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1. Clinical features, treatment and outcomes of Italian children with enthesitis related arthritis and juvenile psoriatic arthritis: a cross-sectional cohort study

Author

Rebollo-Giménez, Ai, Rosina, S, Natoli, V, Burrone, M, Barone, P, Campus, S, Civino, A, Consolini, R, Filocamo, G, Gallizzi, R, Gattinara, M, La Torre, F, Maggio, Mc, Magni-Manzoni, S, Magnolia, Mg, Miniaci, A, Montin, D, Pastore, S, Petaccia, A, Pieropan, S, Rigante, Donato, Simonini, G, Ridella, F, Orsi, Sm, Angioloni, S, Carlini, L, Ruperto, N, Consolaro, A, Rigante D (ORCID:0000-0001-7032-7779), Rebollo-Giménez, Ai, Rosina, S, Natoli, V, Burrone, M, Barone, P, Campus, S, Civino, A, Consolini, R, Filocamo, G, Gallizzi, R, Gattinara, M, La Torre, F, Maggio, Mc, Magni-Manzoni, S, Magnolia, Mg, Miniaci, A, Montin, D, Pastore, S, Petaccia, A, Pieropan, S, Rigante, Donato, Simonini, G, Ridella, F, Orsi, Sm, Angioloni, S, Carlini, L, Ruperto, N, Consolaro, A, and Rigante D (ORCID:0000-0001-7032-7779)

Abstract

Objectives: Limited information is available on the clinical features, treatment modalities and outcomes of the juvenile idiopathic arthritis (JIA) categories of enthesitis-related arthritis (ERA) and juvenile psoriatic arthritis (JPsA). This study was aimed to describe the characteristics of Italian children with ERA and JPsA and to compare them with those of patients with the other categories of JIA. Methods: Patients were part of a multinational sample included in a study aimed to investigate the prevalence of disease categories, treatment approaches, and disease status in patients from across different geographical areas (EPOCA Study). All patients underwent a retrospective assessment, based on the review of clinical chart, and a cross-sectional evaluation, which included assessment of physician- and parent-reported outcomes and laboratory tests, and recording of ongoing therapies. Results: Of the 9081 children with JIA enrolled in the EPOCA Study, 1300 were recruited at 18 paediatric rheumatology centres in Italy. 45 (3.5%) had ERA and 49 (3.8%) had JPsA. Several remarkable differences in demographic features and frequency of articular and extra-articular manifestations, disease damage, impairment in physical function and health-related quality of life, school-related problems, comorbidities, and ongoing treatments were observed between ERA and JPsA and the other JIA categories. Conclusions: We described the characteristics of Italian children with ERA and JPsA and highlighted their peculiarities and their differences from the other JIA subsets. These data provide useful insights for future revisions of JIA classification and a benchmarking against which the features from other cohorts may be compared.

Published
2024

2. Opportunistic infections in immunosuppressed patients with juvenile idiopathic arthritis: analysis by the Pharmachild Safety Adjudication Committee

Author

Giancane G1, Swart JF2, Castagnola E3, Groll AH4, Horneff G5, 6, Huppertz HI7, Lovell DJ8, Wolfs T2, Herlin T9, Dolezalova P10, Sanner H11, Susic G13, Sztajnbok F14, Maritsi D15, Constantin T16, Vargova V17, Sawhney S18, Rygg M19, K Oliveira S21, Cattalini M22, Bovis F1, Bagnasco F1, Pistorio A23, Martini A24, Wulffraat N2, Ruperto N25, Paediatric Rheumatology International Trials Organisation (PRINTO). Cuttica R, Garay SM, Brunner J, Emminger W, Appenzeller S, Len C, Saad Magalhaes C, Telcharova-Mihaylovska A, Harjacek M, Jelusic M, Estmann A, Nielsen S, Herrera Mora C, Gervais E, Koné-Paut I, Quartier P, Foeldvari I, Horneff G, Lutz T, Minden K, Tzaribachev N, Trachana M, Tsitsami E, Vougiouka O, Orban I, Harel L, Hashkes P, Uziel Y, Cimaz R, Civino A, Consolini R, D'Angelo G, De Benedetti F, Filocamo G, Fueri E, Gallizzi R, Maggio MC, Magnolia MG, Miniaci A, Montin D, Olivieri A. N., Pastore S, Rigante D, Zulian F, Rumba-Rozenfelde I, Stanevicha V, Panaviene V, Rodriguez Lozano AL, Rubio-Perez N, Vega Cornejo G, Hoppenreijs E, Kamphuis S, Flato B, Nordal EB, Abdwani R, Miraval T, Paz Gastanaga ME, Smolewska E, Ailioaie C, Cochino AV, Laday M, Lazar C, Alexeeva E, Chasnyk V, Keltsev V, Suwairi WMS, Vijatov-Djuric G, Vojinovic J, Arkachaisri T, Koskova E, Avcin T, Ally M, Van Rensburg CJ, Louw I, Lopez JA, Boteanu AL, Calvo Penades I, De Inocencio J, Mesa-Del-Castillo P, Moreno E, Remesal A, Hofer M, Gok F, Ozen S, Ramanan A, Pallotti C, Villa L., Giancane, G1, Swart, Jf2, Castagnola, E3, Groll, Ah4, Horneff, G5, Huppertz, Hi7, Lovell, Dj8, Wolfs, T2, Herlin, T9, Dolezalova, P10, Sanner, H11, Susic, G13, Sztajnbok, F14, Maritsi, D15, Constantin, T16, Vargova, V17, Sawhney, S18, Rygg, M19, K Oliveira, S21, Cattalini, M22, Bovis, F1, Bagnasco, F1, Pistorio, A23, Martini, A24, Wulffraat, N2, Ruperto, N25, Paediatric Rheumatology International Trials Organisation (PRINTO)., Cuttica R, Garay, Sm, Brunner, J, Emminger, W, Appenzeller, S, Len, C, Saad Magalhaes, C, Telcharova-Mihaylovska, A, Harjacek, M, Jelusic, M, Estmann, A, Nielsen, S, Herrera Mora, C, Gervais, E, Koné-Paut, I, Quartier, P, Foeldvari, I, Horneff, G, Lutz, T, Minden, K, Tzaribachev, N, Trachana, M, Tsitsami, E, Vougiouka, O, Orban, I, Harel, L, Hashkes, P, Uziel, Y, Cimaz, R, Civino, A, Consolini, R, D'Angelo, G, De Benedetti, F, Filocamo, G, Fueri, E, Gallizzi, R, Maggio, Mc, Magnolia, Mg, Miniaci, A, Montin, D, Olivieri, A. N., Pastore, S, Rigante, D, Zulian, F, Rumba-Rozenfelde, I, Stanevicha, V, Panaviene, V, Rodriguez Lozano, Al, Rubio-Perez, N, Vega Cornejo, G, Hoppenreijs, E, Kamphuis, S, Flato, B, Nordal, Eb, Abdwani, R, Miraval, T, Paz Gastanaga, Me, Smolewska, E, Ailioaie, C, Cochino, Av, Laday, M, Lazar, C, Alexeeva, E, Chasnyk, V, Keltsev, V, Suwairi, Wm, Vijatov-Djuric, G, Vojinovic, J, Arkachaisri, T, Koskova, E, Avcin, T, Ally, M, Van Rensburg, Cj, Louw, I, Lopez, Ja, Boteanu, Al, Calvo Penades, I, De Inocencio, J, Mesa-Del-Castillo, P, Moreno, E, Remesal, A, Hofer, M, Gok, F, Ozen, S, Ramanan, A, Pallotti, C, Villa, L., Giancane, Gabriella, Swart, Joost F, Castagnola, Elio, Groll, Andreas H, Horneff, Gerd, Huppertz, Hans-Iko, Lovell, Daniel J, Wolfs, Tom, Herlin, Troel, Dolezalova, Pavla, Sanner, Helga, Susic, Gordana, Sztajnbok, Flavio, Maritsi, Despoina, Constantin, Tama, Vargova, Veronika, Sawhney, Sujata, Rygg, Marite, K Oliveira, Sheila, Cattalini, Marco, Bovis, Francesca, Bagnasco, Francesca, Pistorio, Angela, Martini, Alberto, Wulffraat, Nico, Ruperto, Nicolino, Cuttica, Ruben, Garay, Stella Mari, Brunner, Jurgen, Emminger, Wolfgang, Appenzeller, Simone, Len, Claudio, Saad Magalhaes, Claudia, Telcharova-Mihaylovska, Albena, Harjacek, Miroslav, Jelusic, Marija, Estmann, Anne, Nielsen, Susan, Herrera Mora, Cristina, Gervais, Elisabeth, Koné-Paut, Isabelle, Quartier, Pierre, Foeldvari, Ivan, Lutz, Thoma, Minden, Kirsten, Tzaribachev, Nikolay, Trachana, Maria, Tsitsami, Elena, Vougiouka, Olga, Orban, Ilonka, Harel, Liora, Hashkes, Philip, Uziel, Yosef, Cimaz, Rolando, Civino, Adele, Consolini, Rita, D'Angelo, Gianfranco, De Benedetti, Fabrizio, Filocamo, Giovanni, Fueri, Elena, Gallizzi, Romina, Maggio, Maria Cristina, Magnolia, Maria Greca, Miniaci, Angela, Montin, Davide, Olivieri, Alma Nunzia, Pastore, Serena, Rigante, Donato, Zulian, Francesco, Rumba-Rozenfelde, Ingrida, Stanevicha, Valda, Panaviene, Violeta, Rodriguez Lozano, Ana Luisa, Rubio-Perez, Nadina, Vega Cornejo, Gabriel, Hoppenreijs, Esther, Kamphuis, Sylvia, Flato, Berit, Nordal, Ellen Berit, Abdwani, Reem, Miraval, Tatiana, Paz Gastanaga, Maria Eliana, Smolewska, Elzbieta, Ailioaie, Constantin, Cochino, Alexis-Virgil, Laday, Matilda, Lazar, Calin, Alexeeva, Ekaterina, Chasnyk, Vyacheslav, Keltsev, Vladimir, Suwairi, Wafaa Mohammed Saad, Vijatov-Djuric, Gordana, Vojinovic, Jelena, Arkachaisri, Thaschawee, Koskova, Elena, Avcin, Tadej, Ally, Mahmood, Van Rensburg, Christa Janse, Louw, Ingrid, Lopez, Jordi Anton, Boteanu, Alina Lucica, Calvo Penades, Inmaculada, De Inocencio, Jaime, Mesa-Del-Castillo, Pablo, Moreno, Estefania, Remesal, Agustin, Hofer, Michael, Gok, Faysal, Ozen, Seza, Ramanan, Athimalaipet, Pallotti, Chiara, Villa, Luca, and Pediatrics

Subjects
Male, lcsh:Diseases of the musculoskeletal system, Biologic, Paediatrics: 760 [VDP], Artritis infecciosa, MedDRA, Infants malalts, Arthritis, Severity of Illness Index, Hospital patients, Cohort Studies, Pharmacovigilance, 0302 clinical medicine, 030212 general & internal medicine, Registries, Child, Biologics, Immunosuppressive therapy, Infections, Juvenile idiopathic arthritis, Opportunistic, biologics, immunosuppressive therapy, infections, juvenile idiopathic arthritis, opportunistic, Barneleddgikt, 3. Good health, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Antirheumatic Agents, Child, Preschool, Cohort, Pediatric Infectious Disease, Female, Infection, Research Article, medicine.medical_specialty, Tuberculosis, juvenil idiopathic arthritis, Opportunistic Infections, Herpes Zoster, 03 medical and health sciences, Immunocompromised Host, Juvenile idiopathic arthriti, Internal medicine, medicine, Humans, book, 030203 arthritis & rheumatology, Malalts hospitalitzats, Immunosupressió, business.industry, Sick children, medicine.disease, Rheumatology, Arthritis, Juvenile, Infectious arthritis, Pediatri: 760 [VDP], Orthopedic surgery, Opportunistiske infeksjoner, book.journal, lcsh:RC925-935, business, Infeccions oportunistes, Immunosuppression
Abstract

Background: To derive a list of opportunistic infections (OI) through the analysis of the juvenile idiopathic arthritis (JIA) patients in the Pharmachild registry by an independent Safety Adjudication Committee (SAC). Methods: The SAC (3 pediatric rheumatologists and 2 pediatric infectious disease specialists) elaborated and approved by consensus a provisional list of OI for use in JIA. Through a 5 step-procedure, all the severe and serious infections, classified as per MedDRA dictionary and retrieved in the Pharmachild registry, were evaluated by the SAC by answering six questions and adjudicated with the agreement of 3/5 specialists. A final evidence-based list of OI resulted by matching the adjudicated infections with the provisional list of OI. Results: A total of 772 infectious events in 572 eligible patients, of which 335 serious/severe/very severe non-OI and 437 OI (any intensity/severity), according to the provisional list, were retrieved. Six hundred eighty-two of 772 (88.3%) were adjudicated as infections, of them 603/682 (88.4%) as common and 119/682 (17.4%) as OI by the SAC. Matching these 119 opportunistic events with the provisional list, 106 were confirmed by the SAC as OI, and among them infections by herpes viruses were the most frequent (68%), followed by tuberculosis (27.4%). The remaining events were divided in the groups of non-OI and possible/patient and/or pathogen-related OI. Conclusions: We found a significant number of OI in JIA patients on immunosuppressive therapy. The proposed list of OI, created by consensus and validated in the Pharmachild cohort, could facilitate comparison among future pharmacovigilance studies. © The Author(s). 2020 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

Published
2020
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3. Persistence of disease flares is associated with an inadequate colchicine dose in familial Mediterranean fever: A national multicenter longitudinal study

Author

Bustaffa, M, Mazza, F, Sutera, D, Carrabba, M, Alessio, M, Cantarini, L, Obici, L, Rigante, D, Maggio, M, Insalaco, A, Simonini, G, Cattalini, M, Conti, G, Olivieri, A, Barone, P, Miniaci, A, Moressa, V, Magnolia, M, Breda, L, Montin, D, Spagnolo, A, Fabio, G, Orlando, F, Gaggiano, C, Mussinelli, R, Capozio, G, Celani, C, Marrani, E, Ricci, F, Calzatini, F, Lancieri, M, Ruperto, N, Gattorno, M, Gallizzi, R, Bustaffa M, Mazza F, Sutera D, Carrabba MD, Alessio M, Cantarini L, Obici L, Rigante D, Maggio MC, Insalaco A, Simonini G, Cattalini M, Conti G, Olivieri AN, Barone P, Miniaci A, Moressa V, Magnolia MG, Breda L, Montin D, Spagnolo A, Fabio G, Orlando F, Gaggiano C, Mussinelli R, Capozio G, Celani C, Marrani E, Ricci F, Calzatini F, Lancieri M, Ruperto N, Gattorno M, Gallizzi R, Bustaffa, M, Mazza, F, Sutera, D, Carrabba, M, Alessio, M, Cantarini, L, Obici, L, Rigante, D, Maggio, M, Insalaco, A, Simonini, G, Cattalini, M, Conti, G, Olivieri, A, Barone, P, Miniaci, A, Moressa, V, Magnolia, M, Breda, L, Montin, D, Spagnolo, A, Fabio, G, Orlando, F, Gaggiano, C, Mussinelli, R, Capozio, G, Celani, C, Marrani, E, Ricci, F, Calzatini, F, Lancieri, M, Ruperto, N, Gattorno, M, Gallizzi, R, Bustaffa M, Mazza F, Sutera D, Carrabba MD, Alessio M, Cantarini L, Obici L, Rigante D, Maggio MC, Insalaco A, Simonini G, Cattalini M, Conti G, Olivieri AN, Barone P, Miniaci A, Moressa V, Magnolia MG, Breda L, Montin D, Spagnolo A, Fabio G, Orlando F, Gaggiano C, Mussinelli R, Capozio G, Celani C, Marrani E, Ricci F, Calzatini F, Lancieri M, Ruperto N, Gattorno M, and Gallizzi R

Published
2021

4. Persistence of disease flares is associated to an inadequate colchicine dose in familial Mediterranean fever: a national multicentre longitudinal study

Author

Bustaffa, M, Mazza, F, Sutera, D, Carrabba, Md, Alessio, M, Cantarini, L, Obici, L, Rigante, D, Maggio, Mc, Insalaco A., Simonini G, Cattalini, M, Conti, G, Olivieri, An, Barone, P, Miniaci, A, Moressa, V, Magnolia, Mg, Breda, L, Montin, D, Spagnolo, A, Fabio, G, Orlando, F, Gaggiano, C, Mussinelli, R, Capozio, G, Celani, C, Marrani, E, Ricci, F, Calzatini, F, Lancieri, M, Ruperto, N, Gattorno, M, Gallizzi, R, Rigante D (ORCID:0000-0001-7032-7779), Conti G (ORCID:0000-0002-8566-9365), Bustaffa, M, Mazza, F, Sutera, D, Carrabba, Md, Alessio, M, Cantarini, L, Obici, L, Rigante, D, Maggio, Mc, Insalaco A., Simonini G, Cattalini, M, Conti, G, Olivieri, An, Barone, P, Miniaci, A, Moressa, V, Magnolia, Mg, Breda, L, Montin, D, Spagnolo, A, Fabio, G, Orlando, F, Gaggiano, C, Mussinelli, R, Capozio, G, Celani, C, Marrani, E, Ricci, F, Calzatini, F, Lancieri, M, Ruperto, N, Gattorno, M, Gallizzi, R, Rigante D (ORCID:0000-0001-7032-7779), and Conti G (ORCID:0000-0002-8566-9365)

Abstract

This study aimed at identifying the oersistence od disease in patients with FMF treated with colchicine. Almost 30% of FMF patients display a persistent disease activity not fulfilling the definition of colchicine resistance, but impacting on their quality of life. In most of these patients colchicine is under-dosed and maximum recommended dose is rarely used.

Published
2021

5. Opportunistic infections in immunosuppressed patients with juvenile idiopathic arthritis: analysis by the Pharmachild Safety Adjudication Committee

Author

Giancane, G, Swart, Jf, Castagnola, E, Groll, Ah, Horneff, G, Huppertz, Hi, Lovell, Dj, Wolfs, T, Herlin, T, Dolezalova, P, Sanner, H, Susic, G, Sztajnbok, F, Maritsi, D, Constantin, T, Vargova, V, Sawhney, S, Rygg, M, K Oliveira, S, Cattalini, M, Bovis, F, Bagnasco, F, Pistorio, A, Martini, A, Wulffraat, N, Ruperto, N, Cuttica, R, Garay, Sm, Brunner, J, Emminger, W, Appenzeller, S, Len, C, Saad Magalhaes, C, Telcharova-Mihaylovska, A, Harjacek, M, Jelusic, M, Estmann, A, Nielsen, S, Herrera Mora, C, Gervais, E, Koné-Paut, I, Quartier, P, Foeldvari, I, Lutz, T, Minden, K, Tzaribachev, N, Trachana, M, Tsitsami, E, Vougiouka, O, Orban, I, Harel, L, Hashkes, P, Uziel, Y, Cimaz, R, Civino, A, Consolini, R, D'Angelo, G, De Benedetti, F, Filocamo, G, Fueri, E, Gallizzi, R, Maggio, Mc, Magnolia, Mg, Miniaci, A, Montin, D, Olivieri, An, Pastore, S, Rigante, Donato, Zulian, F, Rumba-Rozenfelde, I, Stanevicha, V, Panaviene, V, Rodriguez Lozano, Al, Rubio-Perez, N, Vega Cornejo, G, Hoppenreijs, E, Kamphuis, S, Flato, B, Nordal, Eb, Abdwani, R, Miraval, T, Paz Gastanaga, Me, Smolewska, E, Ailioaie, C, Cochino, Av, Laday, M, Lazar, C, Alexeeva, E, Chasnyk, V, Keltsev, V, Suwairi, Wm, Vijatov-Djuric, G, Vojinovic, J, Arkachaisri, T, Koskova, E, Avcin, T, Ally, M, Van Rensburg, Cj, Louw, I, Lopez, Ja, Boteanu, Al, Calvo Penades, I, De Inocencio, J, Mesa-Del-Castillo, P, Moreno, E, Remesal, A, Hofer, M, Gok, F, Ozen, S, Ramanan, A, Pallotti, C, Villa, L &amp, Paediatric Rheumatology International Trials Organisation, (PRINTO), Rigante D (ORCID:0000-0001-7032-7779), Giancane, G, Swart, Jf, Castagnola, E, Groll, Ah, Horneff, G, Huppertz, Hi, Lovell, Dj, Wolfs, T, Herlin, T, Dolezalova, P, Sanner, H, Susic, G, Sztajnbok, F, Maritsi, D, Constantin, T, Vargova, V, Sawhney, S, Rygg, M, K Oliveira, S, Cattalini, M, Bovis, F, Bagnasco, F, Pistorio, A, Martini, A, Wulffraat, N, Ruperto, N, Cuttica, R, Garay, Sm, Brunner, J, Emminger, W, Appenzeller, S, Len, C, Saad Magalhaes, C, Telcharova-Mihaylovska, A, Harjacek, M, Jelusic, M, Estmann, A, Nielsen, S, Herrera Mora, C, Gervais, E, Koné-Paut, I, Quartier, P, Foeldvari, I, Lutz, T, Minden, K, Tzaribachev, N, Trachana, M, Tsitsami, E, Vougiouka, O, Orban, I, Harel, L, Hashkes, P, Uziel, Y, Cimaz, R, Civino, A, Consolini, R, D'Angelo, G, De Benedetti, F, Filocamo, G, Fueri, E, Gallizzi, R, Maggio, Mc, Magnolia, Mg, Miniaci, A, Montin, D, Olivieri, An, Pastore, S, Rigante, Donato, Zulian, F, Rumba-Rozenfelde, I, Stanevicha, V, Panaviene, V, Rodriguez Lozano, Al, Rubio-Perez, N, Vega Cornejo, G, Hoppenreijs, E, Kamphuis, S, Flato, B, Nordal, Eb, Abdwani, R, Miraval, T, Paz Gastanaga, Me, Smolewska, E, Ailioaie, C, Cochino, Av, Laday, M, Lazar, C, Alexeeva, E, Chasnyk, V, Keltsev, V, Suwairi, Wm, Vijatov-Djuric, G, Vojinovic, J, Arkachaisri, T, Koskova, E, Avcin, T, Ally, M, Van Rensburg, Cj, Louw, I, Lopez, Ja, Boteanu, Al, Calvo Penades, I, De Inocencio, J, Mesa-Del-Castillo, P, Moreno, E, Remesal, A, Hofer, M, Gok, F, Ozen, S, Ramanan, A, Pallotti, C, Villa, L &amp, Paediatric Rheumatology International Trials Organisation, (PRINTO), and Rigante D (ORCID:0000-0001-7032-7779)

Abstract

BACKGROUND: To derive a list of opportunistic infections (OI) through the analysis of the juvenile idiopathic arthritis (JIA) patients in the Pharmachild registry by an independent Safety Adjudication Committee (SAC). METHODS: The SAC (3 pediatric rheumatologists and 2 pediatric infectious disease specialists) elaborated and approved by consensus a provisional list of OI for use in JIA. Through a 5 step-procedure, all the severe and serious infections, classified as per MedDRA dictionary and retrieved in the Pharmachild registry, were evaluated by the SAC by answering six questions and adjudicated with the agreement of 3/5 specialists. A final evidence-based list of OI resulted by matching the adjudicated infections with the provisional list of OI. RESULTS: A total of 772 infectious events in 572 eligible patients, of which 335 serious/severe/very severe non-OI and 437 OI (any intensity/severity), according to the provisional list, were retrieved. Six hundred eighty-two of 772 (88.3%) were adjudicated as infections, of them 603/682 (88.4%) as common and 119/682 (17.4%) as OI by the SAC. Matching these 119 opportunistic events with the provisional list, 106 were confirmed by the SAC as OI, and among them infections by herpes viruses were the most frequent (68%), followed by tuberculosis (27.4%). The remaining events were divided in the groups of non-OI and possible/patient and/or pathogen-related OI. CONCLUSIONS: We found a significant number of OI in JIA patients on immunosuppressive therapy. The proposed list of OI, created by consensus and validated in the Pharmachild cohort, could facilitate comparison among future pharmacovigilance studies.

Published
2020

6. Phenotypic variability and disparities in treatment and outcomes of childhood arthritis throughout the world: an observational cohort study

Author

Consolaro, A, Giancane, G, Alongi, A, van Dijkhuizen, Ehp, Aggarwal, A, Al-Mayouf, Sm, Bovis, F, De Inocencio, J, Demirkaya, E, Flato, B, Foell, D, Garay, Sm, Lazăr, C, Lovell, Dj, Montobbio, C, Miettunen, P, Mihaylova, D, Nielsen, S, Orban, I, Rumba-Rozenfelde, I, Magalhães, C, Shafaie, N, Susic, G, Trachana, M, Wulffraat, N, Pistorio, A, Martini, A, Ruperto, N, Ravelli, A &amp, Abdwani R, Aghighi, Y, Aiche, Mf, Ailioaie, C, Aktay Ayaz, N, Al-Abrawi, S, Alexeeva, E, Anton, J, Apostol, A, Arguedas, O, Avcin, T, Barone, P, Berntson, L, Boteanu, Al, Boyko, Y, Burgos-Vargas, R, Calvo Penades, I, Chédeville, G, Cimaz, R, Civino, A, Consolini, R, Constantin, T, Cuttica, R, Dallos, T, Martin, N, Magni-Manzoni, S, De Cunto, C, Dolezalova, P, Ekelund, M, El Miedany, Y, Espada, G, Estmann Christensen, A, Foeldvari, I, Gallizzi, R, Ganser, G, Gerloni, V, Haas, Jp, Harel, L, Harjacek, M, Hashad, S, Herlin, T, Herrera, C, Hofer, M, Holzinger, D, Horneff, G, Huppertz, Hi, Iagăru, N, Ibanez Estrella, A, Ioseliani, M, Joos, R, Knupp Oliveira, S, Kamphuis, S, Kasapcopur, O, Katsicas, Mm, Khubchandani, R, Kondi, A, Kröger, L, La Torre, F, Laday, M, Lahdenne, P, Maggio, Mc, Magnolia, Mg, Malagon, C, Malin, M, Martino, S, Melo-Gomes, Ja, Mesa-Del-Castillo, P, Militaru, A, Minden, K, Miniaci, A, Moradinejad, Mh, Morel Ayala, Z, Nikishina, I, Norambuena, X, Nordal, Eb, Pagava, K, Panaviene, V, Pastore, S, Pieropan, S, Podda, Ra, Pruunsild, C, Putto-Laurila, A, Quartier, P, Remesal, A, Rigante, Donato, Ringold, S, Rutkowska-Sak, L, Rygg, M, Saurenmann, Rk, Sawhney, S, Scott, C, Shiari, R, Smolewska, E, Sozeri, B, Swart, Jf, Sztajnbok, F, Torcoletti, M, Tsitsami, E, Tzaribachev, N, Unsal, E, Uziel, Y, Vähäsalo, P, Varbanova, B, Vargova, V, Vesely, R, Vijatov-Djuric, G, Vilaiyuk, S, Vojinovic, J, Vougiouka, O, Weiss, P, Wouters, C, Rigante D (ORCID:0000-0001-7032-7779), Consolaro, A, Giancane, G, Alongi, A, van Dijkhuizen, Ehp, Aggarwal, A, Al-Mayouf, Sm, Bovis, F, De Inocencio, J, Demirkaya, E, Flato, B, Foell, D, Garay, Sm, Lazăr, C, Lovell, Dj, Montobbio, C, Miettunen, P, Mihaylova, D, Nielsen, S, Orban, I, Rumba-Rozenfelde, I, Magalhães, C, Shafaie, N, Susic, G, Trachana, M, Wulffraat, N, Pistorio, A, Martini, A, Ruperto, N, Ravelli, A &amp, Abdwani R, Aghighi, Y, Aiche, Mf, Ailioaie, C, Aktay Ayaz, N, Al-Abrawi, S, Alexeeva, E, Anton, J, Apostol, A, Arguedas, O, Avcin, T, Barone, P, Berntson, L, Boteanu, Al, Boyko, Y, Burgos-Vargas, R, Calvo Penades, I, Chédeville, G, Cimaz, R, Civino, A, Consolini, R, Constantin, T, Cuttica, R, Dallos, T, Martin, N, Magni-Manzoni, S, De Cunto, C, Dolezalova, P, Ekelund, M, El Miedany, Y, Espada, G, Estmann Christensen, A, Foeldvari, I, Gallizzi, R, Ganser, G, Gerloni, V, Haas, Jp, Harel, L, Harjacek, M, Hashad, S, Herlin, T, Herrera, C, Hofer, M, Holzinger, D, Horneff, G, Huppertz, Hi, Iagăru, N, Ibanez Estrella, A, Ioseliani, M, Joos, R, Knupp Oliveira, S, Kamphuis, S, Kasapcopur, O, Katsicas, Mm, Khubchandani, R, Kondi, A, Kröger, L, La Torre, F, Laday, M, Lahdenne, P, Maggio, Mc, Magnolia, Mg, Malagon, C, Malin, M, Martino, S, Melo-Gomes, Ja, Mesa-Del-Castillo, P, Militaru, A, Minden, K, Miniaci, A, Moradinejad, Mh, Morel Ayala, Z, Nikishina, I, Norambuena, X, Nordal, Eb, Pagava, K, Panaviene, V, Pastore, S, Pieropan, S, Podda, Ra, Pruunsild, C, Putto-Laurila, A, Quartier, P, Remesal, A, Rigante, Donato, Ringold, S, Rutkowska-Sak, L, Rygg, M, Saurenmann, Rk, Sawhney, S, Scott, C, Shiari, R, Smolewska, E, Sozeri, B, Swart, Jf, Sztajnbok, F, Torcoletti, M, Tsitsami, E, Tzaribachev, N, Unsal, E, Uziel, Y, Vähäsalo, P, Varbanova, B, Vargova, V, Vesely, R, Vijatov-Djuric, G, Vilaiyuk, S, Vojinovic, J, Vougiouka, O, Weiss, P, Wouters, C, and Rigante D (ORCID:0000-0001-7032-7779)

Abstract

BACKGROUND: To our knowledge, the characteristics and burden of childhood arthritis have never been studied on a worldwide basis. We aimed to investigate, with a cross-sectional study, the prevalence of disease categories, treatment methods, and disease status in patients from across different geographical areas and from countries with diverse wealth status. METHODS: In this multinational, cross-sectional, observational cohort study, we asked international paediatric rheumatologists from specialised centres to enrol children with a diagnosis of juvenile idiopathic arthritis, according to International League of Associations for Rheumatology criteria, who were seen consecutively for a period of 6 months. Each patient underwent retrospective and cross-sectional assessments, including measures of disease activity and damage and questionnaires on the wellbeing and quality of life of the children. We qualitatively compared the collected data across eight geographical areas, and we explored an association between disease activity and damage and a country's gross domestic product (GDP) with a multiple logistic regression analysis. FINDINGS: Between April 4, 2011, and Nov 21, 2016, 9081 patients were enrolled at 130 centres in 49 countries, grouped into eight geographical areas. Systemic arthritis (125 [33·0%] of 379 patients) and enthesitis-related arthritis (113 [29·8%] of 379) were more common in southeast Asia, whereas oligoarthritis was more prevalent in southern Europe (1360 [56·7%] of 2400) and rheumatoid factor-negative polyarthritis was more frequent in North America (165 [31·5%] of 523) than in the other areas. Prevalence of uveitis was highest in northern Europe (161 [19·1%] of 845 patients) and southern Europe (450 [18·8%] of 2400) and lowest in Latin America (54 [6·4%] of 849), Africa and Middle East (71 [5·9%] of 1209), and southeast Asia (19 [5·0%] of 379). Median age at disease onset was lower in southern Europe (3·5 years, IQR 1·9-7·3) than in other regions

Published
2019

7. Is the interruption of antiretroviral treatment during pregnancy an additional major risk factor for mother-to-child transmission of HIV type 1?

Author

Galli, L, Puliti, D, Chiappini, E, Gabiano, C, Ferraris, G, Mignone, F, Viganò, A, Giaquinto, C, Genovese, O, Anzidei, G, Badolato, R, Buffolano, W, Maccabruni, A, Salvini, F, Cellini, M, Ruggeri, M, Manzionna, M, Bernardi, S, Tovo, P, de Martino, M, De Benedictis, F, Osimani, P, La Rovere, D, Quercia, M, Baldi, F, Ciccia, M, Faldella, A, Masi, M, Plebani, A, Spinelli, E, Dedoni, M, Gariel, D, Chiarello, P, Magnolia, Mg, Sticca, M, Vivalda, L, Bezzi, Teresa Maria, Fiumana, Elisa, Bianchi, L, Battiglia, N, Gervaso, P, Bondi, E, Cosso, D, Gotta, C, Ginocchio, L, Rosso, R, Viscoli, C, Amoretti, C, Esposito, S, Farina, F, Giacomet, V, Lipreri, R, Salvatici, E, Stucchi, S, Palazzi, G, Paolucci, P, De Luca, G, Giannattasio, A, Tancredi, F, Tarallo, L, Rampon, O, Dalle Nogare, E, Romano, A, Saitta, M, Mariani, B, Biver, P, Consolini, R, Palla, G, De Fanti, A, Dodi, I, Verna, M, Bove, G, Casadei, Am, Castelli Gattinara, G, Catania, S, Martino, Am, Sirufo, Mm, Ganau, A, Cristiano, L, Scolfaro, C, Versace, A, Portelli, V, Gentilini, L, Mazza, A, Bernardon, M, Bua, J, Rabusin, M, Pellegatta, A, Fortunati, P., Galli, L, Puliti, D, Chiappini, E, Gabiano, C, Ferraris, G, Mignone, F, Viganò, A, Giaquinto, C, Genovese, O, Anzidei, G, Badolato, R, Buffolano, Wilma, Maccabruni, A, Salvini, F, Cellini, M, Ruggeri, M, Manzionna, M, Bernardi, S, Tovo, P, de Martino, M, and Italian Register for HIV Infection in, C. h. i. l. d. r. e. n.

Subjects
Microbiology (medical), antiretroviral treatment, medicine.medical_specialty, Anti-HIV Agents, Pregnancy Trimester, Third, HIV Infections, transmission mother-to-child, HIV infection, Cohort Studies, HIV, Italian Register for HIV Infection in Children, therapeutic use, Cohort Studies, Delivery, Obstetric, Female, HIV Infections, drug therapy/transmission, HIV-1, isolation /&/ purification, Humans, Infant, Newborn, Infectious Disease Transmission, Vertical, Pregnancy, Pregnancy Complications, Infectious, drug therapy, Pregnancy Trimester, First, Pregnancy Trimester, Third, Prospective Studies, Risk Factors, Viral Load, Withholding Treatment, Pregnancy, Risk Factors, medicine, Elective Cesarean Delivery, Humans, Prospective Studies, Risk factor, Pregnancy Complications, Infectious, Maternal Transmission, Obstetrics, business.industry, Infant, Newborn, Viral Load, medicine.disease, Delivery, Obstetric, Confidence interval, Infectious Disease Transmission, Vertical, Surgery, Pregnancy Trimester, First, Infectious Diseases, Withholding Treatment, Cohort, HIV-1, Gestation, Female, business, Viral load
Abstract

There is currently an experts' agreement discouraging interruption of antiretroviral treatment (ART) during the first trimester of pregnancy in women infected with human immunodeficiency virus type 1 (HIV-1). However, this recommendation is poorly supported by data. We evaluated the effects of discontinuing ART during pregnancy on the rate of mother-to-child transmission.Logistic regression models were performed in a prospective cohort of 937 children who were perinatally exposed to HIV-1 to estimate adjusted odds ratios for confounding factors on mother-to-child transmission, including maternal interruption of ART.Among 937 pregnant women infected with HIV-1, ART was interrupted in 81 (8.6%) in the first trimester and in 11 (1.2%) in the third trimester. In the first trimester, the median time at suspension of ART was 6 weeks (interquartile range [IQR], 5-6 weeks) and the time without treatment was 8 weeks (IQR, 7-11 weeks). In the third trimester, the median time at suspension of ART was 32 weeks (IQR, 23-36 weeks) and the time without treatment was 6 weeks (IQR, 2-9 weeks). The plasma viral load was similar in women who had treatment interrupted in the first trimester and in those who did not have treatment interrupted. Overall, the rate of mother-to-child transmission in the whole cohort was 1.3% (95% confidence interval [CI], 0.7%-2.3%), whereas it was 4.9% (95% CI, 1.9%-13.2%) when ART was interrupted in the first trimester and 18.2% (95% CI, 4.5%-72.7%) when ART was interrupted in the third trimester. In the multiple logistic regression models, only interruption of ART during either the first or the third trimester, maternal mono- or double therapy, delivery by a mode other than elective cesarean delivery, and a viral load at delivery4.78 log(10) copies/mL were independently associated with an increased rate of mother-to-child transmission.Discontinuing ART during pregnancy increases the rate of mother-to-child transmission of HIV-1, either when ART is stopped in the first trimester and subsequently restarted or when it is interrupted in the third trimester. This finding supports recommendations to continue ART in pregnant women who are already receiving treatment for their health.

Published
2009

8. Persistence of disease flares is associated with an inadequate colchicine dose in familial Mediterranean fever: A national multicenter longitudinal study

Author

Angela Miniaci, Nicolino Ruperto, Maria Greca Magnolia, Alessandra Spagnolo, Giovanni Conti, Davide Montin, Maria Carrabba, Laura Obici, Giovanna Fabio, Edoardo Marrani, Giovanna Capozio, Marco Cattalini, Patrizia Barone, Maddalena Lancieri, Francesco Calzatini, Valentina Moressa, Maria Cristina Maggio, Gabriele Simonini, Camilla Celani, Luca Cantarini, Francesca Mazza, Diana Sutera, Roberta Mussinelli, Luciana Breda, Marta Bustaffa, Romina Gallizzi, Francesca Orlando, Maria Alessio, Antonella Insalaco, Alma Nunzia Olivieri, Donato Rigante, Carla Gaggiano, Francesca Ricci, Marco Gattorno, Bustaffa, M, Mazza, F, Sutera, D, Carrabba, Md, Alessio, M, Cantarini, L, Obici, L, Rigante, D, Maggio, Mc, Insalaco, A, Simonini, G, Cattalini, M, Conti, G, Olivieri, An, Barone, P, Miniaci, A, Moressa, V, Magnolia, Mg, Breda, L, Montin, D, Spagnolo, A, Fabio, G, Orlando, F, Gaggiano, C, Mussinelli, R, Capozio, G, Celani, C, Marrani, E, Ricci, F, Calzatini, F, Lancieri, M, Ruperto, N, Gattorno, M, Gallizzi, R, Bustaffa M., Mazza F., Sutera D., Carrabba M.D., Alessio M., Cantarini L., Obici L., Rigante D., Maggio M.C., Insalaco A., Simonini G., Cattalini M., Conti G., Olivieri A.N., Barone P., Miniaci A., Moressa V., Magnolia M.G., Breda L., Montin D., Spagnolo A., Fabio G., Orlando F., Gaggiano C., Mussinelli R., Capozio G., Celani C., Marrani E., Ricci F., Calzatini F., Lancieri M., Ruperto N., Gattorno M., and Gallizzi R.

Subjects
Longitudinal study, business.industry, Familial Mediterranean fever, Interleukin, Disease, Familial Mediterranea fever, medicine.disease, Symptom Flare Up, Colchicine, Humans, Interleukin 1 Receptor Antagonist Protein, Longitudinal Studies, Familial Mediterranean Fever, Persistence (computer science), chemistry.chemical_compound, Settore MED/38 - Pediatria Generale E Specialistica, chemistry, Colchicine, Humans, Interleukin 1 Receptor Antagonist Protein, Longitudinal Studies, Symptom Flare Up, Familial Mediterranean Fever, Immunology, COLCHICINE RESISTANCE, Immunology and Allergy, Medicine, business
Abstract

Familial Mediterranean fever (FMF) is characterized by self limited episodes of fever and polyserositis.1 MEFV gene en codes for a protein named Pyrin, which plays a pivotal role in the activation and secretion of IL-1.2 Daily colchicine is highly effective in preventing attacks in this disorder in a dose-related fashion.3 Many definitions of colchicine resistance are available in the literature. The European League Against Rheumatism (EULAR) guidelines defined resistance as one or more attacks per month in compliant patients who had been receiving the maxi mally tolerated dose for at least 6 months.4 A similar definition was confirmed by a recent consensus among experts.5 In the present national multicentric longitudinal study, we analyze the impact of colchicine treatment on disease activity and quality of life in real life in pediatric and adult patients with FMF.

Published
2021

9. Clinical features, treatment and outcomes of Italian children with enthesitis-related arthritis and juvenile psoriatic arthritis: a cross-sectional cohort study.

Author

Rebollo-Giménez AI, Rosina S, Natoli V, Burrone M, Barone P, Campus S, Civino A, Consolini R, Filocamo G, Gallizzi R, Gattinara M, La Torre F, Maggio MC, Magni-Manzoni S, Magnolia MG, Miniaci A, Montin D, Pastore S, Petaccia A, Pieropan S, Rigante D, Simonini G, Ridella F, Orsi SM, Angioloni S, Carlini L, Ruperto N, and Consolaro A

Subjects
Child, Humans, Retrospective Studies, Cross-Sectional Studies, Quality of Life, Treatment Outcome, Arthritis, Juvenile diagnosis, Arthritis, Juvenile drug therapy, Arthritis, Juvenile epidemiology
Abstract

Objectives: Limited information is available on the clinical features, treatment modalities and outcomes of the juvenile idiopathic arthritis (JIA) categories of enthesitis-related arthritis (ERA) and juvenile psoriatic arthritis (JPsA). This study was aimed to describe the characteristics of Italian children with ERA and JPsA and to compare them with those of patients with the other categories of JIA., Methods: Patients were part of a multinational sample included in a study aimed to investigate the prevalence of disease categories, treatment approaches, and disease status in patients from across different geographical areas (EPOCA Study). All patients underwent a retrospective assessment, based on the review of clinical chart, and a cross-sectional evaluation, which included assessment of physician- and parent-reported outcomes and laboratory tests, and recording of ongoing therapies., Results: Of the 9081 children with JIA enrolled in the EPOCA Study, 1300 were recruited at 18 paediatric rheumatology centres in Italy. 45 (3.5%) had ERA and 49 (3.8%) had JPsA. Several remarkable differences in demographic features and frequency of articular and extra-articular manifestations, disease damage, impairment in physical function and health-related quality of life, school-related problems, comorbidities, and ongoing treatments were observed between ERA and JPsA and the other JIA categories., Conclusions: We described the characteristics of Italian children with ERA and JPsA and highlighted their peculiarities and their differences from the other JIA subsets. These data provide useful insights for future revisions of JIA classification and a benchmarking against which the features from other cohorts may be compared.

Published
2024
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10. Persistence of disease flares is associated with an inadequate colchicine dose in familial Mediterranean fever: A national multicenter longitudinal study.

Author

Bustaffa M, Mazza F, Sutera D, Carrabba MD, Alessio M, Cantarini L, Obici L, Rigante D, Maggio MC, Insalaco A, Simonini G, Cattalini M, Conti G, Olivieri AN, Barone P, Miniaci A, Moressa V, Magnolia MG, Breda L, Montin D, Spagnolo A, Fabio G, Orlando F, Gaggiano C, Mussinelli R, Capozio G, Celani C, Marrani E, Ricci F, Calzatini F, Lancieri M, Ruperto N, Gattorno M, and Gallizzi R

Subjects
Colchicine, Humans, Interleukin 1 Receptor Antagonist Protein, Longitudinal Studies, Symptom Flare Up, Familial Mediterranean Fever drug therapy, Familial Mediterranean Fever epidemiology
Published
2021
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11. A clinical prediction model for estimating the risk of developing uveitis in patients with juvenile idiopathic arthritis.

Author

van Straalen JW, Giancane G, Amazrhar Y, Tzaribachev N, Lazar C, Uziel Y, Telcharova-Mihaylovska A, Len CA, Miniaci A, Boteanu AL, Filocamo G, Mastri MV, Arkachaisri T, Magnolia MG, Hoppenreijs E, de Roock S, Wulffraat NM, Ruperto N, and Swart JF

Subjects
Child, Child, Preschool, Female, Humans, Male, Predictive Value of Tests, Risk Factors, Arthritis, Juvenile complications, Clinical Decision Rules, Uveitis diagnosis, Uveitis etiology
Abstract

Objective: To build a prediction model for uveitis in children with JIA for use in current clinical practice., Methods: Data from the international observational Pharmachild registry were used. Adjusted risk factors as well as predictors for JIA-associated uveitis (JIA-U) were determined using multivariable logistic regression models. The prediction model was selected based on the Akaike information criterion. Bootstrap resampling was used to adjust the final prediction model for optimism., Results: JIA-U occurred in 1102 of 5529 JIA patients (19.9%). The majority of patients that developed JIA-U were female (74.1%), ANA positive (66.0%) and had oligoarthritis (59.9%). JIA-U was rarely seen in patients with systemic arthritis (0.5%) and RF positive polyarthritis (0.2%). Independent risk factors for JIA-U were ANA positivity [odds ratio (OR): 1.88 (95% CI: 1.54, 2.30)] and HLA-B27 positivity [OR: 1.48 (95% CI: 1.12, 1.95)] while older age at JIA onset was an independent protective factor [OR: 0.84 (9%% CI: 0.81, 0.87)]. On multivariable analysis, the combination of age at JIA onset [OR: 0.84 (95% CI: 0.82, 0.86)], JIA category and ANA positivity [OR: 2.02 (95% CI: 1.73, 2.36)] had the highest discriminative power among the prediction models considered (optimism-adjusted area under the receiver operating characteristic curve = 0.75)., Conclusion: We developed an easy to read model for individual patients with JIA to inform patients/parents on the probability of developing uveitis., (© The Author(s) 2020. Published by Oxford University Press on behalf of the British Society for Rheumatology.)

Published
2021
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