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Open Access 01-12-2023 | Sarcoma | Research

Nomogram development and external validation for predicting overall survival and cancer-specific survival in patients with primary retroperitoneal sarcoma: a retrospective cohort study

Authors: Jialiang Zheng, Aobo Zhuang, Xiaogang Xia, Fenglin Miao, Zhao Wang, Xu Kong, Yantao Ren, Yuan Ma, Zhenhang Lin, Weiqi Lu, Wengang Li

Published in: Discover Oncology | Issue 1/2023

Abstract

Background

Primary retroperitoneal sarcoma (RPS) comprises over 70 histologic subtypes, yet there are limited studies that have developed prognostic nomograms for RPS patients to predict overall survival (OS) and cancer-specific survival (CSS). The objective of this study was to construct prognostic nomograms for predicting OS and CSS in RPS patients.

Methods

We identified a total of 1166 RPS patients from the Surveillance, Epidemiology and End Results (SEER) database, and an additional 261 cases were collected from a tertiary cancer center. The study incorporated various clinicopathological and epidemiologic features as variables, and prediction windows for overall survival (OS) and cancer-specific survival (CSS) were set at 3, 5, and 7 years. Multivariable Cox models were utilized to develop the nomograms, and variable selection was performed using a backward procedure based on the Akaike Information Criterion. To evaluate the performance of the nomograms in terms of calibration and discrimination, we used calibration plots, coherence index, and area under the curve.

Findings

The study included 818 patients in the development cohort, 348 patients in the internal validation cohort, and 261 patients in the external validation cohort. The backward procedure selected the following variables: age, French Federation of Cancer Centers Sarcoma Group (FNCLCC) grade, pre-/postoperative chemotherapy, tumor size, primary site surgery, and tumor multifocality. The validation results demonstrated that the nomograms had good calibration and discrimination, with C-indices of 0.76 for OS and 0.81 for CSS. Calibration plots also showed good consistency between the predicted and actual survival rates. Furthermore, the areas under the time-dependent receiver operating characteristic curves for the 3-, 5-, and 7-year OS (0.84, 0.82, and 0.78, respectively) and CSS (0.88, 0.88, and 0.85, respectively) confirmed the accuracy of the nomograms.

Interpretation

Our study developed accurate nomograms to predict OS and CSS in patients with RPS. These nomograms have important clinical implications and can assist healthcare providers in making informed decisions regarding patient care and treatment options. They may also aid in patient counseling and stratification in clinical trials.

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Swallow CJ, Strauss DC, Bonvalot S, Rutkowski P, Desai A, Gladdy RA, Gonzalez R, Gyorki DE, Fairweather M, van Houdt WJ, Stoeckle E, Park JB, Albertsmeier M, Nessim C, Cardona K, Fiore M, Hayes A, Tzanis D, Skoczylas J, Ford SJ, Ng D, Mullinax JE, Snow H, Haas RL, Callegaro D, Smith MJ, Bouhadiba T, Stacchiotti S, Jones RL, DeLaney T, Roland CL, Raut CP, Gronchi A. Management of primary retroperitoneal sarcoma (RPS) in the adult: an updated consensus approach from the transatlantic australasian rps working group. Ann Surg Oncol. 2021;28:7873–88.PubMedPubMedCentralCrossRef

Álvarez Álvarez R, Manzano A, Agra Pujol C, Artigas Raventós V, Correa R, Cruz Jurado J, Fernandez JA, Garcia Del Muro X, Gonzalez JA, Hindi N, Lozano Lominchar P, Martínez-Trufero J, Méndez R, Muñoz M, Muñoz Casares C, Orbis Castellanos F, Orellana Fernandez R, Paniagua González M, Redondo A, Valverde Morales C, Asencio JM. Updated review and clinical recommendations for the diagnosis and treatment of patients with retroperitoneal sarcoma by the Spanish Sarcoma Research Group (GEIS). Cancers (Basel). 2023;15:3194.PubMedCrossRef

Gamboa AC, Gronchi A, Cardona K. Soft-tissue sarcoma in adults: an update on the current state of histiotype-specific management in an era of personalized medicine. CA Cancer J Clin. 2020;70:200–29.PubMedCrossRef

Bonvalot S, Gronchi A, Le Pechoux C, Swallow CJ, Strauss D, Meeus P, van Coevorden F, Stoldt S, Stoeckle E, Rutkowski P, Rastrelli M, Raut CP, Hompes D, De Paoli A, Sangalli C, Honore C, Chung P, Miah A, Blay JY, Fiore M, Stelmes JJ, Dei Tos AP, Baldini EH, Litiere S, Marreaud S, Gelderblom H, Haas RL. Preoperative radiotherapy plus surgery versus surgery alone for patients with primary retroperitoneal sarcoma (EORTC-62092: STRASS): a multicentre, open-label, randomised, phase 3 trial. Lancet Oncol. 2020;21:1366–77.PubMedCrossRef

Munoz P, Bretcha-Boix P, Artigas V, Asencio JM. Surgical principles of primary retroperitoneal sarcoma in the era of personalized treatment: a review of the frontline extended surgery. Cancers (Basel). 2022;14:4091.PubMedCrossRef

von Mehren M, Kane JM, Bui MM, Choy E, Connelly M, Dry S, Ganjoo KN, George S, Gonzalez RJ, Heslin MJ, Homsi J, Keedy V, Kelly CM, Kim E, Liebner D, McCarter M, McGarry SV, Meyer C, Pappo AS, Parkes AM, Paz IB, Petersen IA, Poppe M, Riedel RF, Rubin B, Schuetze S, Shabason J, Sicklick JK, Spraker MB, Zimel M, Bergman MA, George GV. NCCN guidelines insights: soft tissue sarcoma, version 1.2021. J Natl Compr Canc Netw. 2020;18:1604–12.CrossRef

Derbel O, Heudel PE, Cropet C, Meeus P, Vaz G, Biron P, Cassier P, Decouvelaere AV, Ranchere-Vince D, Collard O, De Laroche E, Thiesse P, Farsi F, Cellier D, Gilly FN, Blay JY, Ray-Coquard I. Survival impact of centralization and clinical guidelines for soft tissue sarcoma (A prospective and exhaustive population-based cohort). PLoS ONE. 2017;12: e0158406.PubMedPubMedCentralCrossRef

Anaya DA, Lahat G, Wang X, Xiao L, Pisters PW, Cormier JN, Hunt KK, Feig BW, Lev DC, Pollock RE. Postoperative nomogram for survival of patients with retroperitoneal sarcoma treated with curative intent. Ann Oncol. 2010;21:397–402.PubMedCrossRef

Gronchi A, Miceli R, Shurell E, Eilber FC, Eilber FR, Anaya DA, Kattan MW, Honore C, Lev DC, Colombo C, Bonvalot S, Mariani L, Pollock RE. Outcome prediction in primary resected retroperitoneal soft tissue sarcoma: histology-specific overall survival and disease-free survival nomograms built on major sarcoma center data sets. J Clin Oncol. 2013;31:1649–55.PubMedCrossRef

10.

Raut CP, Callegaro D, Miceli R, Barretta F, Rutkowski P, Blay JY, Lahat G, Strauss DC, Gonzalez R, Ahuja N, Grignani G, Quagliuolo V, Stoeckle E, De Paoli A, Pillarisetty VG, Nessim C, Swallow CJ, Bagaria S, Canter R, Mullen J, Gelderblom HJ, Pennacchioli E, van Coevorden F, Cardona K, Fiore M, Fairweather M, Gronchi A. Predicting survival in patients undergoing resection for locally recurrent retroperitoneal sarcoma: a study and novel nomogram from TARPSWG. Clin Cancer Res. 2019;25:2664–71.PubMedCrossRef

11.

Callegaro D, Barretta F, Swallow CJ, Strauss DC, Bonvalot S, Honore C, Stoeckle E, van Coevorden F, Haas R, Rutkowski P, Schrage Y, Fairweather M, Conti L, Vassos N, Gladdy RA, Ng D, van Houdt WJ, Miceli R, Raut CP, Gronchi A. Longitudinal prognostication in retroperitoneal sarcoma survivors: development and external validation of two dynamic nomograms. Eur J Cancer. 2021;157:291–300.PubMedCrossRef

12.

Ardoino I, Miceli R, Berselli M, Mariani L, Biganzoli E, Fiore M, Collini P, Stacchiotti S, Casali PG, Gronchi A. Histology-specific nomogram for primary retroperitoneal soft tissue sarcoma. Cancer. 2010;116:2429–36.PubMedCrossRef

13.

Tan MC, Brennan MF, Kuk D, Agaram NP, Antonescu CR, Qin LX, Moraco N, Crago AM, Singer S. Histology-based classification predicts pattern of recurrence and improves risk stratification in primary retroperitoneal sarcoma. Ann Surg. 2016;263:593–600.PubMedCrossRef

14.

Doll KM, Rademaker A, Sosa JA. Practical guide to surgical data sets: surveillance, epidemiology, and end results (SEER) database. JAMA Surg. 2018;153:588–9.PubMedCrossRef

15.

Camp RL, Dolled-Filhart M, Rimm DL. X-tile: a new bio-informatics tool for biomarker assessment and outcome-based cut-point optimization. Clin Cancer Res. 2004;10:7252–9.PubMedCrossRef

16.

Akaike H. Information theory and an extension of the maximum likelihood principle. In: Parzen E, Tanabe K, Kitagawa G, editors. Selected papers of Hirotugu Akaike. New York: Springer; 1998. p. 199–213.CrossRef

17.

Venables WN, Ripley BD, Venables WN. Modern applied statistics with S. 4th ed. New York: Springer; 2002.CrossRef

18.

Therneau TM, Grambsch PM. Modeling survival data: extending the Cox model. New York: Springer; 2000.CrossRef

19.

Gronchi A, Strauss DC, Miceli R, Bonvalot S, Swallow CJ, Hohenberger P, Van Coevorden F, Rutkowski P, Callegaro D, Hayes AJ, Honore C, Fairweather M, Cannell A, Jakob J, Haas RL, Szacht M, Fiore M, Casali PG, Pollock RE, Raut CP. Variability in patterns of recurrence after resection of primary retroperitoneal sarcoma (RPS): a report on 1007 patients from the multi-institutional collaborative RPS Working Group. Ann Surg. 2016;263:1002–9.PubMedCrossRef

20.

Tyler R, Wanigasooriya K, Taniere P, Almond M, Ford S, Desai A, Beggs A. A review of retroperitoneal liposarcoma genomics. Cancer Treat Rev. 2020;86: 102013.PubMedCrossRef

21.

Zuco V, Pasquali S, Tortoreto M, Brich S, Percio S, Dagrada GP, Colombo C, Sanfilippo R, Lauricella C, Gounder M, El Bezawy R, Barisella M, Dei Tos AP, Casali PG, Gronchi A, Stacchiotti S, Zaffaroni N. Selinexor versus doxorubicin in dedifferentiated liposarcoma PDXs: evidence of greater activity and apoptotic response dependent on p53 nuclear accumulation and survivin down-regulation. J Exp Clin Cancer Res. 2021;40:83.PubMedPubMedCentralCrossRef

22.

Almond LM, Tirotta F, Tattersall H, Hodson J, Cascella T, Barisella M, Marchiano A, Greco G, Desai A, Ford SJ, Gronchi A, Fiore M, Morosi C. Diagnostic accuracy of percutaneous biopsy in retroperitoneal sarcoma. Br J Surg. 2019;106:395–403.PubMedCrossRef

23.

Chen H, Wei J, Wang R, Xiang Y, Fan C. Retroperitoneal Liposarcoma on 99mTc-DTPA Renal Scintigraphy. Clin Nucl Med. 2020;45:129–30.PubMedCrossRef

24.

Tyler R, Dilworth MP, James J, Blakeway D, Stockton JD, Morton DG, Taniere P, Gourevitch D, Desai A, Beggs AD. The molecular landscape of well differentiated retroperitoneal liposarcoma. J Pathol. 2021;255:132–40.PubMedCrossRef

25.

Xu C, Yan L, Guan X, Wang Z, Wu J, Lv A, Liu D, Liu F, Dong B, Zhao M, Jia L, Tian X, Hao C. Tsp2 facilitates tumor-associated fibroblasts formation and promotes tumor progression in retroperitoneal liposarcoma. Int J Biol Sci. 2022;18:5038–55.PubMedPubMedCentralCrossRef

26.

Tucci JJ, Dashti NK, Cates JMM. A proposed staging system for improved prognostication of MDM2-amplified liposarcoma. Am J Surg Pathol. 2021;45:101–7.PubMedCrossRef

27.

Marko J, Wolfman DJ. Retroperitoneal leiomyosarcoma from the radiologic pathology archives. Radiographics. 2018;38:1403–20.PubMedCrossRef

28.

Carvalho JC, Thomas DG, Lucas DR. Cluster analysis of immunohistochemical markers in leiomyosarcoma delineates specific anatomic and gender subgroups. Cancer. 2009;115:4186–95.PubMedCrossRef

29.

Kezios KL, Lu P, Calonico S, Al Hazzouri AZ. History of low hourly wage and all-cause mortality among middle-aged workers. JAMA. 2023;329:561–73.PubMedPubMedCentralCrossRef

30.

Landon BE, Hatfield LA, Bakx P, Banerjee A, Chen YC, Fu C, Gordon M, Heine R, Huang N, Ko DT, Lix LM, Novack V, Pasea L, Qiu F, Stukel TA, Uyl-de Groot C, Yan L, Weinreb G, Cram P. Differences in treatment patterns and outcomes of acute myocardial infarction for low- and high-income patients in 6 countries. JAMA. 2023;329:1088–97.PubMedPubMedCentralCrossRef

31.

Gundle KR, Kafchinski L, Gupta S, Griffin AM, Dickson BC, Chung PW, Catton CN, O’Sullivan B, Wunder JS, Ferguson PC. Analysis of margin classification systems for assessing the risk of local recurrence after soft tissue sarcoma resection. J Clin Oncol. 2018;36:704–9.PubMedCrossRef

32.

von Mehren M, Kane JM, Agulnik M, Bui MM, Carr-Ascher J, Choy E, Connelly M, Dry S, Ganjoo KN, Gonzalez RJ, Holder A, Homsi J, Keedy V, Kelly CM, Kim E, Liebner D, McCarter M, McGarry SV, Mesko NW, Meyer C, Pappo AS, Parkes AM, Petersen IA, Pollack SM, Poppe M, Riedel RF, Schuetze S, Shabason J, Sicklick JK, Spraker MB, Zimel M, Hang LE, Sundar H, Bergman MA. Soft tissue sarcoma, version 2.2022, NCCN clinical practice guidelines in oncology. J Natl Compr Canc Netw. 2022;20:815–33.CrossRef

33.

Ecker BL, Peters MG, McMillan MT, Sinnamon AJ, Zhang PJ, Fraker DL, Levin WP, Roses RE, Karakousis GC. Preoperative radiotherapy in the management of retroperitoneal liposarcoma. Br J Surg. 2016;103:1839–46.PubMedCrossRef

34.

Callegaro D, Raut CP, Ajayi T, Strauss D, Bonvalot S, Ng D, Stoeckle E, Fairweather M, Rutkowski P, van Houdt WJ, Gelderblom H, Sangalli C, Hayes A, Honore C, Gladdy RA, Fau M, Haas R, Tzanis D, Miah AB, Chung P, Baldini EH, Marreaud S, Litiere S, Swallow CJ, Gronchi A. Preoperative radiotherapy in patients with primary retroperitoneal sarcoma: EORTC-62092 trial (STRASS) versus off-trial (STREXIT) results. Ann Surg. 2022;278(1):127–34.PubMedCrossRef

35.

Datta J, Ecker BL, Neuwirth MG, Geha RC, Fraker DL, Roses RE, Karakousis GC. Contemporary reappraisal of the efficacy of adjuvant chemotherapy in resected retroperitoneal sarcoma: evidence from a nationwide clinical oncology database and review of the literature. Surg Oncol. 2017;26:117–24.PubMedCrossRef

36.

Kelly CM, Antonescu CR, Bowler T, Munhoz R, Chi P, Dickson MA, Gounder MM, Keohan ML, Movva S, Dholakia R, Ahmad H, Biniakewitz M, Condy M, Phelan H, Callahan M, Wong P, Singer S, Ariyan C, Bartlett EK, Crago A, Yoon S, Hwang S, Erinjeri JP, Qin LX, Tap WD, D’Angelo SP. Objective response rate among patients with locally advanced or metastatic sarcoma treated with talimogene laherparepvec in combination with pembrolizumab: a phase 2 clinical trial. JAMA Oncol. 2020;6:402–8.PubMedPubMedCentralCrossRef

37.

Gronchi A, Palmerini E, Quagliuolo V, Martin Broto J, Lopez Pousa A, Grignani G, Brunello A, Blay JY, Tendero O, Diaz Beveridge R, Ferraresi V, Lugowska I, Merlo DF, Fontana V, Marchesi E, Braglia L, Donati DM, Palassini E, Bianchi G, Marrari A, Morosi C, Stacchiotti S, Bague S, Coindre JM, Dei Tos AP, Picci P, Bruzzi P, Casali PG. Neoadjuvant chemotherapy in high-risk soft tissue sarcomas: final results of a randomized trial from Italian (ISG), Spanish (GEIS), French (FSG), and Polish (PSG) Sarcoma Groups. J Clin Oncol. 2020;38:2178–86.PubMedCrossRef

38.

C.D.M. Fletcher, World Health Organization, International Agency for Research on Cancer. WHO classification of tumours of soft tissue and bone. 4th ed. Lyon: IARC Press; 2013.

39.

Gronchi A, Miceli R, Colombo C, Stacchiotti S, Collini P, Mariani L, Sangalli C, Radaelli S, Sanfilippo R, Fiore M, Casali PG. Frontline extended surgery is associated with improved survival in retroperitoneal low- to intermediate-grade soft tissue sarcomas. Ann Oncol. 2012;23:1067–73.PubMedCrossRef

40.

Bonvalot S, Miceli R, Berselli M, Causeret S, Colombo C, Mariani L, Bouzaiene H, Le Pechoux C, Casali PG, Le Cesne A, Fiore M, Gronchi A. Aggressive surgery in retroperitoneal soft tissue sarcoma carried out at high-volume centers is safe and is associated with improved local control. Ann Surg Oncol. 2010;17:1507–14.PubMedCrossRef

41.

Bonvalot S, Rivoire M, Castaing M, Stoeckle E, Le Cesne A, Blay JY, Laplanche A. Primary retroperitoneal sarcomas: a multivariate analysis of surgical factors associated with local control. J Clin Oncol. 2009;27:31–7.PubMedCrossRef

42.

Gronchi A, Lo Vullo S, Fiore M, Mussi C, Stacchiotti S, Collini P, Lozza L, Pennacchioli E, Mariani L, Casali PG. Aggressive surgical policies in a retrospectively reviewed single-institution case series of retroperitoneal soft tissue sarcoma patients. J Clin Oncol. 2009;27:24–30.PubMedCrossRef

43.

Lochan R, French JJ, Manas DM. Surgery for retroperitoneal soft tissue sarcomas: aggressive re-resection of recurrent disease is possible. Ann R Coll Surg Engl. 2011;93:39–43.PubMedCrossRef

44.

R.P.S.W.G. Trans-Atlantic. Management of primary retroperitoneal sarcoma (RPS) in the adult: a consensus approach from the Trans-Atlantic RPS Working Group. Ann Surg Oncol. 2015;22:256–63.CrossRef

45.

Ma SJ, Oladeru OT, Farrugia MK, Shekher R, Iovoli AJ, Singh AK. Evaluation of preoperative chemotherapy or radiation and overall survival in patients with nonmetastatic, resectable retroperitoneal sarcoma. JAMA Netw Open. 2020;3: e2025529.PubMedPubMedCentralCrossRef

46.

Italiano A, Penel N, Robin YM, Bui B, Le Cesne A, Piperno-Neumann S, Tubiana-Hulin M, Bompas E, Chevreau C, Isambert N, Leyvraz S, du Chatelard PP, Thyss A, Coindre JM, Blay JY. Neo/adjuvant chemotherapy does not improve outcome in resected primary synovial sarcoma: a study of the French Sarcoma Group. Ann Oncol. 2009;20:425–30.PubMedCrossRef

47.

Bonvalot S, Raut CP, Pollock RE, Rutkowski P, Strauss DC, Hayes AJ, Van Coevorden F, Fiore M, Stoeckle E, Hohenberger P, Gronchi A. Technical considerations in surgery for retroperitoneal sarcomas: position paper from E-Surge, a master class in sarcoma surgery, and EORTC-STBSG. Ann Surg Oncol. 2012;19:2981–91.PubMedCrossRef

48.

Haas RLM, Bonvalot S, Miceli R, Strauss DC, Swallow CJ, Hohenberger P, van Coevorden F, Rutkowski P, Callegaro D, Hayes AJ, Honore C, Fairweather M, Gladdy R, Jakob J, Szacht M, Fiore M, Chung PW, van Houdt WJ, Raut CP, Gronchi A. Radiotherapy for retroperitoneal liposarcoma: a report from the Transatlantic Retroperitoneal Sarcoma Working Group. Cancer. 2019;125:1290–300.PubMedCrossRef

49.

Bremjit PJ, Jones RL, Chai X, Kane G, Rodler ET, Loggers ET, Pollack SM, Pillarisetty VG, Mann GN. A contemporary large single-institution evaluation of resected retroperitoneal sarcoma. Ann Surg Oncol. 2014;21:2150–8.PubMedCrossRef

50.

R.P.S.W.G. Trans-Atlanctic. Management of recurrent retroperitoneal sarcoma (RPS) in the adult: a consensus approach from the Trans-Atlantic RPS Working Group. Ann Surg Oncol. 2016;23:3531–40.CrossRef

51.

Tseng WW, Madewell JE, Wei W, Somaiah N, Lazar AJ, Ghadimi MP, Hoffman A, Pisters PW, Lev DC, Pollock RE. Locoregional disease patterns in well-differentiated and dedifferentiated retroperitoneal liposarcoma: implications for the extent of resection? Ann Surg Oncol. 2014;21:2136–43.PubMedCrossRef

Title: Nomogram development and external validation for predicting overall survival and cancer-specific survival in patients with primary retroperitoneal sarcoma: a retrospective cohort study
Authors: Jialiang Zheng
Aobo Zhuang
Xiaogang Xia
Fenglin Miao
Zhao Wang
Xu Kong
Yantao Ren
Yuan Ma
Zhenhang Lin
Weiqi Lu
Wengang Li
Publication date: 01-12-2023
Publisher: Springer US
Keyword: Sarcoma
Published in: Discover Oncology / Issue 1/2023
Print ISSN: 1868-8497
Electronic ISSN: 2730-6011
DOI: https://doi.org/10.1007/s12672-023-00804-1

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