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1. Low prevalence of lipodystrophy in HIV-infected Senegalese children on long-term antiretroviral treatment: the ANRS 12279 MAGGSEN Pediatric Cohort Study

Author: Cecile Cames, Lea Pascal, Aissatou Ba, Hélène Mbodj, Baly Ouattara, Ndeye-Fatou Diallo, Philippe Msellati, Ngagne Mbaye, Haby Sy Signate, Stephane Blanche, Aminata Diack, and for the MAGGSEN Cohort Study Group
Subjects: Lipodystrophy, Lipoatrophy, Children, Adolescents, HIV-infection, ART, Infectious and parasitic diseases, RC109-216
Abstract: Abstract Background The long-term benefits of antiretroviral treatment (ART) are associated with metabolic complications, especially lipodystrophy, which has been well described among HIV-infected adults and children on ART in developed settings. Specifically, stavudine, and to a lesser extent zidovudine and protease inhibitors (PI), have been consistently implicated in the development of lipodystrophy. In 2006, following advice from the WHO, Senegal began phasing out stavudine from first-line ART. The objectives of this cross-sectional analysis are to assess and identify risk factors affecting the prevalence of lipodystrophy in Senegalese children and adolescents on long-term ART participating in a cohort study. Methods Lipodystrophy was clinically assessed in two- to 18-year-old children on ART for at least six months and with no concurrent severe acute malnutrition. Risk factors for lipodystrophy were identified using stepwise multivariable logistic regression. Explanatory variables included clinical and personal data, immunovirologic status, and therapeutic history. Results Overall, 254 children were assessed for lipodystrophy. The median age was 10.9 years (IQR: 8.1–14.2) and the median duration on ART was 54 months (32–84). Only 18% had been previously treated with stavudine, with a median treatment duration of 8 months (5–25). Ongoing treatment included 76% of children receiving zidovudine (median duration of 48 months (26–74)) and 27% receiving PI (lopinavir/ritonavir; median duration of 49 months (23–59)). Mild signs of lipodystrophy were observed in 33 children (13%): 28 with lipoatrophy, 4 with lipohypertrophy and one with combined type. Boys were more likely to present with lipoatrophy than girls (aOR: 4.3, 95% CI: 1.6–11.7). Children previously treated with stavudine for ≥1 year had a greater risk for lipoatrophy than those never exposed (3.8, 1.0–14.0), although the association was weak. There was no association between lipodystrophy and age or current or cumulative treatment with lopinavir/ritonavir or zidovudine. Conclusions We report low prevalence of mild lipodystrophy in children and adolescents on long-term ART receiving a stavudine-sparing regimen. These findings are reassuring for clinicians in low-income settings where zidovudine is massively prescribed and lopinavir/ritonavir is the only widely available PI. Trial registration ClinicalTrials.gov identifier: NCT01771562 (registration date: 01/18/2013).
Published: 2018
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2. No overall impact on body mass index for age change after dolutegravir initiation in a French paediatric cohort

Author: Pierre Frange, Veronique Avettand‐Fenoel, Florence Veber, and Stephane Blanche
Subjects: Adult, Adolescent, Pyridones, Health Policy, HIV Infections, Piperazines, Body Mass Index, Infectious Diseases, Oxazines, Humans, Pharmacology (medical), Child, Heterocyclic Compounds, 3-Ring, Retrospective Studies
Abstract: Increased weight gain with dolutegravir use is increasingly scrutinized in adults, but published data in paediatrics are limited and conflicting. This study aimed to provide long-term data about changes in body mass index (BMI) in French children (aged 3-9 years) and adolescents (aged 10-17 years) receiving dolutegravir.This retrospective monocentric study included 97 subjects who received a dolutegravir-based regimen for ≥12 months in 2014-2021. We evaluated the mean change in age- and sex-matched standardized BMI z score (BMIz) per year of dolutegravir exposure and compared the dynamics of BMIz change during the 12 months pre- vs. post-dolutegravir use when these data were available.At the time of dolutegravir initiation, most of the subjects were antiretroviral therapy (ART) experienced (89.7%), displayed virological suppression (73.2%), and had normal weight for their age (78.4%). Median follow-up was 30 months (interquartile range [IQR] 19-45). The mean rate of change in BMIz was +0.03 z score/year of dolutegravir exposure (95% confidence interval [CI] -0.08-0.13) in the entire cohort. It was lower in children than in adolescents (-0.08 [95% CI -0.23-0.08] vs. +0.16 [95% CI 0.06-0.26], respectively; p = 0.04) and in individuals with baseline BMI ≥50We found no evidence of change in BMIz in French children initiating dolutegravir. These reassuring findings maintain the primary position of dolutegravir among paediatric therapeutic options.
Published: 2022
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3. Late-onset enteric virus infection associated with hepatitis (EVAH) in transplanted SCID patients

Author: Quentin Riller, Jacques Fourgeaud, Julie Bruneau, Suk See De Ravin, Grace Smith, Mathieu Fusaro, Samy Meriem, Aude Magerus, Marine Luka, Ghaith Abdessalem, Ludovic Lhermitte, Anne Jamet, Emmanuelle Six, Alessandra Magnani, Martin Castelle, Romain Lévy, Mathilde M. Lecuit, Benjamin Fournier, Sarah Winter, Michaela Semeraro, Graziella Pinto, Hanène Abid, Nizar Mahlaoui, Nathalie Cheikh, Benoit Florkin, Pierre Frange, Eric Jeziorski, Felipe Suarez, Françoise Sarrot-Reynauld, Dalila Nouar, Dominique Debray, Florence Lacaille, Capucine Picard, Philippe Pérot, Béatrice Regnault, Nicolas Da Rocha, Camille de Cevins, Laure Delage, Brieuc P. Pérot, Angélique Vinit, Francesco Carbone, Camille Brunaud, Manon Marchais, Marie-Claude Stolzenberg, Vahid Asnafi, Thierry Molina, Frédéric Rieux-Laucat, Luigi D. Notarangelo, Stefania Pittaluga, Jean Philippe Jais, Despina Moshous, Stephane Blanche, Harry Malech, Marc Eloit, Marina Cavazzana, Alain Fischer, Mickaël M. Ménager, and Bénédicte Neven
Subjects: Immunology, Immunology and Allergy
Published: 2023
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4. Adaptation and Coping of HIV Infected Adolescents Living in France

Author: Isabelle Funck-Brentano, Florence Veber, Marianne Debre, and Stephane Blanche
Published: 2022
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5. Young children still to treat, unfortunately

Author: Stephane Blanche
Subjects: Infectious Diseases, Epidemiology, Virology, Child, Preschool, Immunology, Humans, HIV Infections, Child
Published: 2022

6. Severe hematopoietic stem cell inflammation compromises chronic granulomatous disease gene therapy

Author: Steicy Sobrino, Alessandra Magnani, Michaela Semeraro, Loredana Martignetti, Akira Cortal, Adeline Denis, Chloé Couzin, Capucine Picard, Jacinta Bustamante, Elisa Magrin, Laure Joseph, Cécile Roudaut, Aurélie Gabrion, Tayebeh Soheili, Corinne Cordier, Olivier Lortholary, François Lefrere, Frédéric Rieux-Laucat, Jean-Laurent Casanova, Sylvain Bodard, Nathalie Boddaert, Adrian J. Thrasher, Fabien Touzot, Sophie Taque, Felipe Suarez, Ambroise Marcais, Agathe Guilloux, Chantal Lagresle-Peyrou, Anne Galy, Antonio Rausell, Stephane Blanche, Marina Cavazzana, and Emmanuelle Six
Subjects: General Biochemistry, Genetics and Molecular Biology
Published: 2023
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7. The DIANA Study: Continued Access to Darunavir/Ritonavir (DRV/r) and Long-Term Safety Follow-Up in HIV-1-Infected Pediatric Patients Aged 3 to 18 Years

Author: Avy, Violari, Maysseb, Masenya, Stephane, Blanche, Simon, Vanveggel, Veerle, Hufkens, Polan, Chetty, and Magda, Opsomer
Subjects: Ritonavir, Adolescent, Child, Preschool, HIV-1, Humans, HIV Infections, HIV Protease Inhibitors, Child, Health Services Accessibility, Darunavir, Follow-Up Studies
Abstract: Darunavir is a human immunodeficiency virus type 1 (HIV-1) protease inhibitor boosted with ritonavir (DRV/r) or cobicistat.This study provided continued access to DRV/r and assessed long-term safety in patients aged 3 to 18 years.Patients who had completed treatment in the DELPHI (TMC114-C212), DIONE (TMC114-TiDP29-C230), or ARIEL (TMC114-TiDP29-C228) studies were eligible to participate if they derived benefit from using DRV/r in countries where it was not available to them. DRV/r dosing was continued based on original study protocols. Only safety data were collected. Reportable adverse events (AEs) included AEs considered at least possibly related to treatment with DRV/r, AEs leading to discontinuation or treatment interruption, and serious AEs (SAEs).Forty-six patients rolled over to this study and received at least one dose of DRV/r. Median duration of DRV/r intake was 4.2 years. Overall, 15/46 patients experienced one or more reportable AEs, 10/46 patients experienced one or more grade 3 or 4 AEs, and 12/46 patients experienced one or more SAEs. The most common grade 3 or 4 and SAEs were pneumonia (3/46) and asthma (2/46). Only one AE (grade 1 lipoatrophy) was considered probably related to DRV/r (DIONE, n = 1). Overall, 3/46 patients experienced an HIV-related AE (grade 3 pneumonia SAE; grade 2 tuberculosis SAE, and grade 2 lipoatrophy AE), none of which were considered related to DRV/r or led to study discontinuation. Two AEs leading to discontinuation were pregnancies.These long-term safety results continue to support DRV/r as a valuable therapeutic option for the treatment of HIV-1 infection in pediatric patients aged ≥ 3 years.ClinicalTrials.gov: NCT01138605/EudraCT number: 2009-017013-29; first submitted 8 April 2010.
Published: 2020

8. Life-Saving, Dose-Adjusted, Targeted Therapy in a Patient with a STAT3 Gain-of-Function Mutation

Author: Eytan, Sarfati, Jérome, Hadjadj, Mathieu, Fusaro, Roman, Klifa, Marion, Grimaud, Laureline, Berteloot, Alice, Hadchouel, Cécile, Godot, Marie-Claude, Stolzenberg, Marie-Louise, Frémond, Claire, Pressiat, Thierry, Molina, Alain, Fischer, Capucine, Picard, Sylvain, Renolleau, Frederic, Rieux-Laucat, Stephane, Blanche, and Benedicte, Neven
Subjects: STAT3 Transcription Factor, Genotype, Disease Management, Phenotype, Treatment Outcome, Immune System Diseases, Gain of Function Mutation, Humans, Janus Kinase Inhibitors, Genetic Predisposition to Disease, Molecular Targeted Therapy, Tomography, X-Ray Computed, Alleles, Genetic Association Studies, Germ-Line Mutation
Published: 2020

9. Immunovirological status in people with perinatal and adult-acquired HIV-1 infection: a multi-cohort analysis from FranceResearch in context

Author: Rémonie Seng, Pierre Frange, Albert Faye, Catherine Dollfus, Jérôme le Chenadec, Faroudy Boufassa, Asma Essat, Tessa Goetghebuer, Elisa Arezes, Véronique Avettand-Fènoël, Jean-Joël Bigna, Stéphane Blanche, Cécile Goujard, Laurence Meyer, Josiane Warszawski, Jean-Paul Viard, H. Aumaitre, E. Froguel, F. Caby, S. Dellion, L. Gerard, F. Lucht, C. Chirouze, M. Dupon, Jl Schmit, C. Goujard, T. Allegre, B. Cazenave, G. Hittinger, P. De Truchis, J. Cailhol, C. Duvivier, A. Canestri, O. Bouchaud, M. Karmochkine, D. Salmon-Ceron, D. Zucman, E. Mortier, R. Tubiana, P.M. Girard, C. Pintado, A. Cabie, V. Rabier, P. Morlat, D. Neau, C. Genet, D. Makhloufi, S Bregigeon Ronot, J. Ghosn, V. Reliquet, P. Perré, Jl Pellegrin, C. Arvieux, C. Cheneau, L. Bernard, P. Delobel, R. Verdon, C. Jacomet, L. Piroth, F. Ajana, S. Bevilacqua, Y. Debab, A.L. Lecapitaine, L. Cotte, S. Mokhtari, P. Mercie, P. Poubeau, V. Garrait, Ma Khuong, G. Beck-Wirth, L. Blum, S. Blanche, F. Boccara, T. Prazuck, C. Barbuat, J.P. Viard, S. Stegmann-Planchard, B. Martha, J.M. Treluyer, E. Dore, C. Gaud, M. Niault, E. Fernandes, H. Hitoto, A. Compagnucci, N. Elenga, A. Faye, C. Dollfus, A. Chace, M. Levine, S.A. Martha, C. Floch-Tudal, K. Kebaïli, N. Entz-Werle, J. Tricoire, F. Mazingue, P. Bolot, P. Brazille, T. Goetghebuer, A.F. Gennotte, D. Van Der Linden, V. Schmitz, M. Moutschen, C. Crenn-Hebert, F. Habibi, A. Coursol, E. Guesdon, P.F. Ceccaldi, M. Dehlinger – Paul, E. Pannier, V. Marcou, C. Elleau, M. Achkar, M.O. Vareil, S. Couderc, C. Routier, M.A. Bouldouyre, L. Selleret, A. Chabrol, C. Bellahcene, C. Pluchart, A. Yangui, D. Vignes, A. Alissa, A. Johnson, E. Lachassinne, A. Benbara, L. Karaoui, A. Bongain, B. Yakeu, J.L. Schmit, L. Cravello, C. Hubert, P. Faucher, D. Pinquier, C. Borie, D. Rocchi, C. Brunet-Cartier, C. Briandet, J. Brouard, A. Chalvon-Demersay, M. Rajguru, K. Billiemaz, A. Fresard, A. Moulin, P. Fialaire, L. Mesnard, E. Werner, E. Vintejoux, J. Marian, S. Ranaivojaona, F. Bissuel, M. Abdelhadi, Y. Hammou, C. Genet-Villeger, Y. Hatchuel, G. Bachelard, M. Medus, J. Dendale – Nguyen, T.S. Guimard, A. Martha, M. Rouha, P. Perfezou, L. De Saint Martin, S. Jaffuel, R. Buzele, M. Gousseff, C. Cudeville, V. Vitrat, C. Michau, G. Palenzuela, M. Driessen, B. Heller-Roussin, J.M. Labaune, B. Muanza, J. Massardier, M. Partisani, I. Hau, C. Runel-Belliard, C. Brehin, K. Kebaili, M. Lalande, M. Lagree, K. Lacombe, J.-M. Molina, J. Reynes, O. Robineau, F. Raffi, A. Becker, L. Weiss, T. Allègre, G. Pialoux, F. Souala, A. Rami, C. Katlama, A. Cabié, J.-P. Viard, F. Bastides, C. Michel, D. Salmon, J-D Le Lièvre, A. Sotto, E. Rouveix, A. Naqvi, S. Brégigeon, R. Rodet, A. Simon-Coutelier, J.-L. Esnault, R. Buzelé, A. Stein, C. Godin-Colet, G. Pichancourt, P. Caraux-Paz, M Mohseni Zadeh, L. Gérard, C. Lascaux-Cametz, L. Bodard, J.-L. Pellegrin, N. Ettahar, A. Uludag, E. Rosenthal, F. Prevoteau du Clary, S. Jaureguiberry, P. Philibert, A.-L. Lecapitaine, E. Chakvetadze, H. Champagne, V. Daneluzzi, J. Goupil de Bouillé, A. Leprêtre, I. Lamaury, I. Darasteanu, B. Abraham, D. Garipuy, J.-L. Berger, J.-L. Schmit, K. Diallo, F. Gourdon, O. Vaillant, V. Gaborieau, J. Doll, D. Quinsat, L. Geffray, J.-J. Girard, D. Houlbert, V. Perronne, E. Klement, O. Antioniotti, C. Rouzioux, V. Avettand-Fenoel, O. Lortholary, S. Boucly, A. Maignan, R. Thiebaut, L. Meyer, F. Boufassa, M.A. Charles, R. Dray-Spira, C. Legeai, V. Amon, N. Benammar, R. Seng, L. Slama, P. Bonnard, C. Chakvetadze, T. L’Yavanc, J. Capeau, C. Vigouroux, S. Fellahi, J.P. Bastard, E. Oksenhendler, J.F. Bourge, V. Bajzik, D. Sereni, C. Lascoux-Combe, O. Taulera, L.V. Dien, J. Delgado, J.M. Molina, T. Saint-Marc, S. Ferret, J. Pavie, J.F. Bergmann, M. Parrinello, BLefebvre, C. Boudraa, B. Diallo, C. Lupin, S. Herson, A. Simon, N. Edeb, L. Guillevin, T. Tahi, M.P. Pietri, D. Tisne-Dessus, C. Jalbert, P. Yeni, S. Matheron, G. Pahlavan, B. Phung, N. El-Alami Talbi, Z. Ramani, G. Catalano, C. Godard, F. Boue, V. Chambrin, D. Bornarel, H. Schoen, R. Carlier, B. Fantin, C. Poder, R. Dhote, M. Bentata, P. Honore, Xuan Tuyet, J.F. Delfraissy, F. Chaix, M.T. Rannou, Y. Levy, A. Sobel, C. Dumont, S. Abel, S. Pierre-François, V. Beaujolais, I. Poizot-Martin, O. Zaegel-Faucher, C. Debreux, J. Moreau, E. Van Der Gheynst, M.C. Thiebaut-Drobacheff, A. Foltzer, B. Hoen, J.F. Faucher, H. Gil, J.M. Ragnaud, I. Raymond, I. Louis, M. Hessamfar, V. Baillat, C Merle De Boever, C. Tramoni, A. Soufflet, P. Guadagnin, P. Choutet, O. Mounoury, D. Brosseau, H. Hue, T. May, S. Wassoumbou, M. Stenzel, M.P. Bouillon, Y. Yazdanpanah, T. Huleux, E. Aissi, S. Pavel, D. Rey, P. Fischer, G. Blaison, M. Martinot, A. Pachart, F. Jeanblanc, J.L. Touraine, C. Trepo, P. Miailhes, K. Kouadjo, V. Thoirain, C. Brochier, P. Perre, S. Leautez, J.L. Esnault, and I. Suaud
Subjects: Perinatal HIV infection, Cohort, Viral failure, Immunological outcome, Epidemiology, Public aspects of medicine, RA1-1270
Abstract: Summary: Background: No study has compared the virological and immunological status of young people with perinatally-acquired HIV infection (P-HIV) with that of people with HIV adulthood (A-HIV) having a similar duration of infection. Methods: 5 French cohorts of P-HIV and A-HIV patients with a known date of HIV-infection and receiving antiretroviral treatment (ART), were used to compare the following proportions of: virological failure (VF) defined as plasma HIV RNA ≥ 50 copies/mL, CD4 cell percentages and CD4:CD8 ratios, at the time of the most recent visit since 2012. The analysis was stratified on time since infection, and multivariate models were adjusted for demographics and treatment history. Findings: 310 P-HIV were compared to 1515 A-HIV (median current ages 20.9 [IQR:14.4–25.5] and 45.9 [IQR:37.9–53.5] respectively). VF at the time of the most recent evaluation was significantly higher among P-HIV (22.6%, 69/306) than A-HIV (3.3%, 50/1514); p ≤ 0.0001. The risk of VF was particularly high among the youngest children (2–5 years), adolescents (13–17 years) and young adults (18–24 years), compared to A-HIV with a similar duration of infection: adjusted Odds-Ratio (aOR) 7.0 [95% CI: 1.7; 30.0], 11.4 [4.2; 31.2] and 3.3 [1.0; 10.8] respectively. The level of CD4 cell percentages did not differ between P-HIV and A-HIV. P-HIV aged 6–12 and 13–17 were more likely than A-HIV to have a CD4:CD8 ratio ≥ 1: 84.1% vs. 58.8% (aOR = 3.5 [1.5; 8.3]), and 60.9% vs. 54.7% (aOR = 1.9 [0.9; 4.2]) respectively. Interpretation: P-HIV were at a higher risk of VF than A-HIV with a similar duration of infection, even after adjusting for treatment history, whereas they were not at a higher risk of immunological impairment. Exposure to viral replication among young patients living with HIV since birth or a very early age, probably because of lower adherence, could have an impact on health, raising major concerns about the selection of resistance mutations and the risk of HIV transmission. Funding: Inserm - ANRS MIE.
Published: 2024
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10. Mother to child HIV transmission

Author: Stephane, Blanche
Subjects: Anti-Retroviral Agents, Pregnancy, Infant, Newborn, Humans, Female, HIV Infections, Pregnancy Complications, Infectious, Infectious Disease Transmission, Vertical
Abstract: Thirty years after the first descriptions of AIDS in children in 1983, the risk of viral transmission from mother to child has been reduced to almost zero, thanks to antiretroviral drugs during pregnongh. This extraordinary prophylactic efficacy progressively spread to poor countries with high prevalence. The hope of a virtual eradication of HIV transmission appears as a "feasible utopia".
Published: 2018

11. Low prevalence of lipodystrophy in HIV-infected Senegalese children on long-term antiretroviral treatment: the ANRS 12279 MAGGSEN Pediatric Cohort Study

Author: Cecile Cames, Lea Pascal, Aissatou Ba, Hélène Mbodj, Baly Ouattara, Ndeye-Fatou Diallo, Philippe Msellati, Ngagne Mbaye, Haby Sy Signate, Stephane Blanche, Aminata Diack, for the MAGGSEN Cohort Study Group, Recherches Translationnelles sur le VIH et les maladies infectieuses endémiques er émergentes (TransVIHMI), Institut de Recherche pour le Développement (IRD)-Université de Yaoundé I-Université Cheikh Anta Diop [Dakar, Sénégal] (UCAD)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Montpellier (UM), Health Service and Performance Research (HESPER), Université Claude Bernard Lyon 1 (UCBL), Université de Lyon-Université de Lyon, Centre Hospitalier National d'Enfants Albert Royer, Centre Hospitalier Roi Baudouin, CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Agence Nationale de Recherche sur le SIDA et les hépatites virales (ANRS)., MAGGSEN Cohort Study Group, Duchange, Nathalie, Recherches Translationnelles sur le VIH et les maladies infectieuses endémiques et émergentes (TransVIHMI), and Université Cheikh Anta Diop [Dakar, Sénégal] (UCAD)-Institut de Recherche pour le Développement (IRD)-Université de Yaoundé I-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Montpellier (UM)-Université Montpellier 1 (UM1)
Subjects: Male, Adolescent, Lipodystrophy, Anti-HIV Agents, HIV Infections, Adolescents, lcsh:Infectious and parasitic diseases, Cohort Studies, [SDV.MHEP.MI]Life Sciences [q-bio]/Human health and pathology/Infectious diseases, Risk Factors, Prevalence, Humans, lcsh:RC109-216, Child, HIV-infection, Children, Infant, HIV Protease Inhibitors, Senegal, Stavudine, Cross-Sectional Studies, Protease inhibitor, Child, Preschool, [SDV.MHEP.MI] Life Sciences [q-bio]/Human health and pathology/Infectious diseases, Female, Lipoatrophy, Zidovudine, ART, Research Article
Abstract: International audience; BACKGROUND:The long-term benefits of antiretroviral treatment (ART) are associated with metabolic complications, especially lipodystrophy, which has been well described among HIV-infected adults and children on ART in developed settings. Specifically, stavudine, and to a lesser extent zidovudine and protease inhibitors (PI), have been consistently implicated in the development of lipodystrophy. In 2006, following advice from the WHO, Senegal began phasing out stavudine from first-line ART. The objectives of this cross-sectional analysis are to assess and identify risk factors affecting the prevalence of lipodystrophy in Senegalese children and adolescents on long-term ART participating in a cohort study.METHODS:Lipodystrophy was clinically assessed in two- to 18-year-old children on ART for at least six months and with no concurrent severe acute malnutrition. Risk factors for lipodystrophy were identified using stepwise multivariable logistic regression. Explanatory variables included clinical and personal data, immunovirologic status, and therapeutic history.RESULTS:Overall, 254 children were assessed for lipodystrophy. The median age was 10.9 years (IQR: 8.1-14.2) and the median duration on ART was 54 months (32-84). Only 18% had been previously treated with stavudine, with a median treatment duration of 8 months (5-25). Ongoing treatment included 76% of children receiving zidovudine (median duration of 48 months (26-74)) and 27% receiving PI (lopinavir/ritonavir; median duration of 49 months (23-59)). Mild signs of lipodystrophy were observed in 33 children (13%): 28 with lipoatrophy, 4 with lipohypertrophy and one with combined type. Boys were more likely to present with lipoatrophy than girls (aOR: 4.3, 95% CI: 1.6-11.7). Children previously treated with stavudine for ≥1 year had a greater risk for lipoatrophy than those never exposed (3.8, 1.0-14.0), although the association was weak. There was no association between lipodystrophy and age or current or cumulative treatment with lopinavir/ritonavir or zidovudine.CONCLUSIONS:We report low prevalence of mild lipodystrophy in children and adolescents on long-term ART receiving a stavudine-sparing regimen. These findings are reassuring for clinicians in low-income settings where zidovudine is massively prescribed and lopinavir/ritonavir is the only widely available PI.TRIAL REGISTRATION:ClinicalTrials.gov identifier: NCT01771562 (registration date: 01/18/2013).
Published: 2017
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12. Extended pre-exposure prophylaxis with lopinavir–ritonavir versus lamivudine to prevent HIV-1 transmission through breastfeeding up to 50 weeks in infants in Africa (ANRS 12174): a randomised controlled trial

Author: Nicolas Nagot, Chipepo Kankasa, James K Tumwine, Nicolas Meda, G Justus Hofmeyr, Roselyne Vallo, Mwiya Mwiya, Mary Kwagala, Hugues Traore, Amwe Sunday, Mandisa Singata, Chafye Siuluta, Eric Some, David Rutagwera, Desire Neboua, Grace Ndeezi, Debra Jackson, Valérie Maréchal, Dorine Neveu, Ingunn M S Engebretsen, Carl Lombard, Stéphane Blanche, Halvor Sommerfelt, Claire Rekacewicz, Thorkild Tylleskär, Philippe Van de Perre, Valerie Marechal, Marianne Peries, Vincent Foulongne, Michel Segondy, Stephane Blanche, Jean-Marc Treluyer, Deborah Hirt, Charles Karamagi, Philippa Musoke, Proscovia M Mugaba, Joan Murungi, Hawa Nabuuma Muweesi, Evelyn Ninsiima, Simon Baryeija, Frederic Juma, Caleb Bwengye Kata, Stuart Katushabe, Rasmata Ouédraogo, Diarra Yé, Eric Somé, Hugues A Traoré, Christelle Nadembega, Justin Konaté, Arsène Zongo, Abass Ouédraogo, Désiré Néboua, Aissatou Bélemviré, Armel Bambara, Justine Boncoungou, Danielle Zoungrana, Cheryl Nikodem, Justus Hofmeyr, Kim Harper, David Sanders, Amwe Aku, Collins Okegbe-Eze, Xoliswa Williams, Nolundi Mshweshwe, Vatiswa Henge, Fikiswa Gomba, Tapiwa Gundu, Oswell Khondowe, Mildred Lusaka, Mary Chizyuka, Mary Phiri, Billies Imakando, Mwenechanya Musaku, Monica Kapasa, Gondwe Clement, Hilton Mwila Mwaba, Japhet Matoba, Katai Chola, Patricia Mwamutanda, Ingunn Engebretsen, Jørn Klungsøyr, Jan van den Broeck, Jörn Blume, Pathogénèse et contrôle des infections chroniques (PCCI), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre Hospitalier Universitaire de Montpellier (CHU Montpellier )-Université de Montpellier (UM), Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Service d'immuno-hématologie pédiatrique [CHU Necker], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), CIC - Mère Enfant Necker Cochin Paris Centre (CIC 1419), Hôpital Cochin [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM), Université Paris Descartes - Paris 5 (UPD5), CHU Necker - Enfants Malades [AP-HP], Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Montpellier (UM)-Centre Hospitalier Universitaire de Montpellier (CHU Montpellier ), Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-CHU Necker - Enfants Malades [AP-HP], and CHU Cochin [AP-HP]-Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM)
Subjects: Pediatrics, medicine.medical_specialty, Anti-HIV Agents, Population, Breastfeeding, Lopinavir/ritonavir, HIV Infections, 030204 cardiovascular system & hematology, Drug Administration Schedule, Lopinavir, law.invention, 03 medical and health sciences, Pre-exposure prophylaxis, 0302 clinical medicine, Randomized controlled trial, Acquired immunodeficiency syndrome (AIDS), law, medicine, Humans, 030212 general & internal medicine, education, Africa South of the Sahara, education.field_of_study, Ritonavir, business.industry, Infant, Newborn, Infant, Obstetrics and Gynecology, Lamivudine, virus diseases, General Medicine, medicine.disease, Infectious Disease Transmission, Vertical, 3. Good health, Clinical trial, Breast Feeding, HIV-1, [SDV.SP.PHARMA]Life Sciences [q-bio]/Pharmaceutical sciences/Pharmacology, Drug Therapy, Combination, Female, Pre-Exposure Prophylaxis, [SDV.SPEE]Life Sciences [q-bio]/Santé publique et épidémiologie, business, Breast feeding, medicine.drug
Abstract: International audience; BACKGROUND: Strategies to prevent postnatal mother-to-child transmission of HIV-1 in Africa, including infant prophylaxis, have never been assessed past 6 months of breastfeeding, despite breastfeeding being recommended up to 12 months after birth. We aimed to compare the efficacy and safety of infant prophylaxis with the two drug regimens (lamivudine or lopinavir-ritonavir) to prevent postnatal HIV-1 transmission up to 50 weeks of breastfeeding.METHODS: We did a randomised controlled trial in four sites in Burkina Faso, South Africa, Uganda, and Zambia in children born to HIV-1-infected mothers not eligible for antiretroviral therapy (CD4 count >350 cells per μL). An independent researcher electronically generated a randomisation schedule; we then used sequentially numbered envelopes to randomly assign (1:1) HIV-1-uninfected breastfed infants aged 7 days to either lopinavir-ritonavir or lamivudine (paediatric liquid formulations, twice a day) up to 1 week after complete cessation of breastfeeding or at the final visit at week 50. We stratified the randomisation by country and used permuted blocks of four and six. We used a study label on drug bottles to mask participants, study physicians, and assessors to the treatment allocation. The primary outcome was infant HIV-1 infection between age 7 days and 50 weeks, diagnosed every 3 months with HIV-1 DNA PCR, in the modified intention-to-treat population (all who attended at least one follow-up visit). This trial is registered with ClinicalTrials.gov, number NCT00640263.FINDINGS: Between Nov 16, 2009, and May 7, 2012, we enrolled and randomised 1273 infants and analysed 1236; 615 assigned to lopinavir-ritonavir or 621 assigned to lamivudine. 17 HIV-1 infections were diagnosed in the study period (eight in the lopinavir-ritonavir group and nine in the lamivudine group), resulting in cumulative HIV-1 infection of 1.4% (95% CI 0.4-2.5) and 1.5% (0.7-2.5), respectively. Infection rates did not differ between the two drug regimens (hazard ratio [HR] of lopinavir-ritonavir versus lamivudine of 0.90, 95% CI 0.35-2.34; p=0.83). Clinical and biological severe adverse events did not differ between groups; 251 (51%) infants had a grade 3-4 event in the lopinavir-ritonavir group compared with 246 (50%) in the lamivudine group.INTERPRETATION: Infant HIV-1 prophylaxis with lopinavir-ritonavir was not superior to lamivudine and both drugs led to very low rates of HIV-1 postnatal transmission for up to 50 weeks of breastfeeding. Infant pre-exposure prophylaxis should be extended until the end of HIV-1 exposure and mothers should be informed about the persistent risk of transmission throughout breastfeeding.FUNDING: INSERM/National Agency for Research on AIDS and Viral Hepatitis (including funds from the Total Foundation), European Developing Countries Clinical Trials Partnership, Research Council of Norway.
Published: 2016
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13. Prevention of mother-to-child HIV transmission: similar access for sub-Sahara African immigrants and for French women?

Author: Carine, Jasseron, Laurent, Mandelbrot, Roland, Tubiana, Jean-Paul, Teglas, Albert, Faye, Catherine, Dollfus, Jerome, Le Chenadec, Christine, Rouzioux, Stephane, Blanche, Josiane, Warszawski, and A, Chacé
Subjects: medicine.medical_specialty, Anti-HIV Agents, Immunology, Breastfeeding, Black People, HIV Infections, Prenatal care, Drug Administration Schedule, Health Services Accessibility, Acquired immunodeficiency syndrome (AIDS), Pregnancy, Antiretroviral Therapy, Highly Active, Epidemiology, Humans, Mass Screening, Immunology and Allergy, Medicine, Prospective Studies, Pregnancy Complications, Infectious, business.industry, Obstetrics, Infant, Newborn, Prenatal Care, Odds ratio, Viral Load, medicine.disease, Drug Utilization, Infectious Disease Transmission, Vertical, Infectious Diseases, Cohort, HIV-1, Female, France, business, Viral load
Abstract: OBJECTIVE : To investigate whether mother-to-child transmission (MTCT) management and rate differed between African immigrants and French-born women delivering in France. METHODS : MTCT strategies were studied among human immunodeficiency virus type 1-infected women delivering between 1984 and 2007 in the multicenter French Perinatal Cohort, according to geographical origin. RESULTS : Among 9245 pregnancies (in 7090 women), the proportion of African mothers increased from 12% in 1984-1986 to 64% in 2003-2004. African women had later access to care than French women, even in recent years (1997-2004). They more often discovered their HIV infection during pregnancy (40.6 vs. 11.5%, P < 0.001), started prenatal care in the third trimester (14.1 vs. 9.8%, P < 0.001) and started antiretroviral therapy after 32 weeks gestation (7.6 vs. 4.1%, P < 0.001). The association with late treatment initiation disappeared when adjusted for late HIV diagnosis and prenatal care (adjusted odds ratio 1.0, 95% confidence interval 0.7-1.4). African and French women did not differ in terms of access to highly active antiretroviral therapy, nor for substandard management such as vaginal delivery with uncontrolled viral load, lack of intrapartum and postpartum treatment or breastfeeding. The MTCT rate was higher for African than for French women receiving antiretroviral therapy (1.8 vs. 0.8%, P = 0.02), but the difference was no longer significant after adjustment for main transmission risk factors (adjusted odds ratio = 1.7, 95% confidence interval 0.8-3.7, P = 0.17). MTCT did not differ among 2110 term deliveries with maternal viral load less than 400 copies/ml, (0.8 vs. 0.6%, P = 0.5). CONCLUSION : African immigrants more often had late HIV screening in pregnancy than French-born women, but had similar access to MTCT prevention, once the infection was diagnosed.
Published: 2008
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14. Prediction of human fetal pharmacokinetics using ex vivo human placenta perfusion studies and physiologically based models

Author: Maïlys, De Sousa Mendes, Deborah, Hirt, Cécile, Vinot, Elodie, Valade, Gabrielle, Lui, Claire, Pressiat, Naïm, Bouazza, Frantz, Foissac, Stephane, Blanche, Minh Patrick, Lê, Gilles, Peytavin, Jean-Marc, Treluyer, Saik, Urien, and Sihem, Benaboud
Subjects: Anti-HIV Agents, Placenta, Fetal Blood, Models, Biological, Perfusion, Fetus, Predictive Value of Tests, Pregnancy, Emtricitabine, Humans, Computer Simulation, Female, Placental Circulation, Pharmacokinetics, Tenofovir, Maternal-Fetal Exchange
Abstract: Pregnant women can be exposed to numerous drugs during the gestational period. For obvious ethical reasons, in vivo studies of fetal exposure to drugs are limited. Information about the transplacental transfer of drugs prior to their administration to pregnant women would be highly useful. In the present study, a novel approach was developed quantitatively predict or to predict the fetal exposure to drugs administered to the mother quantitatively.Transplacental parameters estimated from ex vivo human placenta perfusion experiments were implemented in pregnancy-physiologically based pharmacokinetic (p-PBPK) models in order to predict fetal PK. Thereafter, fetal PK profiles for two antiretroviral drugs, tenofovir (TFV) and emtricitabine (FTC) were simulated. These predictions were then compared to observed cord blood concentrations, to validate these models.Parameters obtained from the ex vivo experiments enabled a good prediction of observed cord blood concentrations without additional a scaling factor. Moreover, a sensitivity analysis showed that fetal predictions were sensitive to changes in transplacental parameters values obtained ex vivo.The integration of ex vivo human placental perfusion parameters in a p-PBPK model should be a promising new approach for predicting human fetal exposure to xenobiotics.
Published: 2015

15. Interruption of cART in clinical practice is associated with an increase in the long-term risk of subsequent immunosuppression in HIV-1-infected children

Author: Camille, Aupiais, Albert, Faye, Jerome, Le Chenadec, Christine, Rouzioux, Naïma, Bouallag, Corinne, Laurent, Stephane, Blanche, Catherine, Dollfus, and Josiane, Warszawski
Subjects: Microbiology (medical), Cart, Male, medicine.medical_specialty, Pediatrics, Adolescent, medicine.medical_treatment, Context (language use), HIV Infections, Cohort Studies, Antiretroviral Therapy, Highly Active, medicine, Immune Tolerance, Humans, Prospective Studies, Intensive care medicine, Prospective cohort study, Child, Withholding Treatment, business.industry, Infant, Newborn, virus diseases, Infant, Immunosuppression, Viral Load, Long term risk, Infectious Diseases, Treatment Outcome, Anti-Retroviral Agents, Child, Preschool, Pediatrics, Perinatology and Child Health, Female, France, business, Viral load, Cohort study
Abstract: Antiretroviral treatment interruption (TI) is not recommended in HIV-infected children. We aimed to evaluate the context and consequences of TI in clinical practice.We investigated the probability and risk factors of a first TI in the 483 children treated with combined ART (cART) in the ANRS French national pediatric cohort. Immunologic and virologic outcomes were compared between patients with TI (TI group) and those on continuous treatment (matched control group), from a baseline defined as the age at first interruption for the TI child and the corresponding age for the control child.At least one TI ≥ 3 months occurred in 42.4% of patients, at a median age of 8.0 years, for a median duration of 12.1 months. After cART initiation, the risk of TI was 7.0% (5.0-9.6) at 1 year and 30.3% (26.1-35.0) at 5 years and was higher for children starting treatment before 2000 and for children starting cART before 6 months of age. AIDS-free survival was similar, but severe immunosuppression occurred earlier in the TI group than in the control group (adjusted HR = 3.1; 1.0-9.1; P = 0.04). Four years after baseline, the proportion of patients with CD4% ≥ 25% was lower in the TI group than in the control group (52.0% vs. 72.0%; P0.01), even among children restarting cART at least 6 months earlier (aRR = 0.5; 0.3-0.9; P = 0.03).The risk of TI in clinical practice has decreased but remains high. In intent-to-treat analysis, TI was associated with a greater risk of subsequent immunosuppression, even after cART resumption.
Published: 2014

16. X-linked lymphoproliferative disease due to SAP/SH2D1A deficiency: a multicenter study on the manifestations, management and outcome of the disease

Author: Claire, Booth, Kimberly C, Gilmour, Paul, Veys, Andrew R, Gennery, Mary A, Slatter, Helen, Chapel, Paul T, Heath, Colin G, Steward, Owen, Smith, Anna, O'Meara, Hilary, Kerrigan, Nizar, Mahlaoui, Marina, Cavazzana-Calvo, Alain, Fischer, Despina, Moshous, Stephane, Blanche, Jana, Pachlopnik Schmid, Jana, Pachlopnick-Schmid, Sylvain, Latour, Genevieve, de Saint-Basile, Michael, Albert, Gundula, Notheis, Nikolaus, Rieber, Brigitte, Strahm, Henrike, Ritterbusch, Arjan, Lankester, Nico G, Hartwig, Isabelle, Meyts, Alessandro, Plebani, Annarosa, Soresina, Andrea, Finocchi, Claudio, Pignata, Emilia, Cirillo, Sonia, Bonanomi, Christina, Peters, Krzysztof, Kalwak, Srdjan, Pasic, Petr, Sedlacek, Janez, Jazbec, Hirokazu, Kanegane, Kim E, Nichols, I Celine, Hanson, Neena, Kapoor, Elie, Haddad, Morton, Cowan, Sharon, Choo, Joanne, Smart, Peter D, Arkwright, Hubert B, Gaspar, Pediatrics, Booth, C., Gilmour, K. C., Veys, P., Gennery, A. R., Slatter, M. A., Chapel, H., Heath, P. T., Steward, C. G., Smith, O., O'Meara, A., Kerrigan, H., Mahlaoui, N., Cavazzana Calvo, M., Fischer, A., Moshous, D., Blanche, S., Pachlopnik Schmid, J., Latour, S., de Saint Basile, G., Albert, M., Notheis, G., Rieber, N., Strahm, B., Ritterbusch, H., Lankester, A., Hartwig, N. G., Meyts, I., Plebani, A., Soresina, A., Finocchi, A., Pignata, Claudio, Cirillo, E., Bonanomi, S., Peters, C., Kalwak, K., Pasic, S., Sedlacek, P., Jazbec, J., Kanegane, H., Nichols, K. E., Hanson, I. C., Kapoor, N., Haddad, E., Cowan, M., Choo, S., Smart, J., Arkwright, P. D., and Gaspar, H. B.
Subjects: Male, Pediatrics, Epstein-Barr Virus Infections, Herpesvirus 4, Human, Clinical Trials and Observations, medicine.medical_treatment, Hematopoietic stem cell transplantation, medicine.disease_cause, Biochemistry, 0302 clinical medicine, Signaling Lymphocytic Activation Molecule Family Member 1, hemic and lymphatic diseases, Antineoplastic Combined Chemotherapy Protocols, Medicine, X-Linked Lymphoproliferative Syndrome, Signaling Lymphocytic Activation Molecule Associated Protein, Child, Immunodeficiency, 0303 health sciences, Hematopoietic Stem Cell Transplantation, Intracellular Signaling Peptides and Proteins, Hematology, Middle Aged, 3. Good health, Survival Rate, Child, Preschool, Female, SAP, Adult, medicine.medical_specialty, Adolescent, Immunology, Lymphoproliferative disorders, Receptors, Cell Surface, XLP, SAP, Lymphohistiocytosis, Hemophagocytic, 03 medical and health sciences, Young Adult, Antigens, CD, XLP, Humans, Survival rate, 030304 developmental biology, Settore MED/38 - Pediatria Generale e Specialistica, Hemophagocytic lymphohistiocytosis, business.industry, Infant, Newborn, X-linked lymphoproliferative disease, Infant, Cell Biology, Immune dysregulation, medicine.disease, stem-cell transplantation barr-virus infection hemophagocytic lymphohistiocytosis cutting edge t-cells lymphocytic vasculitis encoding gene sap activation mononucleosis, Lymphoproliferative Disorders, Mutation, business, 030215 immunology
Abstract: X-linked lymphoproliferative disease (XLP1) is a rare immunodeficiency characterized by severe immune dysregulation and caused by mutations in the SH2D1A/SAP gene. Clinical manifestations are varied and include hemophagocytic lymphohistiocytosis (HLH), lymphoma and dysgammaglobulinemia, often triggered by Epstein-Barr virus infection. Historical data published before improved treatment regimens shows very poor outcome. We describe a large cohort of 91 genetically defined XLP1 patients collected from centers worldwide and report characteristics and outcome data for 43 patients receiving hematopoietic stem cell transplant (HSCT) and 48 untransplanted patients. The advent of better treatment strategies for HLH and malignancy has greatly reduced mortality for these patients, but HLH still remains the most severe feature of XLP1. Survival after allogeneic HSCT is 81.4% with good immune reconstitution in the large majority of patients and little evidence of posttransplant lymphoproliferative disease. However, survival falls to 50% in patients with HLH as a feature of disease. Untransplanted patients have an overall survival of 62.5% with the majority on immunoglobulin replacement therapy, but the outcome for those untransplanted after HLH is extremely poor (18.8%). HSCT should be undertaken in all patients with HLH, because outcome without transplant is extremely poor. The outcome of HSCT for other manifestations of XLP1 is very good, and if HSCT is not undertaken immediately, patients must be monitored closely for evidence of disease progression.
Published: 2011

17. Perinatal acquisition of drug-resistant HIV-1 infection: mechanisms and long-term outcome

Author: Constance, Delaugerre, Marie-Laure, Chaix, Stephane, Blanche, Josiane, Warszawski, Dorine, Cornet, Catherine, Dollfus, Veronique, Schneider, Marianne, Burgard, Albert, Faye, Laurent, Mandelbrot, Roland, Tubiana, Christine, Rouzioux, S, Tilouche, BMC, Ed., Infections à Vih, Réservoirs, Pharmacologie des Antirétroviraux et Prévention de la Transmission Mère Enfant, Université Paris Descartes - Paris 5 (UPD5), Laboratoire de Virologie [CHU Necker], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Service d'immuno-hématologie pédiatrique [CHU Necker], Santé reproductive, sexualité, infection à VIH - épidémiologie, démographie, sciences sociales, Institut national d'études démographiques (INED)-Université Paris-Sud - Paris 11 (UP11)-Institut National de la Santé et de la Recherche Médicale (INSERM), CHU Trousseau [APHP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), CHU Tenon [AP-HP], Service d'hématologie et immunologie pédiatrique, Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Robert Debré-Université Paris Diderot - Paris 7 (UPD7), Service de gynécologie obstétrique, Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Louis Mourier - AP-HP [Colombes], Service de Maladies Infectieuses et Tropicales [CHU Pitié-Salpêtrière], CHU Pitié-Salpêtrière [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), ANRS French Perinatal Cohort, Université Paris Descartes - Paris 5 ( UPD5 ), Assistance publique - Hôpitaux de Paris (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Institut national d'études démographiques ( INED ) -Université Paris-Sud - Paris 11 ( UP11 ) -Institut National de la Santé et de la Recherche Médicale ( INSERM ), Service de pédiatrie et oncologie, Assistance publique - Hôpitaux de Paris (AP-HP)-CHU Trousseau [APHP], Service de virologie, Assistance publique - Hôpitaux de Paris (AP-HP)-CHU Tenon [APHP], Assistance publique - Hôpitaux de Paris (AP-HP)-Hôpital Robert Debré-Université Paris Diderot - Paris 7 ( UPD7 ), Assistance publique - Hôpitaux de Paris (AP-HP)-Hopital Louis Mourier - AP-HP [Colombes], Service des maladies infectieuses et tropicales [CHU Pitié-Salpêtrière], Assistance publique - Hôpitaux de Paris (AP-HP)-CHU Pitié-Salpêtrière [APHP], CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), and Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)
Subjects: Pediatrics, MESH: Chemoprevention, HIV Infections, MESH : Genotype, Drug resistance, MESH: Genotype, MESH: HIV-1, 0302 clinical medicine, MESH: Pregnancy, Pregnancy, [SDV.MHEP.MI]Life Sciences [q-bio]/Human health and pathology/Infectious diseases, MESH : Chemoprevention, MESH : Female, 030212 general & internal medicine, MESH : DNA, Viral, Pregnancy Complications, Infectious, MESH : Infectious Disease Transmission, Vertical, MESH: Treatment Outcome, 0303 health sciences, education.field_of_study, Maternal Transmission, MESH : Prognosis, MESH: Drug Resistance, Viral, MESH: Infant, Newborn, MESH : Infant, MESH: HIV Infections, Prognosis, MESH: Infant, 3. Good health, MESH : Antiviral Agents, MESH: Infectious Disease Transmission, Vertical, MESH : Drug Resistance, Viral, [ SDV.MHEP.MI ] Life Sciences [q-bio]/Human health and pathology/Infectious diseases, Infectious Diseases, Treatment Outcome, MESH: RNA, Viral, Cohort, [SDV.MHEP.MI] Life Sciences [q-bio]/Human health and pathology/Infectious diseases, RNA, Viral, Female, MESH : HIV-1, lcsh:Immunologic diseases. Allergy, MESH: Antiviral Agents, medicine.medical_specialty, Genotype, Population, MESH : Treatment Outcome, Biology, MESH : Infant, Newborn, Antiviral Agents, Chemoprevention, MESH: Prognosis, 03 medical and health sciences, Pharmacotherapy, Virology, Drug Resistance, Viral, medicine, MESH : HIV Infections, Humans, MESH : RNA, Viral, MESH: Pregnancy Complications, Infectious, education, Genotyping, MESH: Humans, 030306 microbiology, Research, MESH : Humans, Infant, Newborn, Infant, medicine.disease, Reverse transcriptase, Infectious Disease Transmission, Vertical, MESH: DNA, Viral, MESH : Pregnancy, MESH : Pregnancy Complications, Infectious, Immunology, DNA, Viral, HIV-1, lcsh:RC581-607, MESH: Female
Abstract: Background Primary-HIV-1-infection in newborns that occurs under antiretroviral prophylaxis that is a high risk of drug-resistance acquisition. We examine the frequency and the mechanisms of resistance acquisition at the time of infection in newborns. Patients and Methods We studied HIV-1-infected infants born between 01 January 1997 and 31 December 2004 and enrolled in the ANRS-EPF cohort. HIV-1-RNA and HIV-1-DNA samples obtained perinatally from the newborn and mother were subjected to population-based and clonal analyses of drug resistance. If positive, serial samples were obtained from the child for resistance testing. Results Ninety-two HIV-1-infected infants were born during the study period. Samples were obtained from 32 mother-child pairs and from another 28 newborns. Drug resistance was detected in 12 newborns (20%): drug resistance to nucleoside reverse transcriptase inhibitors was seen in 10 cases, non-nucleoside reverse transcriptase inhibitors in two cases, and protease inhibitors in one case. For 9 children, the detection of the same resistance mutations in mothers' samples (6 among 10 available) and in newborn lymphocytes (6/8) suggests that the newborn was initially infected by a drug-resistant strain. Resistance variants were either transmitted from mother-to-child or selected during subsequent temporal exposure under suboptimal perinatal prophylaxis. Follow-up studies of the infants showed that the resistance pattern remained stable over time, regardless of antiretroviral therapy, suggesting the early cellular archiving of resistant viruses. The absence of resistance in the mother of the other three children (3/10) and neonatal lymphocytes (2/8) suggests that the newborns were infected by a wild-type strain without long-term persistence of resistance when suboptimal prophylaxis was stopped. Conclusion This study confirms the importance of early resistance genotyping of HIV-1-infected newborns. In most cases (75%), drug resistance was archived in the cellular reservoir and persisted during infancy, with or without antiretroviral treatment. This finding stresses the need for effective antiretroviral treatment of pregnant women.
Published: 2009
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18. Sustained remission after haploidentical bone marrow transplantation in a child with refractory systemic juvenile idiopathic arthritis

Author: Guillaume Morelle, Martin Castelle, Graziella Pinto, Sylvain Breton, Matthieu Bendavid, Charlotte Boussard, Richard Mouy, Brigitte Bader-Meunier, Michaela Semeraro, Albert Faye, Marina Cavazzana, Bénédicte Neven, Stéphane Blanche, Pierre Quartier, and Despina Moshous
Subjects: Systemic juvenile idiopathic arthritis, Still’s disease, Allogenic hematopoietic stem cell transplantation, Graft versus host disease, Autoimmunity, Pediatrics, RJ1-570, Diseases of the musculoskeletal system, RC925-935
Abstract: Abstract Background Some patients with systemic juvenile idiopathic arthritis (SJIA) and severe, refractory disease achieved remission through intensive immunosuppressive treatment followed by autologous hematopoietic stem cell transplantation (HSCT). However, disease relapsed in most cases. More recently selected SJIA patients received allogenic HSCT from a HLA-identical sibling or a HLA matched unrelated donor. While most transplanted patients achieved sustained SJIA remission off-treatment, the procedure-related morbidity was high. Case report A girl presented SJIA with a severe disease course since the age of 15 months. She was refractory to the combination of methotrexate and steroids to anti-interleukin (IL)-1, then anti-IL-6, tumor necrosis factor alpha inhibitors, and thalidomide. Given the high disease burden and important treatment-related toxicity the indication for a haploidentical HSCT from her mother was validated, as no HLA matched donor was available. The patient received a T replete bone marrow graft at the age of 3.7 years. Conditioning regimen contained Rituximab, Alemtuzumab, Busulfan, and Fludarabine. Cyclophosphamide at D + 3 and + 4 post HSCT was used for graft-versus-host-disease prophylaxis, followed by Cyclosporin A and Mycophenolate Mofetil. Post HSCT complications included severe infections, grade 3 intestinal graft-versus-host-disease, autoimmune thyroiditis, and immune thrombocytopenia. Three years after HSCT, the child is alive and well, notwithstanding persistent hypothyroidy requiring substitution. Immune thrombocytopenia had resolved. Most importantly, SJIA was in complete remission, off immunosuppressive drugs. Conclusion Allogenic HSCT may be a therapeutic option, even with a HLA haplo-identical alternative donor, in patients with inflammatory diseases such as SJIA. Despite increased experience with this treatment, the risk of life-threatening complications restrains its indication to selected patients with severe, refractory disease.
Published: 2021
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19. Analysis of risk factors for myelodysplasias, leukemias and death from infection among patients with congenital neutropenia. Experience of the French Severe Chronic Neutropenia Study Group

Author: Jean, Donadieu, Thierry, Leblanc, Brigitte, Bader Meunier, Mohamed, Barkaoui, Odile, Fenneteau, Yves, Bertrand, Micheline, Maier-Redelsperger, Marguerite, Micheau, Jean Louis, Stephan, Noel, Phillipe, Pierre, Bordigoni, Annie, Babin-Boilletot, Philippe, Bensaid, Anne Marie, Manel, Etienne, Vilmer, Isabelle, Thuret, Stephane, Blanche, Eliane, Gluckman, Alain, Fischer, Françoise, Mechinaud, Bertrand, Joly, Thierry, Lamy, Olivier, Hermine, Bruno, Cassinat, Christine, Bellanné-Chantelot, and Christine, Chomienne
Subjects: Adult, Male, Leukemia, Neutropenia, Adolescent, Infant, Newborn, Infant, Risk Factors, Child, Preschool, Myelodysplastic Syndromes, Sepsis, Acute Disease, Granulocyte Colony-Stimulating Factor, Humans, Female, Prospective Studies, Child
Abstract: The two main complications of severe chronic neutropenia are fatal sepsis and myelodysplasia/acute leukemia (MDS/AL). Granulocyte colony-stimulating factor (G-CSF) therapy has significantly reduced the frequency and severity of infections, but its possible influence on the risk of malignancy is not known.The French Severe Chronic Neutropenia (SCN) Registry has prospectively collected data since 1994 on 231 patients with various forms of SCN, namely severe congenital neutropenia (n=101), cyclic neutropenia (n=60), glycogen storage disease type Ib (GSDIb) (n=15) and Shwachman-Diamond syndrome (SDS)(n=55). The median overall follow-up is 11.1 years. Parameters of exposure to G-CSF therapy, such as the time averaged dose, follow up after first use of G-CSF, and the cumulative dose, have been recorded.Eight septic deaths occurred, of which 6 among patients with severe congenital neutropenia and 2 in patients with cyclic neutropenia; none of these 8 patients was receiving G-CSF therapy. No septic deaths occurred during G-CSF therapy. Thirteen cases of MDS/AL were recorded. The cumulative incidence of MDS/AL was 2.7% (SD 1.3%) at 10 years and 8.1% (SD 2.7%) at 20 years.Risk factors for MDS/AL were the diagnostic category, the severity of neutropenia, younger age at diagnosis, and strong exposure to G-CSF. MDS/AL only occurred in patients with severe congenital neutropenia and SDS. Owing to their particular susceptibility to infections, patients with severe congenital neutropenia had the strongest exposure to G-CSF; the risk of leukemia increased with the degree of G-CSF exposure in this subgroup.
Published: 2005

20. P2-761 Complete adrenocorticotropic hormone and growth hormone deficiency associated with combined immunodeficiency: A novel entity

Author: Papadimitriou, Dimitrios T, Pagnier, Anne, Spiteri, Anne, Marie-Christine Jacob, Spada, Anna, Stephane Blanche, and Thierr Y Brue
Published: 2005
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21. Mini review: Prevention of mother–child transmission of HIV: 25 years of continuous progress toward the eradication of pediatric AIDS?

Author: Stéphane Blanche
Subjects: hiv, pregnancy, children, antiretroviral, prophylaxis, Infectious and parasitic diseases, RC109-216
Abstract: Prevention of mother-to-child transmission with antiretrovirals is extraordinarily effective. When medically well followed, a mother living with human immunodeficiency virus can now expect to avoid transmitting the virus to her child. Despite the immense difficulties inherent in the global implementation of this treatment, the virtual disappearance of pediatric AIDS can be considered in the long term.
Published: 2020
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22. Bayesian Modeling Immune Reconstitution Apply to CD34+ Selected Stem Cell Transplantation for Severe Combined Immunodeficiency

Author: Jean-Sebastien Diana, Naïm Bouazza, Chloe Couzin, Martin Castelle, Alessandra Magnani, Elisa Magrin, Jeremie Rosain, Jean-Marc Treluyer, Capucine Picard, Despina Moshous, Stéphane Blanche, Bénédicte Neven, and Marina Cavazzana
Subjects: Bayesian prediction algorithm, immune reconstitution, severe combined immunodeficiency (SCID), hematopoietic stem cell transplantation, CD34+ selection, Pediatrics, RJ1-570
Abstract: Severe combined immunodeficiencies (SCIDs) correspond to the most severe form of primary immunodeficiency. Allogeneic hematopoietic stem cell transplantation (HSCT) and gene therapy are curative treatments, depending on the donor's availability and molecular diagnostics. A partially human leukocyte antigen (HLA)-compatible donor used has been developed for this specific HSCT indication in the absence of a matched donor. However, the CD34+ selected process induces prolonged post-transplant T-cell immunodeficiency. The aim here was to investigate a modeling approach to predict the time course and the extent of CD4+ T-cell immune reconstitution after CD34+ selected transplantation. We performed a Bayesian approach based on the age-related changes in thymic output and the cell proliferation/loss model. For that purpose, we defined specific individual covariates from the data collected from 10 years of clinical practice and then evaluated the model's predicted performances and accuracy. We have shown that this Bayesian modeling approach predicted the time course and extent of CD4+ T-cell immune reconstitution after SCID transplantation.
Published: 2022
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23. A Mendelian Predisposition to B Cell Lymphoma Caused by IL-10R2 Deficiency

Author: Benedicte Neven, Emilie Mamessier, Julie Bruneau, Sophie Kaltenbach, Felipe Suarez, Francois Doz, Daniel Orbach, Veronique Minard, Pierre Frange, Danielle Canioni, Isabelle Radford-Weiss, Vahid Asnafi, jean-Laurent Casanova, Stephane Blanche, Capucine Picard, Frederic Rieux-Laucat, Frederic Davi, Nicole Brousse, Bertrand Nadel, Frank Ruemmele, and Alain Fischer
Subjects: Immunology, Cell Biology, Hematology, Biochemistry
Abstract: Abstract 5092 Background The fact that monogenic interleukin (IL)-10 and IL-10 receptor deficiencies cause very early-onset, severe inflammatory bowel disease demonstrates the IL-10 pathway's crucial role in preventing microbiota-driven gut inflammation. However, no other severe complications of IL-10/IL-10R deficiencies have been reported to date. Methods A cohort of six patients with IL-10/IL-10R deficiency was retrospectively surveyed for phenotypic expression. The identification of a number of cases of lymphoma prompted an in-depth characterization of available biopsy material, with immunohistochemical staining, cytogenetic studies and gene expression profiling. Results Four patients (all with a complete IL-10R2 deficiency) had developed B cell non-Hodgkin's lymphoma between the ages of 5 and 6 years. Cytogenetic and IgH clonality analyses suggested that one of the patients developed at least 3 distinct lymphomas. The patients' tumors had the characteristics of diffuse large B cell lymphomas and contained monoclonal, Epstein-Barr-virus-negative germinal center B cells. Nuclear expression of the NF-κB factor c-REL was detected in all of the six lymphomas tested. Conclusions IL-10R deficiency is associated with a high risk of developing a B cell lymphoma with an unusual phenotype. Our results highlight an unexpected role of the IL-10R pathway in the control of lymphomagenesis. Disclosures: No relevant conflicts of interest to declare.
Published: 2012
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24. Impact of Early Versus Late Antiretroviral Treatment Initiation on Naive T Lymphocytes in HIV-1-Infected Children and Adolescents – The-ANRS-EP59-CLEAC Study

Author: Pierre Frange, Thomas Montange, Jérôme Le Chenadec, Damien Batalie, Ingrid Fert, Catherine Dollfus, Albert Faye, Stéphane Blanche, Anne Chacé, Corine Fourcade, Isabelle Hau, Martine Levine, Nizar Mahlaoui, Valérie Marcou, Marie-Dominique Tabone, Florence Veber, Alexandre Hoctin, Thierry Wack, Véronique Avettand-Fenoël, Josiane Warszawski, and Florence Buseyne
Subjects: HIV-1, children, adolescents, early ART, T lymphocyte, naive T lymphocyte, Immunologic diseases. Allergy, RC581-607
Abstract: BackgroundThe early initiation of antiretroviral therapy (ART) in HIV-1-infected infants reduces mortality and prevents early CD4 T-cell loss. However, the impact of early ART on the immune system has not been thoroughly investigated in children over five years of age or adolescents. Here, we describe the levels of naive CD4 and CD8 T lymphocytes (CD4/CD8TN), reflecting the quality of immune reconstitution, as a function of the timing of ART initiation (early (
Published: 2021
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25. Different Clinical Presentations and Outcomes of Disseminated Varicella in Children With Primary and Acquired Immunodeficiencies

Author: Paul Bastard, Aurélien Galerne, Alain Lefevre-Utile, Coralie Briand, André Baruchel, Philippe Durand, Judith Landman-Parker, Elodie Gouache, Nathalie Boddaert, Despina Moshous, Joel Gaudelus, Robert Cohen, Georges Deschenes, Alain Fischer, Stéphane Blanche, Loïc de Pontual, and Bénédicte Neven
Subjects: varicella, primary immumunodeficiencies, steroids, innate immunity, disseminated varicella, Immunologic diseases. Allergy, RC581-607
Abstract: Primary infection with varicella-zoster virus (VZV) causes chickenpox, a benign and self-limited disease in healthy children. In patients with primary or acquired immunodeficiencies, primary infection can be life-threatening, due to rapid dissemination of the virus to various organs [lung, gastrointestinal tract, liver, eye, central nervous system (CNS)]. We retrospectively described and compared the clinical presentations and outcomes of disseminated varicella infection (DV) in patients with acquired (AID) (n= 7) and primary (PID) (n= 12) immunodeficiencies. Patients with AID were on immunosuppression (mostly steroids) for nephrotic syndrome, solid organ transplantation or the treatment of hemopathies, whereas those with PID had combined immunodeficiency (CID) or severe CID (SCID). The course of the disease was severe and fulminant in patients with AID, with multiple organ failure, no rash or a delayed rash, whereas patients with CID and SICD presented typical signs of chickenpox, including a rash, with dissemination to other organs, including the lungs and CNS. In the PID group, antiviral treatment was prolonged until immune reconstitution after bone marrow transplantation, which was performed in 10/12 patients. Four patients died, and three experienced neurological sequelae. SCID patients had the worst outcome. Our findings highlight substantial differences in the clinical presentation and course of DV between children with AID and PID, suggesting differences in pathophysiology. Prevention, early diagnosis and treatment are required to improve outcome.
Published: 2020
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26. Safety and efficacy of brentuximab vedotin as a treatment for lymphoproliferative disorders in primary immunodeficiencies

Author: Thomas Pincez, Julie Bruneau, Laureline Berteloot, Eve Piekarski, Caroline Thomas, Ambroise Marçais, Amélie Trinquand, Martin Castelle, Nicolas Garcelon, Dominique Plantaz, Morgane Cheminant, Despina Moshous, Thierry Jo Molina, Olivier Hermine, Elizabeth Macintyre, Alain Fischer, Stéphane Blanche, Felipe Suarez, and Bénédicte Neven
Subjects: Diseases of the blood and blood-forming organs, RC633-647.5
Published: 2020
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27. An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme

Author: Christelle Rémus, Aurélie Stanislas, Naïm Bouazza, Valérie Gauthereau, Michel Polak, Stéphane Blanche, Assa Niakaté, Eliane Gluckman, Jean-Marc Tréluyer, Arnold Munnich, Robert Girot, and Marina Cavazzana
Subjects: sickle cell disease, sickle cell disease trait, parental notification, screening, follow-up, phone call, Pediatrics, RJ1-570
Abstract: Aim: Sickle cell disease (SCD) is the most frequent monogenic disease worldwide; ~5–7% of the world population carry a hemoglobin disorder trait. In the US, one in every 1,941 newborns has SCD, whereas one in every 3,000 newborns in France is affected - resulting in 385 new cases and 5,883 newly identified carriers per year. The objective of the present study was to evaluate three different ways of providing information to parents at risk of having a child with SCD, with a view to increasing the parental screening rate and decreasing the number of new cases per year in France.Method: In a randomized study, we contacted 300 couples of parents after their child had been identified as a SCD carrier in the French national newborn screening programme: 100 couples received an information letter (the standard procedure in France: arm A), 100 couples received a letter and then a follow-up phone call (arm B), and 100 received a letter and then three follow-up text messages at 5-day intervals (arm C). The primary endpoint was the number of parents in each arm screened in the 120 days after the letter had been sent. In a modified intention-to-treat analysis, the screening rate was 17% in arm A, 35% in arm B, and 30% in arm C.Results: Telephone and text message follow-ups were associated with higher screening rates, compared with no follow-up. After being informed of their child's carrier status, some parents had consulted a healthcare professional but had not been referred for screening (16% in arm A, 19% in arm B, and 13% in arm C).Conclusion: A letter followed by a phone call or three text messages is more effective than a letter alone for informing parents at risk of having a child with SCD. The effective implementation of this follow-up programme probably requires better training of all the healthcare professionals involved.
Published: 2020
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28. Not All Tetramer Binding CD8+ T Cells Can Produce Cytokines and Chemokines Involved in the Effector Functions of Virus-Specific CD8+ T Lymphocytes in HIV-1 Infected Children.

Author: DANIEL SCOTT-ALGARA, FLORENCE BUSEYNE, FRANÇOISE PORROT, BEATRICE CORRE, NASSIMA BELLAL, CHRISTINE ROUZIOUX, STEPHANE BLANCHE, and YVES RIVIERE
Abstract: Abstract In the pediatric human immunodeficiency virus type-1 (HIV-1) infection, the presence of cytotoxic T lymphocytes (CTL) is associated with a slow progression to AIDS. The secretion of cytokines by CTLs may be critical in the control of viral infection. We used the combination of cell surface and intracellular staining to study the functionality of tetramer binding CD8+ T cells recognizing two HIV-1 immunodominant epitopes, in peripheral blood mononuclear cells from HIV-1-infected children. A fraction of tetramer positive CD8+ T cells produce cytokines (IFN-?, TNF-a) or chemokines (CCL4, CCL5) after ex vivo stimulation with the cognate peptide. There was a negative correlation between the plasma viral load and the percentage of CD8+ Tetramer Gag+ T cells secreting IFN-?. This is the first report in the context of pediatric HIV-1 infection showing that only a fraction of HIV-1-specific CD8+ T cells have the capacity to produce cytokines and chemokines implicated in their antiviral functions. [ABSTRACT FROM AUTHOR]
Published: 2005

29. Proceedings of the 23rd Paediatric Rheumatology European Society Congress: part one

Author: F. De Benedetti, J. Anton, M. Gattorno, H. Lachmann, I. Kone-Paut, S. Ozen, J. Frenkel, A. Simon, A. Zeft, E. Ben-Chetrit, H. M. Hoffman, Y. Joubert, K. Lheritier, A. Speziale, J. Guido, Roberta Caorsi, Federica Penco, Alice Grossi, Antonella Insalaco, Maria Alessio, Giovanni Conti, Federico Marchetti, Alberto Tommasini, Silvana Martino, Romina Gallizzi, Annalisa Salis, Francesca Schena, Francesco Caroli, Alberto Martini, Gianluca Damonte, Isabella Ceccherini, Marco Gattorno, Marie-Louise Frémond, Carolina Uggenti, Lien Van Eyck, Isabelle Melki, Darragh Duffy, Vincent Bondet, Yoann Rose, Bénédicte Neven, Yanick Crow, Mathieu P. Rodero, Yvonne Kusche, Johannes Roth, Katarzyna Barczyk-Kahlert, Giovanna Ferrara, Annalisa Chiocchetti, Silvio Polizzi, Josef Vuch, Diego Vozzi, Anna Mondino, Erica Valencic, Serena Pastore, Andrea Taddio, Flavio Faletra, Umberto Dianzani, Ugo Ramenghi, Qing Zhou, Xiaomin Yu, Erkan Demirkaya, Natalie Deuitch, Deborah Stone, Wanxia Tsai, Amanda Ombrello, Tina Romeo, Elaine F. Remmers, JaeJin Chae, Massimo Gadina, Steven Welch, Seza Ozen, Rezan Topaloglu, Mario Abinun, Daniel L. Kastner, Ivona Aksentijevich, Donatella Vairo, Rosalba Monica Ferraro, Giulia Zani, Jessica Galli, Micaela De Simone, Marco Cattalini, Elisa Fazzi, Silvia Giliani, Ebun Omoyinmi, Ariane Standing, Dorota Rowczenio, Annette Keylock, Sonia Melo Gomes, Fiona Price-Kuehne, Sira Nanthapisal, Claire Murphy, Thomas Cullup, Lucy Jenkins, Kimberly Gilmour, Despina Eleftheriou, Helen Lachmann, Philip Hawkins, Nigel Klein, Paul Brogan, Anita Dhanrajani, Mercedes Chan, Stephanie Pau, Janet Ellsworth, Jaime Guzman, Florence A. Aeschlimann, Marinka Twilt, Simon W. Eng, Shehla Sheikh, Ronald M. Laxer, Diane Hebert, Damien Noone, Christian Pagnoux, Susanne M. Benseler, Rae S. Yeung, Christoph Kessel, Katrin Lippitz, Toni Weinhage, Claas Hinze, Helmut Wittkowski, Dirk Holzinger, Niklas Grün, Dirk Föll, Pieter Van Dijkhuizen, Federica Del Chierico, Clara Malattia, Alessandra Russo, Denise Pires Marafon, Nienke M. ter Haar, Silvia Magni-Manzoni, Sebastiaan J. Vastert, Bruno Dallapiccola, Berent Prakken, Fabrizio De Benedetti, Lorenza Putignani, Berna Eren Fidanci, Kenan Barut, Serap Arıcı, Dogan Simsek, Mustafa Cakan, Ezgi D. Batu, Sezgin Şahin, Ayşenur Kısaarslan, Ebru Yilmaz, Özge Basaran, Ferhat Demir, Kubra Ozturk, Zübeyde Gunduz, Betül Sozeri, Balahan Makay, Nuray Ayaz, Onder Yavascan, Ozlem Aydog, Yelda Bilginer, Zelal Ekinci, Dilek Yıldız, Faysal Gök, Muferret Erguven, Erbil Unsal, Ozgur Kasapcopur, For the FMF Arthritis Vasculitis and Orphan Disease Research in Paediatric Rheumatology (FAVOR), Hafize E. Sönmez, Betül Sözeri, Yonatan Butbul, Seza Özen, Claudia Bracaglia, Giusi Prencipe, Manuela Pardeo, Geneviève Lapeyre, Emiliano Marasco, Walter Ferlin, Robert Nelson, Cristina de Min, N. Ruperto, H. I. Brunner, P. Quartier, T. Constantin, E. Alexeeva, K. Marzan, N. Wulffraat, R. Schneider, S. Padeh, V. Chasnyk, C. Wouters, J. B. Kuemmerle-Deschner, T. Kallinich, B. Lauwerys, E. Haddad, E. Nasonov, M. Trachana, O. Vougiouka, K. Leon, E. Vritzali, A. Martini, D. Lovell, PRINTO/PRCSG, Stefano Volpi, Claudia Pastorino, Francesca Kalli, Alessia Omenetti, Sabrina Chiesa, Arinna Bertoni, Paolo Picco, Gilberto Filaci, Elisabetta Traggiai, Marie-Louise Fremond, Naoki Kitabayashi, Olivero Sacco, Isabelle Meyts, Marie-Anne Morren, Carine Wouters, Eric Legius, Isabelle Callebaut, Christine Bodemer, Frederic Rieux-Laucat, Mathieu Rodero, Nadia Jeremiah, Alexandre Belot, Eric Jeziorski, Didier Bessis, Guilhem Cros, Gillian I. Rice, Bruno Charbit, Anne Hulin, Nihel Khoudour, Consuelo Modesto Caballero, Monique Fabre, Laureline Berteloot, Muriel Le Bourgeois, Philippe Reix, Thierry Walzer, Despina Moshous, Stéphane Blanche, Alain Fischer, Brigitte Bader-Meunier, Frédéric Rieux-Laucat, K. Annink, N. ter Haar, S. Al-Mayouf, G. Amaryan, K. Barron, S. Benseler, P. Brogan, L. Cantarini, M. Cattalini, A. Cochino, F. Dedeoglu, A. De Jesus, O. Dellacasa, E. Demirkaya, P. Dolezalova, K. Durrant, G. Fabio, R. Gallizzi, R. Goldbach-Mansky, E. Hachulla, V. Hentgen, T. Herlin, M. Hofer, H. Hoffman, A. Insalaco, A. Jansson, I. Koné-Paut, A. Kozlova, J. Kuemmerle-Deschner, R. Laxer, S. Nielsen, I. Nikishina, A. Ombrello, E. Papadopoulou-Alataki, A. Ravelli, D. Rigante, R. Russo, Y. Uziel, Nienke ter Haar, Jerold Jeyaratnam, Anna Simon, Matteo Doglio, Jordi Anton, Consuelo Modesto, Pierre Quartier, Esther Hoppenreijs, Luca Cantarini, Loredana Lepore, Inmaculada Calvo Penades, Christina Boros, Rita Consolini, Donato Rigante, Ricardo Russo, Jana Pachlopnik Schmid, Thirusha Lane, Nicolino Ruperto, Joost Frenkel, Chiara Passarelli, Elisa Pisaneschi, Virginia Messia, Antonio Novelli, Fabrizio Debenedetti, P. A. Brogan, X. Wei, Martina Finetti, Francesca Orlando, Elisabetta Cortis, Angela Miniaci, Nicola Ruperto, Charlotte Eijkelboom, Pavla Dolezalova, Isabelle Koné-Paut, Marija Jelusic-Drazic, Liliana Bezrodnik, Mari Carmen Pinedo, Valda Stanevicha, Marielle van Gijn, Silvia Federici, Hermann Girschick, Gerd Ganser, Susan Nielsen, Troels Herlin, Sulaiman Mohammed Al-Mayouf, Michael Hofer, Jasmin Kuemmerle-Deschner, Susanne Schalm, Annette Jansson, on behalf of PRINTO and Eurofever registry, Marta Marchi, Chiara Marini, Angelo Ravelli, Alberto Garaventa, Sonia Carta, Enrica Balza, Patrizia Castellani, Caterina Pellecchia, Silvia Borghini, Maria Libera Trotta, Anna Rubartelli, Andrew Henrey, Thomas Loughin, Roberta Berard, Natalie Shiff, Roman Jurencak, Susanne Benseler, Lori Tucker, on behalf of ReACCh-Out Investigators, Charalampia Papadopoulou, Ying Hong, Petra Krol, Yiannis Ioannou, Clarissa Pilkington, Hema Chaplin, Stephania Simou, Marietta Charakida, Lucy Wedderburn, Lynn R. Spiegel, Sara Ahola Kohut, Jennifer Stinson, Paula Forgeron, Miriam Kaufman, Nadia Luca, Khush Amaria, Mary Bell, J Swart, F. Boris, E. Castagnola, A. Groll, G. Giancane, G. Horneff, H. I. Huppertz, T. Wolfs, E. Alekseeva, V. Panaviene, F. Uettwiller, V. Stanevicha, L. M. Ailioaie, E. Tsitami, S. Kamphuis, G. Susic, F. Sztajnbok, B. Flato, A. Pistorio, Stephanie J. W. Shoop, Suzanne M. M. Verstappen, Janet E. McDonagh, Wendy Thomson, Kimme L. Hyrich, CAPS, Maarit Tarkiainen, Pirjo Tynjala, Pekka Lahdenne, Janne Martikainen, Acute-JIA Study Group, Meredyth Wilkinson, Christopher Piper, Georg Otto, Claire T. Deakin, Stefanie Dowle, Stefania Simou, Daniel Kelberman, Claudia Mauri, Elizabeth Jury, David Isenberg, Lucy R. Wedderburn, Kiran Nistala, I. Foeldvari, D. J. Lovell, G. Simonini, M. Bereswill, J. Kalabic, Kiem Oen, Brian M. Feldman, Brenden Dufault, Jennifer Lee, Karen Watanabe Duffy, Ciaran Duffy, ReACCh-Out Investigators, N. Tzaribachev, G. Vega-Cornejo, I. Louw, A. Berman, I. Calvo, R. Cuttica, F. Avila-Zapata, R. Cimaz, E. Solau-Gervais, R. Joos, G. Espada, X. Li, M. Nys, R. Wong, S. Banerjee, For Pediatric Rheumatology International Trials Organization (PRINTO)/Pediatric Rheumatology Collaborative Study Group (PRCSG), Rebecca Nicolai, Margherita Verardo, Adele D’Amico, Luisa Bracci-Laudiero, Gian Marco Moneta, Gillian Rice, Anne-Laure Mathieu, Sulliman O. Omarjee, Tracy A. Briggs, James O’Sullivan, Simon Williams, Rolando Cimaz, Eve Smith, Michael W. Beresford, Yanick J. Crow, GENIAL Investigators, UK JSLE Study Group, Madeleine Rooney, Nick Bishop, joyce davidson, Clarissa pilkington, Michael Beresford, Jacqui Clinch, Rangaraj Satyapal, Helen Foster, Janet Gardner Medwin, Janet McDonagh, Sue Wyatt, On Behalf of the British Society for Paediatric and Adolescent Rheumatology, Valentina Litta Modignani, Francesco Baldo, Stefano Lanni, Alessandro Consolaro, Giovanni Filocamo, Helen J. Lachmann, on behalf of Eurofever Registry, Gianmarco Moneta, Camilla Celani, Bilade Cherqaoui, Linda Rossi-Semerano, Perrine Dusser, Véronique Hentgen, Claire Grimwood, Linda Rossi, Isabelle Kone Paut, Veronique Hentgen, Denise Lasigliè, Denise Ferrera, Giulia Amico, Marco Di Duca, Laura Obici, Roberto Ravazzolo, Ryuta Nishikomori, Juan Arostegui, Andrea Petretto, Chiara Lavarello, Elvira Inglese, Federica Vanoni, Michaël Hofer, on behalf of EUROFEVER PROJECT, P. N. Hawkins, T. van der Poll, U. A. Walker, H. H. Tilson, Pascal N. Tyrrell, Raphaela Goldbach-Mansky, Norbert Blank, Hal M. Hoffman, Elisabeth Weissbarth-Riedel, Boris Huegle, Tilmann Kallinich, Ahmet Gul, Marlen Oswald, Fatma Dedeoglu, Aki Hanaya, Takako Miyamae, Manabu Kawamoto, Yumi Tani, Takuma Hara, Yasushi Kawaguchi, Satoru Nagata, Hisashi Yamanaka, Almira Ćosićkić, Fahrija Skokić, Belkisa Čolić, Sanimir Suljendić, Anna Kozlova, Irina Mersiyanova, Mariya Panina, Lily Hachtryan, Vasiliy Burlakov, Elena Raikina, Alexey Maschan, Anna Shcherbina, Banu Acar, Meryem Albayrak, Betul Sozeri, Sezgin Sahin, Amra Adrovic, Nese Inan, Serhan Sevgi, Caroline M. Andreasen, Anne Grethe Jurik, Mia B. Glerup, Christian Høst, Birgitte T. Mahler, Ellen-Margrethe Hauge, Cecilia Lazea, Laura Damian, Calin Lazar, Rodica Manasia, Chloe M. Stephenson, Vimal Prajapati, Paivi M. Miettunen, Dilek Yılmaz, Yavuz Tokgöz, Yasin Bulut, Harun Çakmak, Ferah Sönmez, Elif Comak, Gülşah Kaya Aksoy, Mustafa Koyun, Sema Akman, Yunus Arıkan, Ender Terzioğlu, Osman Nidai Özdeş, İbrahim Keser, Hüseyin Koçak, Ayşen Bingöl, Aygen Yılmaz, Reha Artan, X. Xu, Fatemeh F. Mehregan, Vahid Ziaee, Mohammad H. Moradinejad, Francesco La Torre, Clotilde Alizzi, Pio D’Adamo, G. Junge, J. Gregson, Hasmik Sargsyan, Hulya Zengin, Berna E. Fidanci, Cagla Kaymakamgil, Dilek Konukbay, Dilek Yildiz, Faysal Gok, Iris Stoler, Judith Freytag, Banu Orak, Christine Seib, Lars Esmann, Eva Seipelt, Faekah Gohar, Dirk Foell, Ismail Dursun, Sebahat Tulpar, Sibel Yel, Demet Kartal, Murat Borlu, Funda Bastug, Hakan Poyrazoglu, Zubeyde Gunduz, Kader Kose, Mehmet E. Yuksel, Abdullah Calıskan, Ahmet B. Cekgeloglu, Ruhan Dusunsel, Katerina Bouchalova, Jana Franova, Marcel Schuller, Marie Macku, Katerina Theodoropoulou, Raffaella Carlomagno, Annette von Scheven-Gête, Claudia Poloni, Laura O. Damian, Dan Cosma, Amanda Radulescu, Dan Vasilescu, Liliana Rogojan, Simona Rednic, Mihaela Lupse, Lien De Somer, Pierre Moens, Rocio Galindo Zavala, Laura Martín Pedraz, Esmeralda Núñez Cuadros, Gisela Díaz-Cordovés Rego, Antonio L. Urda Cardona, Ilaria Dal Forno, Sara Pieropan, Ombretta Viapiana, Davide Gatti, Gloria Dallagiacoma, Paola Caramaschi, Domenico Biasi, Daniel Windschall, Ralf Trauzeddel, Hartwig Lehmann, Rainer Berendes, Maria Haller, Manuela Krumrey-Langkammerer, Antje Nimtz-Talaska, Philipp Schoof, Ralf Felix Trauzeddel, Christine Nirschl, Estefania Quesada-Masachs, Carla Aguilar Blancafort, Sara Marsal Barril, Francisca Aguiar, Rita Fonseca, Duarte Alves, Ana Vieira, Alberto Vieira, Jorge A. Dias, Iva Brito, Gordana Susic, Vera Milic, Goran Radunovic, Ivan Boricic, Pauline Marteau, Catherine Adamsbaum, Michel De Bandt, Irène Lemelle, Chantal Deslandre, Tu Anh Tran, Anne Lohse, Elisabeth Solau-Gervais, Pascal Pillet, Julien Wipff, Cécile Gaujoux-Viala, Sylvain Breton, Valérie Devauchelle-Pensec, Sandra Gran, Olesja Fehler, Stefanie Zenker, Michael Schäfers, Thomas Vogl, Severine Guillaume Czitrom, EH Pieter Van Dijkhuizen, Silvia Magni Manzoni, Francesca Magnaguagno, Laura Tanturri de Horatio, Nienke M. Ter Haar, Annemieke S. Littooij, Vitor A. Teixeira, Raquel Campanilho-Marques, Ana F. Mourão, Filipa O. Ramos, Manuela Costa, Wafa A. Madan, Orla G. Killeen, Adriana Rodriguez Vidal, Diana Sueiro Delgado, Maria Isabel Gonzalez Fernandez, Berta Lopez Montesinos, Aleksey Kozhevnikov, Nina Pozdeeva, Mikhail Konev, Evgeniy Melchenko, Vladimir Kenis, Gennadiy Novik, Aysenur Pac Kısaarslan, Butsabong Lerkvaleekul, Suphaneewan Jaovisidha, Witaya Sungkarat, Niyata Chitrapazt, Praman Fuangfa, Thumanoon Ruangchaijatuporn, Soamarat Vilaiyuk, Dan Ø. Pradsgaard, Arne Hørlyck, Anne H. Spannow, Carsten W. Heuck, Talia Diaz, Fernando Garcia, Lorenia De La Cruz, Nadina Rubio, Joanna Świdrowska-Jaros, Elzbieta Smolewska, Mirta Lamot, Lovro Lamot, Mandica Vidovic, Edi Paleka Bosak, Ivana Rados, Miroslav Harjacek, Nikolay Tzaribachev, Polymnia Louka, Romiesa Hagoug, Chiara Trentin, Olga Kubassova, Mark Hinton, Mikael Boesen, Olena A. Oshlianska, Illya A. Chaikovsky, G. Mjasnikov, A. Kazmirchyk, Umberto Garagiola, Irene Borzani, Paolo Cressoni, Fabrizia Corona, Eszter Dzsida, Giampietro Farronato, Antonella Petaccia, Alenka Gagro, Agneza Marija Pasini, Goran Roic, Ozren Vrdoljak, Lucija Lujic, Matija Zutelija-Fattorini, Monika M. Esser, Deepthi R. Abraham, Craig Kinnear, Glenda Durrheim, Mike Urban, Eileen Hoal, Victoria B. Nikolayenko, Kubilay Şahin, Yasar Karaaslan, Adele Civino, Giovanni Alighieri, Sergio Davì, Roberto Rondelli, Andrea Magnolato, Francesca Ricci, Alma Olivieri, Valeria Gerloni, Bianca Lattanzi, Francesca Soscia, Alessandro De Fanti, Stefania Citiso, Lorenzo Quartulli, Maria Cristina Maggio, Manuela Marsili, Maria Antonietta Pelagatti, Valentino Conter, Franca Fagioli, Andrea Pession, Marco Garrone, Mariangela Rinaldi, Jaime De Inocencio, Stella Garay, Daniel J. Lovell, Berit Flato, EPOCA Study Group, Angela Aquilani, Simona Cascioli, Ivan Caiello, Denise Pires-Marafón, Rita Carsetti, Emily Robinson, Salvatore Albani, Wilco de Jager, Sytze de Roock, Trang Duong, Justine Ellis, Kimme Hyrich, Laetitia Jervis, Daniel Lovell, Lucy Marshall, Elizabeth D. Mellins, Kirsten Minden, Jane Munro, Peter A. Nigrovic, Jason Palman, Sunil Sampath, Laura E. Schanberg, Susan D. Thompson, Richard Vesely, Chris Wallace, Chris Williams, Qiong Wu, Nico Wulffraat, Rae S. M. Yeung, M. B. Seyger, D. Arikan, J. K. Anderson, A. Lazar, D. A. Williams, C. Wang, R. Tarzynski-Potempa, J. S. Hymans, Gabriele Simonini, Erika Scoccimarro, Irene Pontikaki, Teresa Giani, Alessandro Ventura, Pier Luigi Meroni, Gaetana Minnone, Marzia Soligo, Luigi Manni, Luisa Bracci Laudiero, Noortje Groot, I. Grein, N. M. Wulffraat, R. Schepp, G. Berbers, C. C. Barbosa Sandoval de Souza, V. Paes Leme Ferriani, G. Pileggi, S. de Roock, Ingrid H. R. Grein, Silvia Scala, Elisa Patrone, Casper Schoemaker, on behalf of Dutch JIA patient organization, Wendy Costello, on behalf of ENCA, Suzanne Parsons, Jean-David Cohen, Damien Bentayou, Marc-Antoine Bernard Brunel, Sonia Trope, Jens Klotsche, Miriam Listing, Martina Niewerth, Gerd Horneff, Angelika Thon, Hans-Iko Huppertz, Kirsten Mönkemöller, Ivan Foeldvari, ICON study group, Achille Marino, Stefano Stagi, Niccolò Carli, Federico Bertini, Adriana S. Díaz-Maldonado, Sally Pino, Pilar Guarnizo, Alfonso Ragnar Torres-Jimenez, Berenice Sanchez-Jara, Eunice Solis-Vallejo, Adriana Ivonne Cespedes-Cruz, Maritza Zeferino-Cruz, Julia Veronica Ramirez-Miramontes, Ankur Kumar, Anju Gupta, Deepti Suri, Amit Rawat, Nandita Kakkar, Surjit Singh, Özge A. Gücenmez, Erbil Ünsal, Bo Magnusson, Karina Mördrup, Anna Vermé, Christina Peterson, Board of the Swedish Pediatric Rheumatology Registry, Caroline Freychet, Jean Louis Stephan, Cathryn E. Harkness, Leanne Foster, Emma Henry, Pauline Taggart, Coskun F. Ozkececi, Esra Kurt, Gokalp Basbozkurt, Daiva Gorczyca, Jacek Postępski, Aleksandra Czajkowska, Bogumiła Szponar, Mariola Paściak, Anna Gruenpeter, Iwona Lachór-Motyka, Daria Augustyniak, Edyta Olesińska, Emediong S. Asuka, Tatyana Golovko, Samuel U. Aliejim, Emilio Inarejos Clemente, Estibaliz Iglesias Jimenez, Joan Calzada Hernandez, Sergi Borlan Fernandez, Clara Gimenez Roca, David Moreno Romo, Natalia Rodriguez Nieva, Juan Manuel Mosquera Angarita, Jordi Anton Lopez, Esmeralda Nuñez-Cuadros, Gisela Diaz-Cordovés, Rocío Galindo-Zavala, Antonio Urda-Cardona, Antonio Fernández-Nebro, Daniel Álvarez de la Sierra, Marina Garcia Prat, Mónica Martínez Gallo, Ricardo Pujol Borrell, Ana M. Marín Sánchez, Etienne Merlin, Sylvie Fraitag, Jean-Louis Stephan, Federico Annoni, Giancarla Di Landro, Sofia Torreggiani, Marta Torcoletti, Georgina Tiller, Jo Buckle, Angela Cox, Peter Gowdie, Roger C. Allen, Jonathan D. Akikusa, Hayde G. Hernández-Huirache, Edel R. Rodea-Montero, William Fahy, Christelle Sordet, Karin B. Berggren, Johanna T. Kembe, Joyce Bos, Wineke Armbrust, Marco van Brussel, Jeanette Cappon, Pieter Dijkstra, Jan Geertzen, Elizabeth Legger, Marion van Rossum, Pieter Sauer, Otto Lelieveld, Levent Buluc, Gur Akansel, Bahar Muezzinoglu, Ljubov Rychkova, Tatyana Knyazeva, Anna Pogodina, Tatyana Belova, Tamara Mandzyak, Ekaterina Kulesh, Alessandro Cafarotti, Cosimo Giannini, Roberta Salvatore, Giuseppe Lapergola, Caterina Di Battista, Maria Loredana Marcovecchio, Raffaella Basilico, Piernicola Pelliccia, Francesco Chiarelli, Luciana Breda, Beverley Almeida, Sarah Tansley, Harsha Gunawardena, Neil McHugh, Juvenile Dermatomyositis Research Group (JDRG), Jessie Aouizerate, Marie De Antonio, Christine Barnerias, Guillaume Bassez, Isabelle Desguerre, Romain Gherardi, Jean-Luc Charuel, François-Jérôme Authier, Cyril Gitiaux, C. H. Spencer, Rabheh Abdul Aziz, Chack-Yung Yu, Brent Adler, Sharon Bout-Tabaku, Katherine Lintner, Melissa Moore-Clingenpeel, Liza McCann, Nicola Ambrose, Mario Cortina-Borja, Juvenile Dermatomyositis Cohort and Biomarker Study (JCDBS), Prasad T. Oommen, Fabian Speth, Johannes-Peter Haas, Working Group “Juvenile Dermatomyositis” of the German Society for Paediatric and Adolescent Rheumatology (GKJR), Claudio Lavarello, Gabriella Giancane, Angela Pistorio, Lisa Rider, Rohit Aggarwal, Sheila K. Oliveira, Ruben Cuttica, Michel Fischbach, Gary Sterba, Karine Brochard, Frank Dressler, Patrizia Barone, Ruben Burgos-Vargas, Elizabeth Candell Chalom, Marine Desjonqueres, Graciela Espada, Anders Fasth, Stella Maris Garay, Rose-Marie Herbigneaux, Claire Hoyoux, Chantal Job Deslandre, Frederick W. Miller, Jiri Vencovsky, Erdal Sag, Gulsev Kale, Haluk Topaloglu, Beril Talim, Francesco Zulian, Tadej Avcin, Roberto Marini, Anne Pagnier, Michel Rodiere, Christine Soler, Rebecca Ten Cate, Yosef Uziel, Jelena Vojinovic, Ana V. Villarreal, Nydia Acevedo, Yuridiana Ramirez, Enrique Faugier, Rocio Maldonado, Bita Arabshahi, John H. Lee, Ian Leibowitz, Lawrence O. Okong’o, Jo Wilmshurst, Monika Esser, Christiaan Scott, Ezgi Deniz Batu, Nagehan Emiroglu, Hafize Emine Sonmez, Gokcen Dilsa Tugcu, Zehra Serap Arici, Ebru Yalcin, Deniz Dogru, Ugur Ozcelik, Mithat Haliloglu, Nural Kiper, Masato Yashiro, Mutsuko Yamada, Toshihiko Yabuuchi, Tomonobu Kikkawa, Nobuyuki Nosaka, Yosuke Fujii, Yukie Saito, Hirokazu Tsukahara, Sulaiman M. Al-Mayouf, Nora AlMutiari, Mohammed Muzaffer, Rawiah shehata, Adel Al-Wahadneh, Reem Abdwani, Safia Al-Abrawi, Mohammed Abu-shukair, Zeyad El-Habahbeh, Abdullah Alsonbul, Aleksandra Szabat, Monika Chęć, Violetta Opoka-Winiarska, Biman Saikia, Ranjana W. Minz, Christine Arango, Clara Malagon, Maria D. P. Gomez, Angela C. Mosquera, Ricardo Yepez, Tatiana Gonzalez, Camilo Vargas, GRIP study group, Marta Balzarin, Biagio Castaldi, Elena Reffo, Francesca Sperotto, Giorgia Martini, Alessandra Meneghel, Ornella Milanesi, Ozgur Kasapçopur, Maria Teresa Terreri, Ekaterina Alexeeva, Maria Katsicas, Mikhail Kostik, Thomas Lehman, W.-Alberto Sifuentes-Giraldo, Vanessa Smith, Flavio Sztajnbok, Tadey Avcin, Maria Jose Santos, Dana Nemcova, Cristina Battagliotti, Liora Harel, Mahesh Janarthanan, Kathryn Torok, Nicola Helmus, Eileen Baildem, Michael Blakley, Kim Fligelstone, Antonia Kienast, Clare Pain, Amanda Saracino, Gabriele Simoni, Lisa Weibel, Maria K. Osminina, Nathalia A. Geppe, Olga V. Niconorova, Olesya V. Karashtina, Oksana V. Abbyasova, Olga V. Shpitonkova, Sinem Durmus, Hafize Uzun, Angela Mauro, Eleonora Fanti, Fabio Voller, Franca Rusconi, Fernando Garcia-Rodriguez, Ana V. Villarreal-Treviño, Angel J. Flores-Pineda, Paola B. Lara-Herrea, Diego R. Salinas-Encinas, Talia Diaz-Prieto, Maria R. Maldonado-Velazquez, Sarbelio Moreno-Espinosa, Enrique Faugier-Fuentes, Mirella Crapanzano, Ilaria Parissenti, Man S. Parihar, Pandiarajan Vignesh, ManojKumar Rohit, Kavitha Gopalan, Savita V. Attri, Alan Salama, David Jayne, Mark Little, Yulia Kostina, Galina Lyskina, Olga Shpitonkova, Alena Torbyak, Olga Shirinsky, Maria Francesca Gicchino, Maria Cristina Smaldone, Mario Diplomatico, Alma Nunzia Olivieri, C H. Spencer, Richard McClead, Hiren Patel, Chung-Yung Yu, Dita Cebecauerová, Tomáš Dallos, Edita Kabíčková, Martin Kynčl, Daniela Chroustová, Jozef Hoza, Dana Němcová, Vladimír Tesař, Pavla Doležalová, Tuncay Hazirolan, Fatih Ozaltin, Fabiola Almeida, Isabela H. Faria de Paula, Maíra M. Sampaio, Fernando N. Arita, Andressa G. Alves, Maria Carolina Santos, Eunice M. Okuda, Silvana B. Sacchetti, Fernanda Falcini, Marini Francesca, Gemma Lepri, Marco Matucci-Cerinic, Maria Luisa Brandi, Hakan Kisaoglu, Sema Misir, Selim Demir, Yuksel Aliyazicioglu, Mukaddes Kalyoncu, Carlos Eduardo Ramalho, Fabiola D. Almeida, Joan Calzada-Hernández, Rosa Bou, Estíbaliz Iglesias, Judith Sánchez-Manubens, Fredy Hermógenes Prada Martínez, Clara Giménez Roca, Sergi Borlan Fernández, Marek Bohm, Kamran Mahmood, Valentina Leone, Mark Wood, Ken-Ichi Yamaguchi, Satoshi Fujikawa, Working Group of Behçet’s Disease, Pediatric Rheumatology Association of Japan (PRAJ), Kyu Yeun Kim, Do Young Kim, Dong Soo Kim, Maka Ioseliani, Ivane Chkhaidze, Maia Lekishvili, Nana Tskhakaia, Shorena Tvalabeishvili, Aleksandre Kajrishvili, Maiko Takakura, Masaki Shimizu, Natsumi Inoue, Mao Mizuta, Akihiro Yachie, Giovanni Corsello, Maryam Piram, Carla Maldini, Sandra Biscardi, Nathalie Desuremain, Catherine Orzechowski, Emilie Georget, Delphine Regnard, Isabelle Kone-Paut, Alfred Mahr, Mihaela Sparchez, Zeno Sparchez, Nydia Acevedo Silva, Ana V. Villarreal Treviño, Yuridiana Ramirez Loyola, Talia Diaz Prieto, Enrique Faugier Fuentes, Maria D. R. Maldonado Velazquez, Pilar Perez, Sagar Bhattad, Ranjana Minz, Jitendra Shandilya, Pediatric Allergy and Immunology Unit, PGIMER, Chandigarh, Ana Villarreal, Yuridiana Ramírez, Zeynep Birsin Özçakar, Suat Fitoz, Fatos Yalcinkaya, Annacarin Horne, Francesca Minoia, Francesca Bovis, Sergio Davi, Priyankar Pal, Kimo Stein, Sandra Enciso, Michael Jeng, Despoina Maritsi, Randy C. Cron, Anne Thorwarth, Sae Lim von Stuckrad, Angela Rösen-Wolff, Hella Luksch, Patrick Hundsdoerfer, Peter Krawitz, Nuray Aktay Ayaz, Doğan Simsek, Şebnem Sara Kılıc, Emine Sonmez, Aysenur Pac Kisaarslan, Ozge Altug Gucenmez, Z. Serap Arıcı, Fatih Kelesoglu, Zelal Ekinci Ekinci, Maria Miranda-Garcia, Carolin Pretzer, Michael Frosch, F. Gohar, Angela McArdle, Niamh Callan, Belinda Hernandez, Miha Lavric, Oliver FitzGerald, Stephen R. Pennington, Joachim Peitz, Joern Kekow, Ariane Klein, Anna C. Schulz, Frank Weller-Heinemann, Anton Hospach, J-Peter Haas, BIKER collaborative group, Karen Put, Jessica Vandenhaute, Anneleen Avau, Annemarie van Nieuwenhuijze, Ellen Brisse, Tim Dierckx, Omer Rutgeerts, Josselyn E. Garcia-Perez, Jaan Toelen, Mark Waer, Georges Leclercq, An Goris, Johan Van Weyenbergh, Adrian Liston, Patrick Matthys, Carine H. Wouters, Yasuo Nakagishi, Michael J. Ombrello, Victoria Arthur, Anne Hinks, Patricia Woo, International Childhood Arthritis Genetics (INCHARGE) Consortium, Barbara Stanimirovic, Biljana Djurdjevic-Banjac, Olivera Ljuboja, Boris Hugle, MArgarita Onoufriou, Olga Vougiouka, Kenza Bouayed, Sanae El Hani, Imane Hafid, Nabiha Mikou, Nunu Shelia, Mari Laan, Jaanika Ilisson, and Chris Pruunsild
Subjects: Pediatrics, RJ1-570, Diseases of the musculoskeletal system, RC925-935
Published: 2017
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30. Efavirenz-based simplification after successful early lopinavir-boosted-ritonavir-based therapy in HIV-infected children in Burkina Faso and Côte d’Ivoire: the MONOD ANRS 12206 non-inferiority randomised trial

Author: Désiré Lucien Dahourou, Madeleine Amorissani-Folquet, Karen Malateste, Clarisse Amani-Bosse, Malik Coulibaly, Carole Seguin-Devaux, Thomas Toni, Rasmata Ouédraogo, Stéphane Blanche, Caroline Yonaba, François Eboua, Philippe Lepage, Divine Avit, Sylvie Ouédraogo, Philippe Van de Perre, Sylvie N’Gbeche, Angèle Kalmogho, Roger Salamon, Nicolas Meda, Marguerite Timité-Konan, Valériane Leroy, and on behalf of the MONOD Study Group
Subjects: Africa, HIV, Early antiretroviral treatment, Infants, Protease inhibitors, Lopinavir, Medicine
Abstract: Abstract Background The 2016 World Health Organization guidelines recommend all children
Published: 2017
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31. Clinical, virological and immunological features of HIV-positive children internationally adopted in France from 2005-2015.

Author: Violaine Corbin, Pierre Frange, Florence Veber, Stéphane Blanche, Camille Runel-Belliard, Muriel Lalande, Virginie Gandemer, Marie Moukagni-Pelzer, Catherine Dollfus, Dilek Coban, Justine Prouteau, Christine Jacomet, Olivier Lesens, and HIV adopted children study group
Subjects: Medicine, Science
Abstract: OBJECTIVE(S):To describe the clinical, virological and immune characteristics of internationally adopted children on arrival in France and after 6-months follow-up. DESIGN:Multicenter retrospective study. METHODS:30 centers from 24 cities were asked to include, after informed consent, HIV+ children living in France and internationally adopted between 1st Jan 2005 and 1st Jan 2015. Sociodemographic, medical and biological variables collected during the first medical evaluation in France and 6 months later were analyzed. RESULTS:41 HIV+ adoptees were included (female: 56%; median age: 3.91 years) in 14 centers. Adoptees tend to represent an increasing part of newly diagnosed HIV positive children over the years. The majority came from East-Asia. At arrival, one child was diagnosed with lymphobronchial tuberculosis and three with latent chronic hepatitis B, cleared HBV infection and chronic active hepatitis C, respectively. The mean CD4% was 32.8 ± 9% (range: 13-49%). The 34 children (83%) have been initiated on treatment from their countries of origin. Of these, 25 (74%) had an undetectable viral load (VL) on arrival. Resistance to ART was detected in five. At 6 months, 36 adoptees received ART, and the VL was undetectable in 29 children (71%), with one acquired resistance to NRTI & NNRTI. CONCLUSIONS:An increasing number of HIV-infected children have been internationally adopted in France since 2005. Most of the children have been initiated on treatment from their countries of origin, had good immunity, with few opportunistic infections, and infrequently detectable VL. Low level of mutation conferring resistance was detected.
Published: 2018
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32. Gag-Specific CD4 and CD8 T-Cell Proliferation in Adolescents and Young Adults with Perinatally Acquired HIV-1 Infection Is Associated with Ethnicity - The ANRS-EP38-IMMIP Study.

Author: Jérôme Le Chenadec, Daniel Scott-Algara, Stéphane Blanche, Céline Didier, Thomas Montange, Jean-Paul Viard, Catherine Dollfus, Véronique Avettand-Fenoel, Christine Rouzioux, Josiane Warszawski, and Florence Buseyne
Subjects: Medicine, Science
Abstract: The ANRS-EP38-IMMIP study aimed to provide a detailed assessment of the immune status of perinatally infected youths living in France. We studied Gag-specific CD4 and CD8 T-cell proliferation and the association between the proliferation of these cells, demographic factors and HIV disease history. We included 93 youths aged between 15 and 24 years who had been perinatally infected with HIV. Sixty-nine had undergone valid CFSE-based T-cell proliferation assays. Gag-specific proliferation of CD4 and CD8 T cells was detected in 12 (16%) and 30 (38%) patients, respectively. The Gag-specific proliferation of CD4 and CD8 T cells was more frequently observed in black patients than in patients from other ethnic groups (CD4: 32% vs. 4%, P = 0.001; CD8: 55% vs. 26%, P = 0.02). Among aviremic patients, the duration of viral suppression was shorter in CD8 responders than in CD8 nonresponders (medians: 54 vs. 20 months, P = 0.04). Among viremic patients, CD8 responders had significantly lower plasma HIV RNA levels than CD8 nonresponders (2.7 vs. 3.7 log10 HIV-RNA copies/ml, P = 0.02). In multivariate analyses including sex and HIV-1 subtype as covariables, Gag-specific CD4 T-cell proliferation was associated only with ethnicity, whereas Gag-specific CD8 T-cell proliferation was associated with both ethnicity and the duration of viral suppression. Both CD4 and CD8 responders reached their nadir CD4 T-cell percentages at younger ages than their nonresponder counterparts (6 vs. 8 years, P = 0.04 for both CD4 and CD8 T-cell proliferation). However, these associations were not significant in multivariate analysis. In conclusion, after at least 15 years of HIV infection, Gag-specific T-cell proliferation was found to be more frequent in black youths than in patients of other ethnic groups, despite all the patients being born in the same country, with similar access to care.
Published: 2015
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33. Circulating Endothelial Cells As a Reliable Marker Of Endothelial Damage In Children Undergoing Hematopoietic Stem Cell Transplantation

Author: Fabien Touzot, Despina Moshous, Guilhem Cros, Pierre Frange, Sebastien Heritier, Benedicte Neven, Marina Cavazzana-Calvo, Alain Fischer, Stephane Blanche, and Dominique Helley
Subjects: medicine.medical_specialty, Hepatic veno-occlusive disease, Thrombotic microangiopathy, business.industry, medicine.medical_treatment, Immunology, Cell Biology, Hematology, Hematopoietic stem cell transplantation, Defibrotide, medicine.disease, Biochemistry, Gastroenterology, Surgery, Transplantation, Graft-versus-host disease, Internal medicine, Medicine, Endothelial dysfunction, business, Complication, medicine.drug
Abstract: The endothelial dysfunction associated with Hematopoietic Stem Cell Transplantation (HSCT) is responsible for pleomorphic complications that arise during the post-tranplant period: sinusoidal obstruction syndrome (SOS), pulmonary arterial hypertension (PAH), capillary leak syndrome (CLS) and transplantation-associated thrombotic microangiopathy (TA-TMA). Numerous factors may induce these endothelial damages. Circulating endothelial cells (CEC) emerge as a potent marker of endothelial lesion in a variety of disorders but their monitoring in HSCT has been little investigated so far. Methods Thirty-six pediatric patients treated in the department of Immuno-Hematology at the Necker-Enfants-Malades Hospital who received allogeneic HSCT after full intensity conditioning regimen for non-malignant disorders including inherited immunodeficiencies, metabolic diseases or hemoglobin disorders were enrolled in the study between April 2011 and December 2012. CEC were monitored at 5 time points: before conditioning (day -15), at time of transplant (day 0) and during the post-transplantation period (at day +15, day +30 and eventually day +60 if the patient was still hospitalized). Blood samples were not collected in period of sepsis to avoid any confounding factor7. Results Among the thirty-six patient enrolled, four patients were excluded because of incomplete follow-up. Thirty-two patients completed the study, and their data were analyzed. Vascular complications occurred in 10 patients (31% patients) and are detailed in Figure 1C. Of note, all patients who developed SOS were under defibrotide prophylaxis. Two patients developed CLS in association with grade IV GVH. TA-TMA occurred in two patients and resolved in both after cyclosporine withdrawal. Patients with vascular events (VE) had significant higher CEC counts as compared to patients without endothelial complication at day +15 (p = 0.0122) and day +30 (p=0.0046). The increase of CEC counts was significantly higher in patients with VE during the first 15 days (p=0.0175) and during the first month post transplantation (p=0.0005) as compared to patient without VE. Strikingly, detection of high CEC counts in patients with major vascular events (ie. PAH, CLS and TA-TAM) was preceding the onset of clinical manifestations by a median of 18.5 days (range, 8-42). We didn’t find any correlation on univariate analysis between CEC counts and the following variables: the underlying disease, the intensity of conditioning regimen, the existence of CMV reactivation, graft versus host disease or death. Conclusion We observed that the occurrence of vascular complications is associated with an early rise (in the first 15 days) of CEC count. Interestingly, the increase in the number of CEC is preceding the onset of major endothelial complications. Thus, monitoring of CEC in routine could be a useful prognostic tool that could allow early therapeutic intervention. Disclosures: No relevant conflicts of interest to declare.

34. Association between prenatal exposure to antiretroviral therapy and birth defects: an analysis of the French perinatal cohort study (ANRS CO1/CO11).

Author: Jeanne Sibiude, Laurent Mandelbrot, Stéphane Blanche, Jérôme Le Chenadec, Naima Boullag-Bonnet, Albert Faye, Catherine Dollfus, Roland Tubiana, Damien Bonnet, Nathalie Lelong, Babak Khoshnood, and Josiane Warszawski
Subjects: Medicine
Abstract: BACKGROUND: Antiretroviral therapy (ART) has major benefits during pregnancy, both for maternal health and to prevent mother-to-child transmission of HIV. Safety issues, including teratogenic risk, need to be evaluated. We estimated the prevalence of birth defects in children born to HIV-infected women receiving ART during pregnancy, and assessed the independent association of birth defects with each antiretroviral (ARV) drug used. METHODS AND FINDINGS: The French Perinatal Cohort prospectively enrolls HIV-infected women delivering in 90 centers throughout France. Children are followed by pediatricians until 2 y of age according to national guidelines. We included 13,124 live births between 1994 and 2010, among which, 42% (n = 5,388) were exposed to ART in the first trimester of pregnancy. Birth defects were studied using both European Surveillance of Congenital Anomalies (EUROCAT) and Metropolitan Atlanta Congenital Defects Program (MACDP) classifications; associations with ART were evaluated using univariate and multivariate logistic regressions. Correction for multiple comparisons was not performed because the analyses were based on hypotheses emanating from previous findings in the literature and the robustness of the findings of the current study. The prevalence of birth defects was 4.4% (95% CI 4.0%-4.7%), according to the EUROCAT classification. In multivariate analysis adjusting for other ARV drugs, maternal age, geographical origin, intravenous drug use, and type of maternity center, a significant association was found between exposure to zidovudine in the first trimester and congenital heart defects: 2.3% (74/3,267), adjusted odds ratio (AOR) = 2.2 (95% CI 1.3-3.7), p = 0.003, absolute risk difference attributed to zidovudine +1.2% (95% CI +0.5; +1.9%). Didanosine and indinavir were associated with head and neck defects, respectively: 0.5%, AOR = 3.4 (95% CI 1.1-10.4), p = 0.04; 0.9%, AOR = 3.8 (95% CI 1.1-13.8), p = 0.04. We found a significant association between efavirenz and neurological defects (n = 4) using the MACDP classification: AOR = 3.0 (95% CI 1.1-8.5), p = 0.04, absolute risk +0.7% (95% CI +0.07%; +1.3%). But the association was not significant using the less inclusive EUROCAT classification: AOR = 2.1 (95% CI 0.7-5.9), p = 0.16. No association was found between birth defects and lopinavir or ritonavir with a power >85% for an odds ratio of 1.5, nor for nevirapine, tenofovir, stavudine, or abacavir with a power >70%. Limitations of the present study were the absence of data on termination of pregnancy, stillbirths, tobacco and alcohol intake, and concomitant medication. CONCLUSIONS: We found a specific association between in utero exposure to zidovudine and heart defects; the mechanisms need to be elucidated. The association between efavirenz and neurological defects must be interpreted with caution. For the other drugs not associated with birth defects, the results were reassuring. Finally, whatever the impact that some ARV drugs may have on birth defects, it is surpassed by the major role of ART in the successful prevention of mother-to-child transmission of HIV. Please see later in the article for the Editors' Summary.
Published: 2014
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35. Missed opportunities for early access to care of HIV-infected infants in Burkina Faso.

Author: Malik Coulibaly, Nicolas Meda, Caroline Yonaba, Sylvie Ouedraogo, Malika Congo, Mamoudou Barry, Elisabeth Thio, Issa Siribié, Fla Koueta, Diarra Ye, Ludovic Kam, Stéphane Blanche, Phillipe Van De Perre, Valériane Leroy, and MONOD Study Group ANRS
Subjects: Medicine, Science
Abstract: OBJECTIVE: The World Health Organization (WHO) has recommended a universal antiretroviral therapy (ART) for all HIV-infected children before the age of two since 2010, but this implies an early identification of these infants. We described the Prevention of Mother-to-Child HIV Transmission (PMTCT) cascade, the staffing and the quality of infrastructures in pediatric HIV care facilities, in Ouagadougou, Burkina Faso. METHODS: We conducted a cross-sectional survey in 2011 in all health care facilities involved in PMTCT and pediatric HIV care in Ouagadougou. We assessed them according to their coverage in pediatric HIV care and WHO standards, through a desk review of medical registers and a semi-structured questionnaire administered to health-care workers (HCW). RESULTS: In 2011, there was no offer of care in primary health care facilities for HIV-infected children in Ouagadougou. Six district hospitals and two university hospitals provided pediatric HIV care. Among the 67 592 pregnant women attending antenatal clinics in 2011, 85.9% were tested for HIV. The prevalence of HIV was 1.8% (95% Confidence Interval: 1.7%-1.9%). Among the 1 064 HIV-infected pregnant women attending antenatal clinics, 41.4% received a mother-to-child HIV transmission prevention intervention. Among the HIV-exposed infants, 313 (29.4%) had an early infant HIV test, and 306 (97.8%) of these infants tested received their result within a four-month period. Among the 40 children initially tested HIV-infected, 33 (82.5%) were referred to a health care facility, 3 (9.0%) were false positive, and 27 (90.0%) were initiated on ART. Although health care facilities were adequately supplied with HIV drugs, they were hindered by operational challenges such as shortage of infrastructures, laboratory reagents, and trained HCW. CONCLUSIONS: The PMTCT cascade revealed bottle necks in PMTCT intervention and HIV early infant diagnosis. The staffing in HIV care and quality of health care infrastructures were also insufficient in 2011 in Ouagadougou.
Published: 2014
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36. Variant of X-Linked Chronic Granulomatous Disease Revealed by a Severe Burkholderia cepacia Invasive Infection in an Infant

Author: Saul Oswaldo Lugo Reyes, Nizar Mahlaoui, Carolina Prando, Lizbeth Blancas Galicia, Marjorie Hubeau, Stéphane Blanche, Capucine Picard, Jean-Laurent Casanova, and Jacinta Bustamante
Subjects: Immunologic diseases. Allergy, RC581-607
Abstract: Chronic granulomatous disease (CGD) is a primary immunodeficiency characterized by increased susceptibility to bacteria and fungi since early in life, caused by mutations in any of the five genes coding for protein subunits in NADPH oxidase. X-linked variant CGD can be missed during routine evaluation or present later in life due to hypomorphic mutations and a residual superoxide production. The case of a 10-month-old boy who died of pneumonia is reported. The isolation of Burkholderia cepacia from his lung, together with a marginally low nitroblue tetrazolium reduction assay (NBT), made us suspect and pursue the molecular diagnosis of CGD. A postmortem genetic analysis finally demonstrated CGD caused by a hypomorphic missense mutation with normal gp91phox expression. In a patient being investigated for unusually severe or recurrent infection, a high index of suspicion of immunodeficiency must be maintained.
Published: 2013
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