, P. ;
Ranchere-Vince, D. ;
Ferrari, A. ;
Collini, P. ;
Suurmeijer, A. J. H. ;
Blay, J. Y. ;
Terrisse, S. A. ;
Piperno-Neumann, S. ;
Averous, G. ;
Bui, B. ;
Orbach, D. ;
Italiano, A. ;
Chibon, F. .
ANNALS OF ONCOLOGY, 2014, 25 (11) :2267-2271
[4]   Validated prediction of clinical outcome in sarcomas and multiple types of cancer on the basis of a gene expression signature related to genome complexity [J].
Chibon, Frederic ;
Lagarde, Pauline ;
Salas, Sebastien ;
Perot, Gaelle ;
Brouste, Veronique ;
Tirode, Franck ;
Lucchesi, Carlo ;
de Reynies, Aurelien ;
Kauffmann, Audrey ;
Bui, Binh ;
Terrier, Philippe ;
Bonvalot, Sylvie ;
Le Cesne, Axel ;
Vince-Ranchere, Dominique ;
Blay, Jean-Yves ;
Collin, Francoise ;
Guillou, Louis ;
Leroux, Agnes ;
Coindre, Jean-Michel ;
Aurias, Alain .
NATURE MEDICINE, 2010, 16 (07) :781-U81
[5]  
DESNECOCHEACAMP.R, 2017, BIOMARK RES, V5, P4
[6]   Synovial sarcoma: A retrospective analysis of 271 patients of all ages treated at a single institution [J].
Ferrari, A ;
Gronchi, A ;
Casanova, M ;
Meazza, C ;
Gandola, L ;
Collini, P ;
Lozza, L ;
Bertulli, R ;
Olmi, P ;
Casali, PG .
CANCER, 2004, 101 (03) :627-634
[7]   Synovial sarcoma in children and adolescents: the European Pediatric Soft Tissue Sarcoma Study Group prospective trial (EpSSG NRSTS 2005) [J].
Ferrari, A. ;
De Salvo, G. L. ;
Brennan, B. ;
van Noesel, M. M. ;
De Paoli, A. ;
Casanova, M. ;
Francotte, N. ;
Kelsey, A. ;
Alaggio, R. ;
Oberlin, O. ;
Carli, M. ;
Ben-Arush, M. ;
Bergeron, C. ;
Merks, J. H. M. ;
Jenney, M. ;
Stevens, M. C. ;
Bisogno, G. ;
Orbach, D. .
ANNALS OF ONCOLOGY, 2015, 26 (03) :567-572
[8]   Synovial sarcoma of children and adolescents: The prognostic role of axial sites [J].
Ferrari, Andrea ;
Bisogno, Gianni ;
Alaggio, Rita ;
Cecchetto, Giovanni ;
Collini, Paola ;
Rosolen, Angelo ;
Meazza, Cristina ;
Indolfi, Paolo ;
Garaventa, Alberto ;
De Sio, Luigi ;
D'Angelo, Paolo ;
Tamaro, Paolo ;
Casanova, Michela ;
Carli, Modesto .
EUROPEAN JOURNAL OF CANCER, 2008, 44 (09) :1202-1209
[9]   Surgery alone is sufficient therapy for children and adolescents with low-risk synovial sarcoma: A joint analysis from the European paediatric soft tissue sarcoma Study Group and the Children's Oncology Group [J].
Ferrari, Andrea ;
Chi, Yueh-Yun ;
De Salvo, Gian Luca ;
Orbach, Daniel ;
Brennan, Bernadette ;
Randall, R. Lor ;
McCarville, M. Beth ;
Black, Jennifer O. ;
Alaggio, Rita ;
Hawkins, Douglas S. ;
Bisogno, Gianni ;
Spunt, Sheri L. .
EUROPEAN JOURNAL OF CANCER, 2017, 78 :1-6
[10]   Histotype-tailored neoadjuvant chemotherapy versus standard chemotherapy in patients with high-risk soft-tissue sarcomas (ISG-STS 1001): an international, open-label, randomised, controlled, phase 3, multicentre trial [J].
Gronchi, Alessandro ;
Ferrari, Stefano ;
Quagliuolo, Vittorio ;
Martin-Broto, J. ;
Lopez Pousa, Antonio ;
Grignani, Giovanni ;
Basso, Umberto ;
Blay, Jean-Yves ;
Tendero, Oscar ;
Diaz Beveridge, Robert ;
Ferraresi, Virginia ;
Lugowska, Iwona ;
Merlo, Domenico Franco ;
Fontana, Valeria ;
Marchesi, Emanuela ;
Donati, Davide Maria ;
Palassini, Elena ;
Palmerini, Emanuela ;
De Sanctis, Rita ;
Morosi, Carlo ;
Stacchiotti, Silvia ;
Bague, Silvia ;
Coindre, Jean Michelle ;
Dei Tos, Angelo Paolo ;
Picci, Piero ;
Bruzzi, Paolo ;
Casali, Paolo Giovanni .
LANCET ONCOLOGY, 2017, 18 (06) :812-822