Expert consensus on dynamics of laboratory tests for diagnosis of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis

Angelo Ravelli, Francesca Minoia, Sergio Davì, Anna Carin Horne, Francesca Bovis, Angela Pistorio, Maurizio Aricò, Tadej Avcin, Edward M. Behrens, Fabrizio De Benedetti, Alexandra Filipovic, Alexei A. Grom, Jan Inge Henter, Norman T. Ilowite, Michael B. Jordan, Raju Khubchandani, Toshiyuki Kitoh, Kai Lehmberg, Daniel J. Lovell, Paivi Miettunen & 33 others Kim E. Nichols, Seza Ozen, Jana Pachlopnik Schmid, Athimalaipet V. Ramanan, Ricardo Russo, Rayfel Schneider, Gary Sterba, Yosef Uziel, Carol Wallace, Carine Wouters, Nico Wulffraat, Erkan Demirkaya, Hermine I. Brunner, Alberto Martini, Nicolino Ruperto, Randy Q. Cron, Simona Angioloni, Chiara Pallotti, Michele Pesce, Mariangela Rinaldi, Luca Villa, Mario Abinun, Amita Aggarwal, Jonathan Akikusa, Sulaiman M. Al-Mayouf, Maria Alessio, Jordi Anton, Fabrizia Corona, Antonella Insalaco, Silvia Magni-Manzoni, on behalf of the Pediatric Rheumatology International Trials Organization, Childhood Arthritis & Rheumatology Research Alliance, Pediatric Rheumatology Collaborative Study Group and the Histiocyte Society

Research output: Contribution to journalArticle

16 Citations (Scopus)

Abstract

Objective: To identify which laboratory tests that change over time are most valuable for the timely diagnosis of macrophage activation syndrome (MAS) complicating systemic juvenile idiopathic arthritis (sJIA). Methods: A multistep process, based on a combination of expert consensus and analysis of real patient data, was conducted. A panel of experts was first asked to evaluate 115 profiles of patients with MAS, which included the values of laboratory tests at the pre-MAS visit and at MAS onset, and the change in values between the two time points. The experts were asked to choose the 5 laboratory tests in which change was most important for the diagnosis of MAS and to rank the 5 selected tests in order of importance. The relevance of change in laboratory parameters was further discussed and ranked by the same experts at a consensus conference. Results: Platelet count was the most frequently selected test, followed by ferritin level, aspartate aminotransferase (AST), white cell count, neutrophil count, and fibrinogen and erythrocyte sedimentation rate. Ferritin was most frequently assigned the highest score. At the end of the process, platelet count, ferritin level and AST were the laboratory tests in which the experts found change over time to be most important. Conclusions: We identified the laboratory tests in which change over time is most valuable for the early diagnosis of MAS in sJIA. The dynamics of laboratory values during the course of MAS should be further scrutinised in a prospective study in order to establish the optimal cut-off values for their variation.

Original languageEnglish
Article numbere000161
JournalRMD Open
Volume2
Issue number1
DOIs
Publication statusPublished - 2016

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Macrophage Activation Syndrome
Juvenile Arthritis
Clinical Laboratory Techniques
Ferritins
Aspartate Aminotransferases
Platelet Count
Blood Sedimentation
Fibrinogen
Early Diagnosis
Neutrophils
Cell Count
Prospective Studies

ASJC Scopus subject areas

  • Rheumatology
  • Immunology
  • Immunology and Allergy

Cite this

Ravelli, A., Minoia, F., Davì, S., Horne, A. C., Bovis, F., Pistorio, A., ... Pediatric Rheumatology Collaborative Study Group and the Histiocyte Society (2016). Expert consensus on dynamics of laboratory tests for diagnosis of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis. RMD Open, 2(1), [e000161]. https://doi.org/10.1136/rmdopen-2015-000161

Expert consensus on dynamics of laboratory tests for diagnosis of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis. / Ravelli, Angelo; Minoia, Francesca; Davì, Sergio; Horne, Anna Carin; Bovis, Francesca; Pistorio, Angela; Aricò, Maurizio; Avcin, Tadej; Behrens, Edward M.; De Benedetti, Fabrizio; Filipovic, Alexandra; Grom, Alexei A.; Henter, Jan Inge; Ilowite, Norman T.; Jordan, Michael B.; Khubchandani, Raju; Kitoh, Toshiyuki; Lehmberg, Kai; Lovell, Daniel J.; Miettunen, Paivi; Nichols, Kim E.; Ozen, Seza; Schmid, Jana Pachlopnik; Ramanan, Athimalaipet V.; Russo, Ricardo; Schneider, Rayfel; Sterba, Gary; Uziel, Yosef; Wallace, Carol; Wouters, Carine; Wulffraat, Nico; Demirkaya, Erkan; Brunner, Hermine I.; Martini, Alberto; Ruperto, Nicolino; Cron, Randy Q.; Angioloni, Simona; Pallotti, Chiara; Pesce, Michele; Rinaldi, Mariangela; Villa, Luca; Abinun, Mario; Aggarwal, Amita; Akikusa, Jonathan; Al-Mayouf, Sulaiman M.; Alessio, Maria; Anton, Jordi; Corona, Fabrizia; Insalaco, Antonella; Magni-Manzoni, Silvia; on behalf of the Pediatric Rheumatology International Trials Organization; Childhood Arthritis & Rheumatology Research Alliance; Pediatric Rheumatology Collaborative Study Group and the Histiocyte Society.

In: RMD Open, Vol. 2, No. 1, e000161, 2016.

Research output: Contribution to journalArticle

Ravelli, A, Minoia, F, Davì, S, Horne, AC, Bovis, F, Pistorio, A, Aricò, M, Avcin, T, Behrens, EM, De Benedetti, F, Filipovic, A, Grom, AA, Henter, JI, Ilowite, NT, Jordan, MB, Khubchandani, R, Kitoh, T, Lehmberg, K, Lovell, DJ, Miettunen, P, Nichols, KE, Ozen, S, Schmid, JP, Ramanan, AV, Russo, R, Schneider, R, Sterba, G, Uziel, Y, Wallace, C, Wouters, C, Wulffraat, N, Demirkaya, E, Brunner, HI, Martini, A, Ruperto, N, Cron, RQ, Angioloni, S, Pallotti, C, Pesce, M, Rinaldi, M, Villa, L, Abinun, M, Aggarwal, A, Akikusa, J, Al-Mayouf, SM, Alessio, M, Anton, J, Corona, F, Insalaco, A, Magni-Manzoni, S, on behalf of the Pediatric Rheumatology International Trials Organization, Childhood Arthritis & Rheumatology Research Alliance & Pediatric Rheumatology Collaborative Study Group and the Histiocyte Society 2016, 'Expert consensus on dynamics of laboratory tests for diagnosis of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis', RMD Open, vol. 2, no. 1, e000161. https://doi.org/10.1136/rmdopen-2015-000161
Ravelli, Angelo ; Minoia, Francesca ; Davì, Sergio ; Horne, Anna Carin ; Bovis, Francesca ; Pistorio, Angela ; Aricò, Maurizio ; Avcin, Tadej ; Behrens, Edward M. ; De Benedetti, Fabrizio ; Filipovic, Alexandra ; Grom, Alexei A. ; Henter, Jan Inge ; Ilowite, Norman T. ; Jordan, Michael B. ; Khubchandani, Raju ; Kitoh, Toshiyuki ; Lehmberg, Kai ; Lovell, Daniel J. ; Miettunen, Paivi ; Nichols, Kim E. ; Ozen, Seza ; Schmid, Jana Pachlopnik ; Ramanan, Athimalaipet V. ; Russo, Ricardo ; Schneider, Rayfel ; Sterba, Gary ; Uziel, Yosef ; Wallace, Carol ; Wouters, Carine ; Wulffraat, Nico ; Demirkaya, Erkan ; Brunner, Hermine I. ; Martini, Alberto ; Ruperto, Nicolino ; Cron, Randy Q. ; Angioloni, Simona ; Pallotti, Chiara ; Pesce, Michele ; Rinaldi, Mariangela ; Villa, Luca ; Abinun, Mario ; Aggarwal, Amita ; Akikusa, Jonathan ; Al-Mayouf, Sulaiman M. ; Alessio, Maria ; Anton, Jordi ; Corona, Fabrizia ; Insalaco, Antonella ; Magni-Manzoni, Silvia ; on behalf of the Pediatric Rheumatology International Trials Organization ; Childhood Arthritis & Rheumatology Research Alliance ; Pediatric Rheumatology Collaborative Study Group and the Histiocyte Society. / Expert consensus on dynamics of laboratory tests for diagnosis of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis. In: RMD Open. 2016 ; Vol. 2, No. 1.
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title = "Expert consensus on dynamics of laboratory tests for diagnosis of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis",
abstract = "Objective: To identify which laboratory tests that change over time are most valuable for the timely diagnosis of macrophage activation syndrome (MAS) complicating systemic juvenile idiopathic arthritis (sJIA). Methods: A multistep process, based on a combination of expert consensus and analysis of real patient data, was conducted. A panel of experts was first asked to evaluate 115 profiles of patients with MAS, which included the values of laboratory tests at the pre-MAS visit and at MAS onset, and the change in values between the two time points. The experts were asked to choose the 5 laboratory tests in which change was most important for the diagnosis of MAS and to rank the 5 selected tests in order of importance. The relevance of change in laboratory parameters was further discussed and ranked by the same experts at a consensus conference. Results: Platelet count was the most frequently selected test, followed by ferritin level, aspartate aminotransferase (AST), white cell count, neutrophil count, and fibrinogen and erythrocyte sedimentation rate. Ferritin was most frequently assigned the highest score. At the end of the process, platelet count, ferritin level and AST were the laboratory tests in which the experts found change over time to be most important. Conclusions: We identified the laboratory tests in which change over time is most valuable for the early diagnosis of MAS in sJIA. The dynamics of laboratory values during the course of MAS should be further scrutinised in a prospective study in order to establish the optimal cut-off values for their variation.",
author = "Angelo Ravelli and Francesca Minoia and Sergio Dav{\`i} and Horne, {Anna Carin} and Francesca Bovis and Angela Pistorio and Maurizio Aric{\`o} and Tadej Avcin and Behrens, {Edward M.} and {De Benedetti}, Fabrizio and Alexandra Filipovic and Grom, {Alexei A.} and Henter, {Jan Inge} and Ilowite, {Norman T.} and Jordan, {Michael B.} and Raju Khubchandani and Toshiyuki Kitoh and Kai Lehmberg and Lovell, {Daniel J.} and Paivi Miettunen and Nichols, {Kim E.} and Seza Ozen and Schmid, {Jana Pachlopnik} and Ramanan, {Athimalaipet V.} and Ricardo Russo and Rayfel Schneider and Gary Sterba and Yosef Uziel and Carol Wallace and Carine Wouters and Nico Wulffraat and Erkan Demirkaya and Brunner, {Hermine I.} and Alberto Martini and Nicolino Ruperto and Cron, {Randy Q.} and Simona Angioloni and Chiara Pallotti and Michele Pesce and Mariangela Rinaldi and Luca Villa and Mario Abinun and Amita Aggarwal and Jonathan Akikusa and Al-Mayouf, {Sulaiman M.} and Maria Alessio and Jordi Anton and Fabrizia Corona and Antonella Insalaco and Silvia Magni-Manzoni and {on behalf of the Pediatric Rheumatology International Trials Organization} and {Childhood Arthritis & Rheumatology Research Alliance} and {Pediatric Rheumatology Collaborative Study Group and the Histiocyte Society}",
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T1 - Expert consensus on dynamics of laboratory tests for diagnosis of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis

AU - Ravelli, Angelo

AU - Minoia, Francesca

AU - Davì, Sergio

AU - Horne, Anna Carin

AU - Bovis, Francesca

AU - Pistorio, Angela

AU - Aricò, Maurizio

AU - Avcin, Tadej

AU - Behrens, Edward M.

AU - De Benedetti, Fabrizio

AU - Filipovic, Alexandra

AU - Grom, Alexei A.

AU - Henter, Jan Inge

AU - Ilowite, Norman T.

AU - Jordan, Michael B.

AU - Khubchandani, Raju

AU - Kitoh, Toshiyuki

AU - Lehmberg, Kai

AU - Lovell, Daniel J.

AU - Miettunen, Paivi

AU - Nichols, Kim E.

AU - Ozen, Seza

AU - Schmid, Jana Pachlopnik

AU - Ramanan, Athimalaipet V.

AU - Russo, Ricardo

AU - Schneider, Rayfel

AU - Sterba, Gary

AU - Uziel, Yosef

AU - Wallace, Carol

AU - Wouters, Carine

AU - Wulffraat, Nico

AU - Demirkaya, Erkan

AU - Brunner, Hermine I.

AU - Martini, Alberto

AU - Ruperto, Nicolino

AU - Cron, Randy Q.

AU - Angioloni, Simona

AU - Pallotti, Chiara

AU - Pesce, Michele

AU - Rinaldi, Mariangela

AU - Villa, Luca

AU - Abinun, Mario

AU - Aggarwal, Amita

AU - Akikusa, Jonathan

AU - Al-Mayouf, Sulaiman M.

AU - Alessio, Maria

AU - Anton, Jordi

AU - Corona, Fabrizia

AU - Insalaco, Antonella

AU - Magni-Manzoni, Silvia

AU - on behalf of the Pediatric Rheumatology International Trials Organization

AU - Childhood Arthritis & Rheumatology Research Alliance

AU - Pediatric Rheumatology Collaborative Study Group and the Histiocyte Society

PY - 2016

Y1 - 2016

N2 - Objective: To identify which laboratory tests that change over time are most valuable for the timely diagnosis of macrophage activation syndrome (MAS) complicating systemic juvenile idiopathic arthritis (sJIA). Methods: A multistep process, based on a combination of expert consensus and analysis of real patient data, was conducted. A panel of experts was first asked to evaluate 115 profiles of patients with MAS, which included the values of laboratory tests at the pre-MAS visit and at MAS onset, and the change in values between the two time points. The experts were asked to choose the 5 laboratory tests in which change was most important for the diagnosis of MAS and to rank the 5 selected tests in order of importance. The relevance of change in laboratory parameters was further discussed and ranked by the same experts at a consensus conference. Results: Platelet count was the most frequently selected test, followed by ferritin level, aspartate aminotransferase (AST), white cell count, neutrophil count, and fibrinogen and erythrocyte sedimentation rate. Ferritin was most frequently assigned the highest score. At the end of the process, platelet count, ferritin level and AST were the laboratory tests in which the experts found change over time to be most important. Conclusions: We identified the laboratory tests in which change over time is most valuable for the early diagnosis of MAS in sJIA. The dynamics of laboratory values during the course of MAS should be further scrutinised in a prospective study in order to establish the optimal cut-off values for their variation.

AB - Objective: To identify which laboratory tests that change over time are most valuable for the timely diagnosis of macrophage activation syndrome (MAS) complicating systemic juvenile idiopathic arthritis (sJIA). Methods: A multistep process, based on a combination of expert consensus and analysis of real patient data, was conducted. A panel of experts was first asked to evaluate 115 profiles of patients with MAS, which included the values of laboratory tests at the pre-MAS visit and at MAS onset, and the change in values between the two time points. The experts were asked to choose the 5 laboratory tests in which change was most important for the diagnosis of MAS and to rank the 5 selected tests in order of importance. The relevance of change in laboratory parameters was further discussed and ranked by the same experts at a consensus conference. Results: Platelet count was the most frequently selected test, followed by ferritin level, aspartate aminotransferase (AST), white cell count, neutrophil count, and fibrinogen and erythrocyte sedimentation rate. Ferritin was most frequently assigned the highest score. At the end of the process, platelet count, ferritin level and AST were the laboratory tests in which the experts found change over time to be most important. Conclusions: We identified the laboratory tests in which change over time is most valuable for the early diagnosis of MAS in sJIA. The dynamics of laboratory values during the course of MAS should be further scrutinised in a prospective study in order to establish the optimal cut-off values for their variation.

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