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Development and Initial Validation of the ONCOREUM Score to Differentiate Childhood Cancer with Arthropathy from Juvenile Idiopathic Arthritis - 15/03/23

Doi : 10.1016/j.jpeds.2022.09.046 
Adele Civino, MD 1, , Francesca Bovis, PhD 2, Marta Ponzano, MSc 2, Giovanni Alighieri, MD 3, Eleonora Prete, MD 4, Stefania Sorrentino, MD 5, Silvia Magni-Manzoni, MD 6, Luciana Vinti, MD, PhD 7, Micol Romano, MD 8, Nicola Santoro, MD 9, Giovanni Filocamo, MD, PhD 10, Tamara Belotti, MD, PhD 11, Francesca Santarelli, MD 12, Chiara Gorio, MD 13, Marco Cattalini, MD 14, Giulia Stabile, MSc 15, Valentino Conter, MD 16, Roberto Rondelli, MD 11, Andrea Pession, MD 17, , Angelo Ravelli, MD 18, 19,
on behalf of the

ONCOREUM Study Group

  A list of additional members of the ONCOREUM Study Group is available at www.jpeds.com ().

1 Pediatric Rheumatology and Immunology, Hospital "Vito Fazzi", Lecce, Italy 
2 Department of Health Sciences, Section of Biostatistics, University of Genova, Italy 
3 Neonatology and Neonatal Intensive Care Unit, Hospital "A. Perrino", Brindisi, Italy 
4 Department of Hematology and Bone Marrow Transplant, Hospital "Card. G. Panico", Tricase, Italy 
5 Department of Paediatric Haemato-Oncology, Scientific Institute for Research, Hospitalization and Healthcare (IRCCS) Giannina Gaslini Institute, Genoa, Italy 
6 Rheumatology Division, Children's Hospital, IRCCS Bambino Gesù, Rome, Italy 
7 Department of Pediatric Hemato-Oncology, IRCCS Bambino Gesù, Rome, Italy 
8 Paediatric Rheumatology, Hospital and Orthopaedic Trauma Centre "G. Pini", Milan, Italy 
9 Department of Pediatric Oncology and Hematology, University "Aldo Moro" Bari, Italy 
10 Pediatric Rheumatology, IRCCS Ca' Granda Maggiore Hospital, Milan, Italy 
11 Pediatric Oncology and Haematology Unit, IRCCS, University of Bologna, Italy 
12 Division of Pediatric Immunology and Rheumatology, Department of Public Health and Pediatrics, "Regina Margherita" Children Hospital, University of Turin, Italy 
13 Pediatric Oncohematology and Bone Marrow Transplant Unit, University of Brescia and "Spedali Civili", Brescia, Italy 
14 Pediatric Clinic, University of Brescia and "Spedali Civili", Brescia, Italy 
15 CINECA Interuniversity Consortium, Casalecchio di Reno, Italy 
16 Pediatric Hematology-Oncology Unit, University of Milano-Bicocca, Monza, Italy 
17 Pediatric Unit, IRCCS S.Orsola-Malpighi Hospital, University of Bologna, Italy 
18 Scientific Director, Giannina Gaslini Institute, Genoa, Italy 
19 Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics and Maternal-Infantile Sciences (DiNOGMI), University of Genoa, Italy 

Reprint requests: Adele Civino, MD, Reumatologia e Immunologia Pediatrica, Ospedale “Vito Fazzi”, Piazza Muratore, 1, 73100 Lecce, Italy.Reumatologia e Immunologia PediatricaOspedale “Vito Fazzi”Piazza Muratore, 1Lecce73100Italy

Abstract

Objective

To develop and validate a weighted score, the ONCOREUM score, that aids physicians in differentiation of cancer with arthropathy from juvenile idiopathic arthritis (JIA).

Study design

Data were extracted from the ONCOREUM Study, a multicenter, cross-sectional investigation aimed at comparing children with cancer and arthropathy to children with JIA. Three statistical approaches were applied to develop the ONCOREUM score and assess the role of each variable in the diagnosis of cancer with arthropathy, including 2 approaches based on multivariable stepwise selection (models 1 and 2) and 1 approach on a Bayesian model averaging method (model 3). The β coefficients estimated in the models were used to assign score points. Considering that not missing a child with cancer is a mandatory clinical objective, discriminating performance was assessed by fixing sensitivity at 100%. Score performance was evaluated in both developmental and validation samples (representing 80% and 20% of the study population, respectively).

Results

Patients with cancer and arthropathy (49 with solid tumors and 46 with hematologic malignancies without peripheral blasts) and 677 patients with JIA were included. The highest area under the receiver operating characteristic (ROC) curve (AUC) in the validation data set was yielded by model 1, which was selected to constitute the ONCOREUM score. The score ranged from −18 to 21.8, and the optimal cutoff obtained through ROC analysis was −6. The sensitivity, specificity, and AUC of the cutoff in the validation sample were 100%, 70%, and 0.85, respectively.

Conclusions

The ONCOREUM score is a powerful and easily applicable tool that may facilitate early differentiation of malignancies with articular complaints from JIA.

Il testo completo di questo articolo è disponibile in PDF.

Keywords : bone pain, cancer, child, adolescent, juvenile idiopathic arthritis, musculoskeletal symptoms

Abbreviations : AUC, BMA, JIA, LDH, ROC, TRIPOD


Mappa


 The authors declare no conflicts of interest.


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