American College of Rheumatology Provisional Criteria for Clinically Relevant Improvement in Children and Adolescents With Childhood-Onset Systemic Lupus Erythematosus

Brunner, H.I.; Holland, M.J.; Beresford, M.W.; Ardoin, S.P.; Appenzeller, S.; Silva, C.A.; Flores, F.; Goilav, B.; Avar Aydin, P.O.; Wenderfer, S.E.; Levy, D.M.; Ravelli, A.; Khubchandani, R.; Avcin, T.; Klein-Gitelman, M.S.; Ruperto, N.; Feldman, B.M.; Ying, J.; Battagliotti, C.; Brusco, M.I.; Cuttica, R.; De Cunto, C.; Espada, G.; Farfan, M.; Garay, S.; Marcantoni, M.; Marcela, A.; Meiorin, S.; Rama, M.E.; Russo, R.; Torre Walsh, C.; Zamparo, C.; Adib, N.; Akikusa, J.; Boros, C.; Lee, S.J.; Mckay, D.; Piper, S.; Joos, R.; Bica, B.; Campos, L.; Cavalcanti, A.; do Prado, R.; Donner-Maliki, A.; Fernandes, T.; Fonseca, A.; Gasparello de Almeida, R.; Guariento, A.; Gusman, C.; Jusan Fiorot, F.; Knupp Oliveira, S.; Len, C.; Arruda Campos, L.M.; Machado, S.; Marques, L.; Martins de Carvalho, L.; Moulin, R.; Pedroso, S.; Pileggi, G.; Romanelli, P.R.S.; Saad-Magalhaes, C.; Sakamoto, A.; Santos, M.C.; Silva, M.F.; Spelling, P.; Sztajnbok, S.; Terreri, M.; Cabral, D.; Chedeville, G.; Ellsworth, J.; Huber, A.; Tucker, L.; Houghton, K.; Borzutzky, A.; Ladino, M.; Norambuena, X.; Eraso, R.; Mosquera, A.; Velasquez, M.; Harjacek, M.; Jelusic, M.; Coto Hermosilla, C.; Dolezalova, P.; Nielsen, S.; Tineo, C.; Alegria, M.; Dressler, F.; Foell, D.; Ganser, G.; Hinze, C.; Hufnagel, M.; Lutz, T.; Trauzeddel, R.; Boiu, S.; Trachana, M.; Tsitsami, E.; Cifuentes, M.; Orban, I.; Aggarwal, A.; Sawhney, S.; Butbul Aviel, Y.; Cimaz, R.; Maggio, M.C.; Rumba-Rozenfelde, I.; Hashad, S.; Lim, S.C.; Abud, C.; Burgos-Vargas, R.; Carreno-Manjarrez, R.; Enciso Pelaez, S.; Hernandez-Huirache, H.; Maldonado Velazquez, R.; Orozco, J.; Rodriguez-Lozano, A.L.; Rojas Pacheco, O.E.; Suarez Larios, L.M.; Vega, G.; Ramirez Miramontes, J.V.; Ruiz Lopez, I.K.; Kamphuis, S.; Schonenberg-Meinema, D.; Concannon, A.; Yan, J.; Jarquin Jaime, M.; Al Abrawi, S.; Vega, C.; Lopez-Benitez, J.; Ibanez Estrella, A.; Miraval, T.; Dans, L.; Kimseng, K.J.; Opoka-Winiarska, V.; Rutkowska-Sak, L.; Smolewska, E.; Conde, M.; Guedes, M.; del Valle, E.; Quintero-Del Rio, A.; Ailioaie, C.; Sparchez, M.; Alekseeva, E.; Keltsev, V.; Al-Mayouf, S.; Asiri, A.; Suwairi, W.; Susic, G.; Vijatov-Djuric, G.; Ang, E.; Arkachaisri, T.; Boteanu, A.-.; Lopez-Robledillo, J.C.; Medrano San Ildefonso, M.; Modesto, C.; Calvo, I.; Sotoca-Fernandez, J.; Bolt, I.; Vilaiyuk, S.; Al-Abadi, E.; Baildam, E.; Fotis, L.; Pain, C.; Pilkington, C.; Abulaban, K.; Barbar-Smiley, F.; Binstadt, B.; Bohnsack, J.; Boneparth, A.; Brown, D.; Chira, P.; Cron, R.; Dedeoglu, F.; Eberhard, A.; Gedalia, A.; Grom, A.; Henrickson, M.; Hom, C.; Huggins, J.; Jerath, R.; Jones, J.; Jung, L.; Kingsbury, D.; Lai, J.; Lovell, D.; Nanda, K.; Nocton, J.; Olson, J.; O'Neil, K.; Onel, K.; Punaro, L.; Reiff, A.; Rouster-Stevens, K.; Ruth, N.; Schikler, K.; Murphy Schmidt, K.; Schulert, G.; Shaham, B.; Singer, N.; Smith, J.; Sundel, R.; Syverson, G.; Vega-Fernandez, P.; Vehe, R.; Wagner-Weiner, L.; Cameto, J.; Jurado, R.; Maldonado, I.

doi:10.1002/acr.23834

Objective: To develop a Childhood Lupus Improvement Index (CHILI) as a tool to measure response to therapy in childhood-onset systemic lupus erythematosus (cSLE), with a focus on clinically relevant improvement (CRI cSLE ). Methods: Pediatric nephrology and rheumatology subspecialists (n = 213) experienced in cSLE management were invited to define CRI cSLE and rate a total of 433 unique patient profiles for the presence/absence of CRI cSLE . Patient profiles included the following cSLE core response variables (CRVs): global assessment of patient well-being (patient-global), physician assessment of cSLE activity (MD-global), disease activity index score (here, we used the Systemic Lupus Erythematosus Disease Activity Index), urine protein-to-creatinine ratio, and Child Health Questionnaire physical summary score. Percentage and absolute changes in these cSLE-CRVs (baseline versus follow-up) were considered in order to develop candidate algorithms and validate their performance (sensitivity, specificity, area under the receiver operating characteristic curve [AUC]; range 0–1). Results: During an international consensus conference, unanimous agreement on a definition of CRI cSLE was achieved; cSLE experts (n = 13) concurred (100%) that the preferred CHILI algorithm considers absolute changes in the cSLE-CRVs. After transformation to a range of 0–100, a CHILI score of ≥54 had outstanding accuracy for identifying CRI cSLE (AUC 0.93, sensitivity 81.1%, and specificity 84.2%). CHILI scores also reflect minor, moderate, and major improvement for values exceeding 15, 68, and 92, respectively (all AUC ≥0.92, sensitivity ≥93.1%, and specificity ≥73.4%). Conclusion: The CHILI is a new, seemingly highly accurate index for measuring CRI in cSLE over time. This index is useful to categorize the degree of response to therapy in children and adolescents with cSLE.

American College of Rheumatology Provisional Criteria for Clinically Relevant Improvement in Children and Adolescents With Childhood-Onset Systemic Lupus Erythematosus / H.I. Brunner, M.J. Holland, M.W. Beresford, S.P. Ardoin, S. Appenzeller, C.A. Silva, F. Flores, B. Goilav, P.O. Avar Aydin, S.E. Wenderfer, D.M. Levy, A. Ravelli, R. Khubchandani, T. Avcin, M.S. Klein-Gitelman, N. Ruperto, B.M. Feldman, J. Ying, C. Battagliotti, M.I. Brusco, R. Cuttica, C. De Cunto, G. Espada, M. Farfan, S. Garay, M. Marcantoni, A. Marcela, S. Meiorin, M.E. Rama, R. Russo, C. Torre Walsh, C. Zamparo, N. Adib, J. Akikusa, C. Boros, S.J. Lee, D. Mckay, S. Piper, R. Joos, B. Bica, L. Campos, A. Cavalcanti, R. do Prado, A. Donner-Maliki, T. Fernandes, A. Fonseca, R. Gasparello de Almeida, A. Guariento, C. Gusman, F. Jusan Fiorot, S. Knupp Oliveira, C. Len, L.M. Arruda Campos, S. Machado, L. Marques, L. Martins de Carvalho, R. Moulin, S. Pedroso, G. Pileggi, P.R.S. Romanelli, C. Saad-Magalhaes, A. Sakamoto, M.C. Santos, M.F. Silva, P. Spelling, S. Sztajnbok, M. Terreri, D. Cabral, G. Chedeville, J. Ellsworth, A. Huber, L. Tucker, K. Houghton, A. Borzutzky, M. Ladino, X. Norambuena, R. Eraso, A. Mosquera, M. Velasquez, M. Harjacek, M. Jelusic, C. Coto Hermosilla, P. Dolezalova, S. Nielsen, C. Tineo, M. Alegria, F. Dressler, D. Foell, G. Ganser, C. Hinze, M. Hufnagel, T. Lutz, R. Trauzeddel, S. Boiu, M. Trachana, E. Tsitsami, M. Cifuentes, I. Orban, A. Aggarwal, S. Sawhney, Y. Butbul Aviel, R. Cimaz, M.C. Maggio, I. Rumba-Rozenfelde, S. Hashad, S.C. Lim, C. Abud, R. Burgos-Vargas, R. Carreno-Manjarrez, S. Enciso Pelaez, H. Hernandez-Huirache, R. Maldonado Velazquez, J. Orozco, A.L. Rodriguez-Lozano, O.E. Rojas Pacheco, L.M. Suarez Larios, G. Vega, J.V. Ramirez Miramontes, I.K. Ruiz Lopez, S. Kamphuis, D. Schonenberg-Meinema, A. Concannon, J. Yan, M. Jarquin Jaime, S. Al Abrawi, C. Vega, J. Lopez-Benitez, A. Ibanez Estrella, T. Miraval, L. Dans, K.J. Kimseng, V. Opoka-Winiarska, L. Rutkowska-Sak, E. Smolewska, M. Conde, M. Guedes, E. del Valle, A. Quintero-Del Rio, C. Ailioaie, M. Sparchez, E. Alekseeva, V. Keltsev, S. Al-Mayouf, A. Asiri, W. Suwairi, G. Susic, G. Vijatov-Djuric, E. Ang, T. Arkachaisri, A.-. Boteanu, J.C. Lopez-Robledillo, M. Medrano San Ildefonso, C. Modesto, I. Calvo, J. Sotoca-Fernandez, I. Bolt, S. Vilaiyuk, E. Al-Abadi, E. Baildam, L. Fotis, C. Pain, C. Pilkington, K. Abulaban, F. Barbar-Smiley, B. Binstadt, J. Bohnsack, A. Boneparth, D. Brown, P. Chira, R. Cron, F. Dedeoglu, A. Eberhard, A. Gedalia, A. Grom, M. Henrickson, C. Hom, J. Huggins, R. Jerath, J. Jones, L. Jung, D. Kingsbury, J. Lai, D. Lovell, K. Nanda, J. Nocton, J. Olson, K. O'Neil, K. Onel, L. Punaro, A. Reiff, K. Rouster-Stevens, N. Ruth, K. Schikler, K. Murphy Schmidt, G. Schulert, B. Shaham, N. Singer, J. Smith, R. Sundel, G. Syverson, P. Vega-Fernandez, R. Vehe, L. Wagner-Weiner, J. Cameto, R. Jurado, I. Maldonado. - In: ARTHRITIS CARE & RESEARCH. - ISSN 2151-464X. - 71:5(2019), pp. 579-590. [10.1002/acr.23834]

American College of Rheumatology Provisional Criteria for Clinically Relevant Improvement in Children and Adolescents With Childhood-Onset Systemic Lupus Erythematosus

Brunner H. I.;Holland M. J.;Beresford M. W.;Ardoin S. P.;Appenzeller S.;Silva C. A.;Flores F.;Goilav B.;Avar Aydin P. O.;Wenderfer S. E.;Levy D. M.;Ravelli A.;Khubchandani R.;Avcin T.;Klein-Gitelman M. S.;Ruperto N.;Feldman B. M.;Ying J.;Battagliotti C.;Brusco M. I.;Cuttica R.;De Cunto C.;Espada G.;Farfan M.;Garay S.;Marcantoni M.;Marcela A.;Meiorin S.;Rama M. E.;Russo R.;Torre Walsh C.;Zamparo C.;Adib N.;Akikusa J.;Boros C.;Lee S. J.;Mckay D.;Piper S.;Joos R.;Bica B.;Campos L.;Cavalcanti A.;do Prado R.;Donner-Maliki A.;Fernandes T.;Fonseca A.;Gasparello de Almeida R.;Guariento A.;Gusman C.;Jusan Fiorot F.;Knupp Oliveira S.;Len C.;Arruda Campos L. M.;Machado S.;Marques L.;Martins de Carvalho L.;Moulin R.;Pedroso S.;Pileggi G.;Romanelli P. R. S.;Saad-Magalhaes C.;Sakamoto A.;Santos M. C.;Silva M. F.;Spelling P.;Sztajnbok S.;Terreri M.;Cabral D.;Chedeville G.;Ellsworth J.;Huber A.;Tucker L.;Houghton K.;Borzutzky A.;Ladino M.;Norambuena X.;Eraso R.;Mosquera A.;Velasquez M.;Harjacek M.;Jelusic M.;Coto Hermosilla C.;Dolezalova P.;Nielsen S.;Tineo C.;Alegria M.;Dressler F.;Foell D.;Ganser G.;Hinze C.;Hufnagel M.;Lutz T.;Trauzeddel R.;Boiu S.;Trachana M.;Tsitsami E.;Cifuentes M.;Orban I.;Aggarwal A.;Sawhney S.;Butbul Aviel Y.;R. Cimaz;Maggio M. C.;Rumba-Rozenfelde I.;Hashad S.;Lim S. C.;Abud C.;Burgos-Vargas R.;Carreno-Manjarrez R.;Enciso Pelaez S.;Hernandez-Huirache H.;Maldonado Velazquez R.;Orozco J.;Rodriguez-Lozano A. L.;Rojas Pacheco O. E.;Suarez Larios L. M.;Vega G.;Ramirez Miramontes J. V.;Ruiz Lopez I. K.;Kamphuis S.;Schonenberg-Meinema D.;Concannon A.;Yan J.;Jarquin Jaime M.;Al Abrawi S.;Vega C.;Lopez-Benitez J.;Ibanez Estrella A.;Miraval T.;Dans L.;Kimseng K. J.;Opoka-Winiarska V.;Rutkowska-Sak L.;Smolewska E.;Conde M.;Guedes M.;del Valle E.;Quintero-Del Rio A.;Ailioaie C.;Sparchez M.;Alekseeva E.;Keltsev V.;Al-Mayouf S.;Asiri A.;Suwairi W.;Susic G.;Vijatov-Djuric G.;Ang E.;Arkachaisri T.;Boteanu A. -L.;Lopez-Robledillo J. C.;Medrano San Ildefonso M.;Modesto C.;Calvo I.;Sotoca-Fernandez J.;Bolt I.;Vilaiyuk S.;Al-Abadi E.;Baildam E.;Fotis L.;Pain C.;Pilkington C.;Abulaban K.;Barbar-Smiley F.;Binstadt B.;Bohnsack J.;Boneparth A.;Brown D.;Chira P.;Cron R.;Dedeoglu F.;Eberhard A.;Gedalia A.;Grom A.;Henrickson M.;Hom C.;Huggins J.;Jerath R.;Jones J.;Jung L.;Kingsbury D.;Lai J.;Lovell D.;Nanda K.;Nocton J.;Olson J.;O'Neil K.;Onel K.;Punaro L.;Reiff A.;Rouster-Stevens K.;Ruth N.;Schikler K.;Murphy Schmidt K.;Schulert G.;Shaham B.;Singer N.;Smith J.;Sundel R.;Syverson G.;Vega-Fernandez P.;Vehe R.;Wagner-Weiner L.;Cameto J.;Jurado R.;Maldonado I.

2019

Abstract

Objective: To develop a Childhood Lupus Improvement Index (CHILI) as a tool to measure response to therapy in childhood-onset systemic lupus erythematosus (cSLE), with a focus on clinically relevant improvement (CRI cSLE ). Methods: Pediatric nephrology and rheumatology subspecialists (n = 213) experienced in cSLE management were invited to define CRI cSLE and rate a total of 433 unique patient profiles for the presence/absence of CRI cSLE . Patient profiles included the following cSLE core response variables (CRVs): global assessment of patient well-being (patient-global), physician assessment of cSLE activity (MD-global), disease activity index score (here, we used the Systemic Lupus Erythematosus Disease Activity Index), urine protein-to-creatinine ratio, and Child Health Questionnaire physical summary score. Percentage and absolute changes in these cSLE-CRVs (baseline versus follow-up) were considered in order to develop candidate algorithms and validate their performance (sensitivity, specificity, area under the receiver operating characteristic curve [AUC]; range 0–1). Results: During an international consensus conference, unanimous agreement on a definition of CRI cSLE was achieved; cSLE experts (n = 13) concurred (100%) that the preferred CHILI algorithm considers absolute changes in the cSLE-CRVs. After transformation to a range of 0–100, a CHILI score of ≥54 had outstanding accuracy for identifying CRI cSLE (AUC 0.93, sensitivity 81.1%, and specificity 84.2%). CHILI scores also reflect minor, moderate, and major improvement for values exceeding 15, 68, and 92, respectively (all AUC ≥0.92, sensitivity ≥93.1%, and specificity ≥73.4%). Conclusion: The CHILI is a new, seemingly highly accurate index for measuring CRI in cSLE over time. This index is useful to categorize the degree of response to therapy in children and adolescents with cSLE.

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	Settori scientifico-disciplinari dell'articolo
	
			Settore MED/16 - Reumatologia
Settore MED/38 - Pediatria Generale e Specialistica
		
	Data di pubblicazione
	
			2019
		
	Rivista in ANCE
	
			ARTHRITIS CARE & RESEARCH
		
	DOI
	
			https://dx.doi.org/10.1002/acr.23834
		
	Tipologia
	
			Article (author)
		
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			01 - Articolo su periodico

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