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1. Quality of life in adolescents with chronic kidney disease who initiate haemodialysis treatment

Author

Stéphanie Clavé, Michel Tsimaratos, Mohamed Boucekine, Bruno Ranchin, Rémi Salomon, Olivier Dunand, Arnaud Garnier, Annie Lahoche, Marc Fila, Gwenaelle Roussey, Francoise Broux, Jérome Harambat, Sylvie Cloarec, Soraya Menouer, Georges Deschenes, Isabelle Vrillon, Pascal Auquier, and Julie Berbis

Subjects
End-stage renal disease, Adolescents, Initiating haemodialysis, Quality of life, Coping strategies, Bayesian models, Diseases of the genitourinary system. Urology, RC870-923
Abstract

Abstract Background To describe the quality of life of adolescents initiating haemodialysis, to determine the factors associated with quality of life, and to assess coping strategies and their impact on quality of life. Methods All adolescents initiating haemodialysis between September 2013 and July 2015 in French paediatric haemodialysis centres were included. Quality of life data were collected using the “Vécu et Santé Perçue de l’Adolescent et l’Enfant” questionnaire, and coping data were collected using the Kidcope questionnaire. Adolescent’s quality of life was compared with age- and sex-matched French control. Results Thirty-two adolescents were included. Their mean age was 13.9 ± 2.0 years. The quality of life score was lowest in leisure activities and highest in relationships with medical staff. Compared with the French control, index, energy-vitality, relationships with friends, leisure activities and physical well-being scores were significantly lower in haemodialysis population. In multivariate analyses, active coping was positively associated with quality of life and especially with energy-vitality, relationships with parents and teachers, and school performance. In contrast, avoidant and negative coping were negatively associated with energy-vitality, psychological well-being and body image for avoidant coping, and body image and relationships with medical staff for negative coping. Conclusions The quality of life of haemodialysis adolescents, and mainly the dimensions of leisure activities, physical well-being, relationships with friends and energy-vitality, were significantly altered compared to that of the French population. The impact of coping strategies on quality of life seems to be important. Given the importance of quality of life and coping strategies in adolescents with chronic disease, health care professionals should integrate these aspects into care management.

Published
2019
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2. Bi‐allelic pathogenic variants in <scp> ITGA8 </scp> cause slowly progressive renal disease of unknown etiology

Author

Sara Gómez‐Conde, Olivier Dunand, Aurélie Hummel, Vincent Morinière, Marion Gauthier, Laurent Mesnard, and Laurence Heidet

Subjects
Genetics, Humans, Integrin alpha Chains, Genetics (clinical)
Abstract

Integrin Subunit Alpha 8 gene (ITGA8) encodes an integrin chain that is known to be critical in the early stage of the kidney development. Bi-allelic pathogenic variants in ITGA8 are associated with bilateral renal agenesis, as well as anomalies involving urogenital system. Here, we report two unrelated patients presenting with slowly progressing chronic kidney disease associated with bilateral renal hypodysplasia carrying homozygous loss of function variants in the ITGA8 gene. These results broaden the clinical and genotypic spectrum of ITGA8 defects, revealing the high and unexpected degree of phenotypic heterogeneity of this autosomal recessive disease. Our study emphasizes the usefulness of Next-Generation Sequencing in unraveling the genetic cause of chronic kidney disease of unknown etiology, and raises the question of genetic modifiers involved in the variation of the phenotypes associated with autosomal recessive ITGA8 pathogenic variants.

Published
2022

3. The genetic landscape and clinical spectrum of nephronophthisis and related ciliopathies

Author

Friederike Petzold, Katy Billot, Xiaoyi Chen, Charline Henry, Emilie Filhol, Yoann Martin, Marina Avramescu, Maxime Douillet, Vincent Morinière, Pauline Krug, Cécile Jeanpierre, Kalman Tory, Olivia Boyer, Anita Burgun, Aude Servais, Remi Salomon, Alexandre Benmerah, Laurence Heidet, Nicolas Garcelon, Corinne Antignac, Mohamad Zaidan, Sophie Saunier, Tania Attié-Bitach, Valerie Comier-Daire, Jean-Michel Rozet, Yaacov Frishberg, Brigitte Llanas, Michel Broyer, Nabil Mohsin, Marie-Alice Macher, Nicole Philip, Véronique Baudouin, Damian Brackman, Chantal Loirat, Marina Charbit, Maud Dehennault, Claude Guyot, Pierre Bataille, Mariet Elting, Georges Deschenes, Andrea Gropman, Geneviève Guest, Marie-France Gagnadoux, Philippe Nicoud, Pierre Cochat, Bruno Ranchin, Albert Bensman, Anne-Marie Guerrot, Bertrand Knebelmann, Ilmay Bilge, Danièle Bruno, Stéphane Burtey, Caroline Rousset Rouvière, Valérie Caudwell, Denis Morin, Hélène Dollfus, Anne Maisin, Christian Hamel, Eric Bieth, Sophie Gie, Judith Goodship, Gwenaelle Roussey, Hermine La Selve, Hubert Nivet, Lucie Bessenay, Mathilde Caillez, Jean Bernard Palcoux, Stéphane Benoît, Philippe Dubot, Marc Fila, Fabienne Giuliano, Daouya Iftene, Michele Kessler, Theresa Kwon, Anine Lahoche, Audrey Laurent, Anne-Laure Leclerc, David Milford, Thomas Neuhaus, Sylvie Odent, Philippe Eckart, Dominique Chauveau, Patrick Niaudet, Horacio Repetto, Sophie Taque, Alexandra Bruel, Alexandra Noel-Botte, Emma Allain Launay, Lisa Allard, Dany Anlicheau, Anne-Laure Adra, Arnaud Garnier, Arvind Nagra, Remy Baatard, Justine Bacchetta, Banu Sadikoglu, Christine Barnerias, Anne Barthelemy, Lina Basel, Nader Bassilios, Hedi Ben Maiz, Fatma Ben Moussa, Faïza Benmati, Romain Berthaud, Aurélia Bertholet, Dominique Blanchier, Jean Jacques Boffa, Karim Bouchireb, Ihab Bouhabel, Zakaria Boukerroucha, Guylhène Bourdat-Michel, Odile Boute, Karine Brochard, Roseline Caumes, Siham Chafai Elalaoui, Bernard Chamontin, Marie Caroline Chastang, Christine Pietrement, Christine Richer, Christophe Legendre, Karin Dahan, Fabienne Dalla-Vale, Damien Thibaudin, Maxime Dauvergne, Salandre Davourie, Martin Debeukelaer, Jean Daniel Delbet, Constantinos Deltas, Denis Graber, Nadège Devillars, Boucar Diouf, Martine Doco Fenzy, Jean-Luc André, Dominique Joly, Alan Fryer, Laetitia Albano, Elisabeth Cassuto, Aline Pincon, Ana Medeira, Annabelle Chaussenot, Anne Mensire-Marinier, Francois Bouissou, Stephane Decramer, Armand Bottani, Aurélie Hummel, Alexandre Karras, Avi Katz, Christine Azema, Bénédicte Janbon, Bernard Roussel, Claude Bonniol, Christiophe Mariat, Gérard Champion, Deborah Chantreuil, Nicolas Chassaing, Christiane Mousson, Christine Baudeau, Delphine Hafdar Cuntz, Cyril Mignot, Laurene Dehoux, Didier Lacombe, Thierry Hannedouche, Elodie Mérieau, Emmanuelle Charlin, Eric Gauthier, Florent Plasse, Stanislas Faguer, Fanny Lebas, Florence Demurger, Francesco Emma, François Cartault, Geneviève Dumont, Nathalie Godefroid, Vincent Guigonis, Sophie Hillaire, Jaap Groothoff, Jan Dudley, Noémie Jourde-Chiche, Khalil El Karoui, Saoussen Krid, Krier Coudert, Larbi Bencheick, Laurent Yver, Marie-Pierre Lavocat, Le Monies De Sagazan, Valerie Leroy, Lise Thibaudin, Liz Ingulli, Lorraine Gwanmesia, Lydie Burglen, Marie-Hélène Saïd-Menthon, Marta Carrera, Mathilde Nizon, Catherine Melander, Michel Foulard, Monique Blayo, Jacques Prinseau, Nadine Jay, Nathalie Brun, Nicolas Camille, François Nobili, Olivier Devuyst, Ouafa Ben Brahim, Paloma Parvex, Laurence Perrin Sabourin, Philippe Blanc, Philippe Vanhille, Pierre Galichon, Sophie Pierrepont, Vincent Planquois, Gwenaelle Poussard, Claire Pouteil Noble, Radia Allal, Raphaelle Bernard, Raynaud Mounet, Rémi Cahen, Renaud Touraine, Claire Rigothier, Amélie Ryckewaert, Mathieu Sacquepee, Salima El Chehadeh, Charlotte Samaille, Shuman Haq, Ari Simckes, Stéphanie Lanoiselée, Stephanie Tellier, Jean-François Subra, Sylvie Cloarec, Julie Tenenbam, Thomas Lamy, Valérie Drouin Garraud, Huguette Valette, Vanina Meyssonnier, Rosa Vargas-Poussou, Yves Snajer, Sandrine Durault, Emmanuelle Plaisier, Etienne Berard, Fadi Fakhouri, Ferielle Louillet, Paul Finielz, Michel Fischbach, Bernard Foliguet, Hélène Francois-Pradier, Florentine Garaix, Marion Gerard, Gianfranco Rizzoni, Brigitte Gilbert, Denis Glotz, Astrid Godron Dubrasquet, Jean-Pierre Grünfeld, Guillaume Bollee, Michelle Hall, Sverker Hansson, Damien Haye, Hélène Taffin, Friedhelm Hildebrandt, Maryvonne Hourmand, Hümya Kayserili, Ivan Tack, Marie Line Jacquemont, Jennifer Fabre-Teste, Cliff Kashtan, Kkoen Van Hoeck, Alexandre Klein, Yannick Knefati, Nine Knoers, Martin Konrad, Alain Lachaux, Isabelle Landru, Gilbert Landthaler, Philippe Lang, Patrick Le Pogamp, Tristan Legris, Catherine Didailler, Thierry Lobbedez, Loïc de Parscau, Lucile Pinson, Hervé Maheut, Marc Duval-Arnould, Marlène Rio, Marie-Claire Gubler, Pierre Merville, Guillaume Mestrallet, Maite Meunier, Karine Moreau, Jérôme Harambat, Graeme Morgan, Georges Mourad, Niksic Stuber, Odile Boespflug-Tanguy, Olivier Dunand, Olivier Niel, Nacera Ouali, Paolo Malvezzi, Pauline Abou Jaoude, Solenne Pelletier, Julie Peltier, M.B. Petersen, Philippe Michel, Philippe Rémy, Jean-Baptiste Philit, Valérie Pichault, Thierry Billette de Villemeur, Bernard Boudailliez, Bruno Leheup, Claire Dossier, Djamal-Dine Djeddi, Yves Berland, Bruno Hurault de Ligny, Susan Rigden, Christophe Robino, Annick Rossi, Sabine Sarnacki, Messaoud Saidani, Albane Brodin Sartorius, Elise Schäfer, Sztriha Laszlo, Marie-Christine Thouret, Angélique Thuillier-Lecouf, Howard Trachtman, Claire Trivin, Michel Tsimaratos, Rita Van Damme-Lombaerts, Marjolaine Willems, Michel Youssef, Ariane Zaloszyc, Alexis Zawodnik, and Marie-Julia Ziliotis

Subjects
Nephrology
Abstract

Nephronophthisis (NPH) is an autosomal-recessive ciliopathy representing one of the most frequent causes of kidney failure in childhood characterized by a broad clinical and genetic heterogeneity. Applied to one of the worldwide largest cohorts of patients with NPH, genetic analysis encompassing targeted and whole exome sequencing identified disease-causing variants in 600 patients from 496 families with a detection rate of 71%. Of 788 pathogenic variants, 40 known ciliopathy genes were identified. However, the majority of patients (53%) bore biallelic pathogenic variants in NPHP1. NPH-causing gene alterations affected all ciliary modules defined by structural and/or functional subdomains. Seventy six percent of these patients had progressed to kidney failure, of which 18% had an infantile form (under five years) and harbored variants affecting the Inversin compartment or intraflagellar transport complex A. Forty eight percent of patients showed a juvenile (5-15 years) and 34% a late-onset disease (over 15 years), the latter mostly carrying variants belonging to the Transition Zone module. Furthermore, while more than 85% of patients with an infantile form presented with extra-kidney manifestations, it only concerned half of juvenile and late onset cases. Eye involvement represented a predominant feature, followed by cerebellar hypoplasia and other brain abnormalities, liver and skeletal defects. The phenotypic variability was in a large part associated with mutation types, genes and corresponding ciliary modules with hypomorphic variants in ciliary genes playing a role in early steps of ciliogenesis associated with juvenile-to-late onset NPH forms. Thus, our data confirm a considerable proportion of late-onset NPH suggesting an underdiagnosis in adult chronic kidney disease.

Published
2023

4. Renal Prognosis in Children With Tubulointerstitial Nephritis and Uveitis Syndrome

Author

Corentin Tanne, François Nobili, Christine Pietrement, Emma Allain-Launay, Olivia Boyer, Claire Duflos, Annie Lahoche, Guylhène Bourdat-Michel, Sylvie Cloarec, Ariane Zaloszyc, Lise Allard, Agnès Chevalier, Djamal Djeddi, Tim Ulinski, Stéphane Decramer, Olivier Dunand, Julien Hogan, Justine Bacchetta, Marc Fila, Camille Faudeux, Stéphanie Clavé, Denis Morin, Isabelle Vrillon, Françoise Broux, Sophie Taque, Herrada, Anthony, Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Université de Montpellier (UM), Assistance Publique - Hôpitaux de Marseille (APHM), CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Imagine - Institut des maladies génétiques (IHU) (Imagine - U1163), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Cité (UPCité), AP-HP Hôpital universitaire Robert-Debré [Paris], Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Centre Hospitalier Universitaire de Toulouse (CHU Toulouse), CHU Rouen, Normandie Université (NU), Centre Hospitalier Régional Universitaire de Nancy (CHRU Nancy), Centre hospitalier universitaire de Nantes (CHU Nantes), Hôpital Femme Mère Enfant [CHU - HCL] (HFME), Hospices Civils de Lyon (HCL), CHU Bordeaux [Bordeaux], Centre Hospitalier Régional Universitaire de Tours (CHRU Tours), Centre Hospitalier Universitaire de Reims (CHU Reims), CHU Grenoble, CHU Amiens-Picardie, Centre Hospitalier Universitaire de La Réunion (CHU La Réunion), Centre Hospitalier Universitaire de Nice (CHU Nice), Service de pédiatrie [CHRU Besançon], Centre Hospitalier Régional Universitaire de Besançon (CHRU Besançon), CHU Pontchaillou [Rennes], CHU Trousseau [APHP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), CHU Strasbourg, Institut de Génomique Fonctionnelle (IGF), and Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Montpellier (UM)-Centre National de la Recherche Scientifique (CNRS)

Subjects
Pediatrics, medicine.medical_specialty, Tubulointerstitial nephritis and uveitis, Renal function, tubulointerstitial nephritis and uveitis syndrome, [SDV.MHEP.UN]Life Sciences [q-bio]/Human health and pathology/Urology and Nephrology, Asymptomatic, Dobrin’s syndrome, [SDV.MHEP.PED] Life Sciences [q-bio]/Human health and pathology/Pediatrics, Clinical Research, medicine, [SDV.MHEP.PED]Life Sciences [q-bio]/Human health and pathology/Pediatrics, business.industry, Retrospective cohort study, chronic kidney failure, medicine.disease, [SDV.MHEP.UN] Life Sciences [q-bio]/Human health and pathology/Urology and Nephrology, pediatric, Nephrology, Dobrin's syndrome, Cohort, outcome, medicine.symptom, business, Uveitis, Kidney disease, Rare disease
Abstract

International audience; Introduction: Tubulointerstitial nephritis (TIN) and uveitis (TINU) syndrome is a rare disease. The renal prognosis is generally thought to be better in children with TINU syndrome than in adults. However, data are scarce. We aimed to investigate the long-term renal prognosis in a French cohort of children with TINU syndrome.Methods: We performed a national retrospective study including 23 French pediatric nephrology centers enrolling patients with TINU syndrome diagnosed between January 2000 and December 2018.Results: A total of 46 patients were included (52% female, median age 13.8 years). At diagnosis of TIN, the median estimated glomerular filtration rate (eGFR) was 30.6 ml/min per 1.73 m2 (4.9-62.8). The median time between diagnosis of uveitis and TIN was 0.4 months (-4.1; +17.1). All patients had anterior uveitis, but 12 (29%) were asymptomatic. Nearly all patients (44 of 46) received steroid treatment, and 12 patients (26%) received a second-line therapy. At last follow-up (median 2.8 years), the median eGFR was 87.5 ml/min per 1.73 m2 (60.3-152.7) and

Published
2021

5. Transition et transfert de la néphrologie pédiatrique à la néphrologie adulte : recommandations de la filière maladies rénales rares ORKiD

Author

Robert Novo, Fitsum Guebre-Egziabher, Loïc De Parscau, Aurélia Bertholet-Thomas, Marie Essig, Gérard Champion, Aude Servais, Pierre Cochat, Jennifer Radenac, Anne-Laure Leclerc, Véronique Baudouin, Olivier Dunand, Nathalie Buebuyck, Béatrice Sartoris, Béatrice Langellier-Bellevue, Justine Perrin, Marina Charbit, Dominique Rousiot, Stéphane Burtey, Bruno Moulin, Françoise Broux, Sandrine Lemoine, Yahsou Delmas, Stéphanie Belaiche, Elodie Merieau, and Arvind Nagra

Subjects
03 medical and health sciences, 0302 clinical medicine, Nephrology, Political science, 030232 urology & nephrology, Humanities, Non adherence
Abstract

Resume Des recommandations specifiques a la nephrologie ont ete redigees sous l’egide de l’Association internationale de nephrologie pediatrique en 2011. Ces recommandations preconisent une transition individualisee, debutant entre 12 et 14 ans, avec des informations sur la maladie et les traitements du patient adaptees a l’âge et au niveau intellectuel. Les recommandations insistent sur l’importance d’identifier un referent pediatre et un referent en service adulte, ainsi qu’une equipe paramedicale et de travailleurs sociaux ; elles insistent egalement sur l’importance d’associer les aidants (les familles et l’entourage proche, etc.), la necessite d’un dossier specifique de transition et d’une visite prealable du service adulte avant le transfert. L’International Pediatric Nephrology Association recommande que le transfert soit realise au « bon moment » de la vie du jeune et non a ses 18 ans ; ce « bon moment » etant defini par le moment ou le jeune se sent pret, en periode de stabilite aussi bien medicale que familiale, scolaire et sociale. Afin de fournir au jeune des competences d’autogestion (empowerment) appropriees de sa maladie et de sa vie, dans le cadre de la filiere maladies renales rares (ORKiD), nous avons elabore un programme avec questionnaire adapte a l’âge en nous inspirant de celui mis en place a Southampton au Royaume-Uni. Ce document presente les outils developpes par le groupe de travail de la filiere de soins ORKiD.

Published
2021

6. School level of children carrying a HNF1B variant or a deletion

Author

Annie Lahoche, Pierre Cochat, Cécile Saint-Martin, Isabelle Vrillon, Sylvie Cloarec, Philippe Eckart, Marie-Pierre Lavocat, Gwenaelle Roussey, Olivier Dunand, Brigitte Llanas, J. Tenenbaum, Vincent Guigonis, François Nobili, Nicolas Chassaing, Véronique Baudouin, Christine Bellanné-Chantelot, Laurence Heidet, Loïc De Parscau, Laurence Michel-Calemard, Christine Pietrement, Valérie Bonneville, Stéphane Decramer, Michel Tsimaratos, Fanny Lalieve, Claire Bahans, Nicolas Rodier, Françoise Broux, Vincent Morinière, and Lucie Bessenay

Subjects
Male, Pediatrics, medicine.medical_specialty, Adolescent, Prenatal counseling, Population, Kidney, Article, Academic Performance, Genetics, medicine, Humans, In patient, School level, Child, education, Prospective cohort study, Genetics (clinical), Hepatocyte Nuclear Factor 1-beta, education.field_of_study, business.industry, Neuropsychology, Syndrome, HNF1B, Neurodevelopmental Disorders, Female, business, Gene Deletion, Neuropsychiatric disease
Abstract

The prevalence of neurological involvement in patients with a deletion of or a variant in the HNF1B gene remains discussed. The aim of this study was to investigate the neuropsychological outcomes in a large cohort of children carrying either a HNF1B whole-gene deletion or a disease-associated variant, revealed by the presence of kidney anomalies. The neuropsychological development—based on school level—of 223 children included in this prospective cohort was studied. Data from 180 children were available for analysis. Patients mean age was 9.6 years, with 39.9% of girls. Among these patients, 119 carried a HNF1B deletion and 61 a disease-associated variant. In the school-aged population, 12.7 and 3.6% of patients carrying a HNF1B deletion and a disease-associated variant had special educational needs, respectively. Therefore, the presence of a HNF1B deletion increases the risk to present with a neuropsychiatric involvement when compared with the general population. On the other hand, almost 90% of patients carrying a HNF1B disease-associated variant or deletion have a normal schooling in a general educational environment. Even if these findings do not predict the risk of neuropsychiatric disease at adulthood, most patients diagnosed secondary to kidney anomalies do not show a neurological outcome severe enough to impede standard schooling at elementary school. These results should be taken into account in prenatal counseling.

Published
2019

7. Vers une extension du registre REIN aux patients avec une maladie rénale chronique au stade 5 non traités par dialyse ou greffe ? Étude pilote

Author

Muriel Siebert, Emma Allain-Launay, Olivier Moranne, Florence Glaudet, Cécile Vigneau, Michèle Kessler, Cécile Couchoud, Véronique Baudoin, Olivier Dunand, Marie-Béatrice Nogier, Vincent Allot, Stéphane Edet, Emmanuel Villar, Julien Hogan, Sebastien Gomis, registre Rein, Carole Ayav, François Glowacki, and Natacha Noël

Subjects
Nephrology, medicine.medical_specialty, education.field_of_study, Referral, business.industry, Public health, medicine.medical_treatment, General surgery, Population, 030232 urology & nephrology, 3. Good health, Conservative treatment, 03 medical and health sciences, 0302 clinical medicine, Median time, Internal medicine, Epidemiology, medicine, Renal replacement therapy, business, education
Abstract

To date, it is important to know more about the population of CKD stage 5 patients in order to better understand the practices of access to renal replacement therapy (RRT) or conservative treatment and to anticipate future needs. In April 2015, at the instigation of the Scientific Committee of REIN, a working group was formed to reflect on the opportunity and feasibility of a data collection on these patients. Between September 2017 and March 2018, 21 participating centers included 390 patients over a period of at least one month. The data collected included the patient's living conditions, level of study, mode of referral, clinical data and the therapeutic project. The median age at baseline was 71.4years (IQR: 58.4-80.4), 39.9% were diabetic. The median eGFR was 12mL/min/1.73m2 (IQR: 9-14). At inclusion, 77% of the patients were already followed in nephrology, 11% had been referred by a general practitioner. For the majority of patients included (81%), there was a RRT project. In 10% of cases, there was a project of conservative care, in 5% of cases the project was not yet decided and in 7% the project had not been yet discussed. At the latest news (median time 4.0months), 35% of patients were dialyzed, 9 (2%) have been pre-emptively transplanted, 25 (6%) died, 210 (54%) were still with a CKD stage 5. Our pilot study has shown the feasibility and interest of setting up such a data collection. Such a registry will provide important public health information regarding the demographic of nephrologists and advanced practices nurses. At the local level, this information will help the department to organize themselves to set-up pre-RRT information, implementation of care pathway nurses and multidisciplinary meetings for difficult cases. However, our pilot study shows that to ensure the completeness of the collection, the tracking upstream or downstream of nephrology consultations for eligible patients is essential and therefore requires dedicated human time on site.

Published
2019

8. Preemptive Kidney Transplantation is Associated With Transplantation Outcomes in Children: Results From the French Kidney Replacement Therapy Registry

Author

Marie-Alice Macher, Karen Leffondré, Tim Ulinski, Elodie Merieau, Véronique Baudouin, Stéphane Decramer, Gwenaelle Roussey, Ariane Zaloszyc, Anne-Laure Sellier-Leclerc, Marios Pongas, Rémi Salomon, Iona Madden, Olivier Dunand, Cécile Couchoud, Florentine Garaix, Marc Fila, Jérôme Harambat, Annie Lahoche, Mathilde Prezelin-Reydit, Etienne Bérard, Bordeaux population health (BPH), and Université de Bordeaux (UB)-Institut de Santé Publique, d'Épidémiologie et de Développement (ISPED)-Institut National de la Santé et de la Recherche Médicale (INSERM)

Subjects
medicine.medical_specialty, medicine.medical_treatment, 030232 urology & nephrology, 030204 cardiovascular system & hematology, Cold Ischemia Time, 03 medical and health sciences, 0302 clinical medicine, Kidney Replacement Therapy, Renal Dialysis, Internal medicine, Medicine, Humans, Registries, Child, Dialysis, Kidney transplantation, Transplantation, Deceased donor, business.industry, Graft Survival, medicine.disease, Kidney Transplantation, 3. Good health, Renal Replacement Therapy, Transplantation outcomes, Treatment Outcome, Kidney Failure, Chronic, [SDV.SPEE]Life Sciences [q-bio]/Santé publique et épidémiologie, business, Kidney disease
Abstract

Kidney transplantation (KT) is the optimal treatment for children with end-stage kidney disease. The aim of this study was to evaluate the impact of preemptive kidney transplantation (PKT) and of pretransplant dialysis duration on graft survival among French pediatric kidney transplant recipients.We analyzed all first pediatric kidney-only transplantations performed in France between 1993 and 2012. A Cox multivariable model was used to investigate the association of PKT and pretransplant dialysis time with the hazard of graft failure defined as death, return to dialysis, or retransplant, whichever occurred first.Patients (n = 1911) were included, of which 380 (19.8%) received a PKT. Median time of follow-up was 7.0 y. PKT was associated with a 55% reduction of the hazard of graft failure at any time after KT compared with patients transplanted after dialysis (hazard ratio, 0.45; 95% confidence interval, 0.33-0.62), after adjustment for recipient sex and age, primary kidney disease, donor age and type (living or deceased donor), number of HLA mismatches, cold ischemia time, and year of transplantation. A reduction of the hazard of graft failure was found in PKT whatever the compared duration of dialysis, even when6 mo and whatever the dialysis modality. Results were similar in multiple sensitivity analyses.In France, PKT among pediatric patients is associated with a better graft survival when compared with KT after dialysis, even when6 mo. Based on these findings, we suggest that PKT should be considered as the treatment of choice for children with end-stage kidney disease.

Published
2021

9. Low incidence of SARS-CoV-2, risk factors of mortality and the course of illness in the French national cohort of dialysis patients

Author

Cécile Couchoud, Florian Bayer, Carole Ayav, Clémence Béchade, Philippe Brunet, François Chantrel, Luc Frimat, Roula Galland, Maryvonne Hourmant, Emmanuelle Laurain, Thierry Lobbedez, Lucile Mercadal, Olivier Moranne, Abdelhamid Abbassi, Alain Debure, Abdallah Guerraoui, Abdelatif Benmoussa, Abdelaziz Hamani, Abdelaziz Ziane, Abdelhamid Nefti, Abdelkader Hadj, Abderrahim El Amari, Abderrahmane Ghazali, Abo Bakr Abd El Fatah Mohamed, Achour Laradi, Adel Ben Ahmed, Adel Sahar, Adele Pillet, Adeline Lacraz, Adnan Moinat, Afshin Massoumi, Agathe Pardon, Agnes Caillette Beaudoin, Agnes Chapelet Debout, Agnes Mariot, Ahmed Rachi, Aida Afiani, Aime Remy Boula, Al Jalaby, Alain Cremault, Alain Fournier, Alain Jeanson, Alain Lyon, Alain Nony, Alain Robert, Alain Slingeneyer, Alanor Agnes Labatide, Albane Brodin Sartorius, Albert Bensman, Albert Fournier, Alex Ranlin, Alex Vido Sandor, Alexandra Colombo, Alexandra Duhem, Alexandra Stancu, Alexandre Dufay, Alexandre Dumoulin, Alexandre Ebel, Alexandre Klein, Alexandre Martin, Alexandre Mouneimne, Alexandre Seidowsky, Alfio De Martin, Alfredo Zannier, Ali Aizel, Ali Hafi, Ali Zineddine Diddaoui, Alim Heyani, Alina Mocanu, Alina Preda, Aline Hafi, Aline Talaszka, Alyette Duquesne, Amar Amaouche, Amel Ghemmour, Amelie Simon, Amina Skalli, Amine Boukadida, Amr Ekhlas Ragab Eid, Ana Fedorca, Anabelle Baillet, Anais Poyet, Ancuta Bouffandeau Giorgita, Anderson Ratsimbazafy, Andre Pruna, Angel Argiles, Angelo Testa, Ann Karolien Vandooren, Anne Jolivot, Anne Kolko Labadens, Anne Lataste, Anne Maisin, Anne Paris, Anne Sechet, Anne Wuillai, Anne Elisabeth Heng, Anne Gaelle Josse, Anne Helene Querard, Anne Helene Reboux, Anne Laure Adra, Anne Laure Faller, Anne Laure Leclerc, Anne Laure Poitou, Annie Lahoche Manucci, Antoine Jacquet, Antoine Pommereau, Antoine Thierry, Arezki Adem, Arielle Chapelet, Arnaud Del Bello, Arnaud Delezire, Arnaud Garnier, Arnaud Guerard, Arnaud Klisnick, Arnaud Lionet, Arnaud Roccabianca, Arnaud Stolz, Arthur Capdeville, Asma Allal, Assem Alrifai, Assetou Diarrassouba, Assia Djema, Assia Ferhat Carre, Astrid Godron Dubrasquet, Atman Haddj Elmrabet, Audrey Jegado, Aurelia Bertholet Thomas, Aurelie Davourie Salandre, Aurelie Pajot, Aurelien Lorthioir, Aurelien Tiple, Aurore Sury, Ayman Abokasem, Ayman Sarraj, Bachir Henaoui, Baher Chaghouri, Bassem Wehbe, Beatrice Ball, Beatrice Viron, Belkassem Issad, Benedicte Hodemon Corne, Benedicte Janbon, Benjamin Deroure, Benjamin Savenkoff, Benoit Jonon, Benoit Vendrely, Benyakoub Djelaleddine, Bernard Ohry, Bernard Painchart, Bernard Strullu, Bernard Temperville, Bertin Ebikili, Bertrand Hacq, Bertrand Morel, Bilal Aoun, Blanca Muniz, Bouchra Chlih, Brahim Amara, Brice Mayor, Brigitte Gilson, Brigitte Llanas, Brigitte Zins, Bruno Bourgeon, Bruno Coevoet, Bruno Guery, Bruno Legallicier, Bruno Paris, Bruno Ranchin, Bruno Seigneuric, Camelia Ghiciuc Dita, Camelia Prelipcean, Carine Achard Hottelart, Carine Diet, Carlos Frangie, Carlos Vela, Carmina Muresan, Carole Deprele, Caroline Araujo, Caroline Bidault, Caroline Creput, Caroline Delclaux, Caroline Du Halgouet, Caroline Favennec, Caroline Freguin, Caroline Gourraud Vercel, Caroline Mesguen, Caroline Ndomo Obama, Caroline Poitou, Caroline Preissig Dirhold, Caroline Roubiou, Catherine Albert, Catherine Bessin, Catherine De Marion Gaja, Catherine Godart, Catherine Lasseur, Catherine Leocardi, Catherine Lumbroso, Catherine Melander, Catherine Michel, Catherine Quere Maurouard, Catherine Rouannet, Catherine Taddei, Cathy Verove, Cecile Guiraud, Cecile Tafelin, Cecile Turc Baron, Cedric Formet, Cedric Pinier, Celia Lessore De Ste Foy, Celine Granolleras, Chaouki Bennini, Charles Cartou, Charles Chazot, Charlotte Jouzel, Cherif Badid, Christa Roubicek, Christel Viaud, Christelle Verrier, Christian Chuet, Christian Combe, Christian Dabot, Christian Duvic, Christian Emond, Christian Lagarde, Christian Lamotte, Christian Pain, Christiane Mousson, Christie Lorriaux, Christine Beauchamp, Christine Fumeron, Christine Le Gurun, Christine Leroy, Christine Pietrement, Christine Richer, Christophe Bouaka, Christophe Charasse, Christophe Goupy, Christophe Ridel, Cindy Castrale, Cindy Detourne, Clair Francois, Claire Presne, Claire Trivin, Clarissa Von Kotze, Claude Bernard, Claude Bonniol, Claude Desvergnes, Claude Raharivelina, Claudia Nistor, Claudine Gueret, Claudine Lloret, Claudine Saltiel, Clelia Rosati, Clementine Rabate, Corina Stanescu, Corinne Ferrandini, Corinne Guibergia, Corinne Lemoine, Corinne Passeron, Cynthia Kahil, Cyril Garrouste, Cyril Vo Van, Cyrille Jolimoy, Dalila Kesraoui, Damien Jolly, Damien Thibaudin, Dan Teboulle, Daniel Daubresse, Daniel Louvet, Daniel Rasamimanantsoa, Daniel Toledano, Daniela Babici, Daniela David, Daniela Dincu, Danielle Bruno, Delia May, Delphine Haussaire, Delphine Henriet Viprey, Denis Bugnon, Denis Fouque, Denis Morin, Derradji Nour, Diab Mohamed Mahmoud, Diana Istrati Cristescu, Didier Aguilera, Didier Coste, Didier Hamel, Didier Le Chapois, Didier Testou, Dilaver Erbilgin, Djamal Dahmane, Doan Bui Quang, Dominique Bertrand, Dominique Besnier, Dominique Blanchier, Dominique Briffa, Dominique Caux, Dominique Durand, Dominique Fleury, Dominique Guerrot, Dominique Hestin, Dominique Jaubert, Dominique Joly, Dominique Lombart, Dominique Pagniez, Dominique Pierre, Dominique Schohn, Donatien Ikonga, Dorina Visanica, Dorothee Bazin, Edouard Boury, Edouard Maksour, Ekoue Agbonon, Elarbi Harrami, Elena Marcu, Elena Tudorache, Elisabeth Caniot, Elisabeth Semjen, Elisabeth Tomkiewicz, Elise Scheidt, Elke Gaboriau, Elodie Lamouroux, Elsa Guiard, Elsa Martin Passos, Emerson Nsembani, Emilie Fache, Emilie Kalbacher, Emilie Pambrun, Emilie Pincon, Emma Allain Launay, Emmanuel Baron, Emmanuel Dupuis, Emmanuel Villar, Emmanuelle Charlin, Emmanuelle Hecquet, Emmanuelle Kohler, Emmanuelle Rosier, Enrique Figueroa, Eric Azoulay, Eric Canivet, Eric Daugas, Eric Gauthier, Eric Laruelle, Eric Le Guen, Eric Legrand, Eric Moumas, Eric Postec, Eric Prinz, Eric Renaudineau, Estelle Desport, Estelle Ricard Sutra, Etienne Berard, Etienne Ged, Etienne Robin, Eve Vilaine, Evelyne Bargas, Evelyne Mac Namara, François Combarnous, Fatima Yazbeck, Fabien Gerard, Fabien Metivier, Fabien Parazols, Fabien Soulis, Fabrice Garnier, Fadhila Pech Messaoudene, Fadi Haidar, Fanny Boullenger, Fanny Lepeytre, Fanny Leroy, Fares Frejate, Farid Bellahsene, Farid Bellhasene, Farid Saidani, Fatouma Toure, Faycal Kriaa, Fazia Nemmar, Fernando Vetromile, Florence Chalmin, Florence Lucats, Florence Sens, Florence Villemain, Florent Plasse, Fouad Lebhour, Francis Schillinger, Franck Berge, Franck Bourdon, Franck Bridoux, Franck Reynaud, Francois Babinet, Francois Basse, Francois Chantrel, Francois Clair, Francois Coulomb, Francois De Cornelissen, Francois Glowacki, Francois Marchal, Francois Maurice, Francois Nobili, Francois Pourreau, Francois Provot, Francois Roux Amani, Francoise Broux, Francoise Bulte, Francoise Heibel, Francoise Leonetti, Francoise Moussion Schott, Frank Le Roy, Frederic Besson, Frederic Lavainne, Frederic Tollis, Frederique Bocquentin, Frederique Meeus, Frederique Vecina, Friederike Von Ey, Gabriel Balit, Gabriel Choukroun, Gabriel Gruget, Gabriel Huchard, Gabriella Golea, Gabrielle Duneau, Gaelle Lefrancois, Gaelle Pelle, Gaetan Lebrun, Genevieve Dumont, Georges Brillet, Georges Deschenes, Georges Mourad, Georges Stamatakis, Geraldine Cazajous, Geraldine D'ythurbide, Geraldine Robitaille Wiart, Gerard Cardon, Gerard Champion, Gerard Deschodt, Gerard Mangenot, Gerard Motte, Gerard Schortgen, Ghada Boulahia, Ghassan Maakaroun, Ghylene Bourdat Michel, Gilbert Zanetta, Gilles Hufnagel, Gilles Messier, Giorgina Piccoli, Gregoire Couvrat Desvergnes, Guillaume Bobrie, Guillaume Bonnard, Guillaume Clement, Guillaume Jean, Guillaume Queffeulou, Guillaume Seret, Guillaume Vernin, Guy Delavaud, Guy Lambrey, Guy Rostoker, Gwenaelle Poussard, Gwenaelle Roussey Kesler, H. Leon, Habib Aboubekr, Hacene Boulechfar, Hacene Sekhri, Hadia Hebibi, Hadjira Benalia, Hafed Fessi, Hafsabhai Atchia, Haiat Bittar, Hakim Maiza, Hakim Mazouz, Hamid El Ali, Hammouche Bougrida, Hans Van Der Pijl, Hassan Lokmane, Hassane Izzedine, Hassen Adda, Helene De Preneuf, Helene Leray, Helene Philippot, Henri Boulanger, Henri Merault, Henri Renaud, Herve Bonarek, Herve Maheut, Hilaire Nzeyimana, Hocine Mehama, Hocine Zaidi, Hugo Weclawiak, Hugues Flodrops, Huseyin Karaaslan, Ibrahim Haskour, Ihssen Belhadj, Imad Almoubarak, Imad Haddad, Ines Castellano, Ines Ferrandiz, Ioana Daniliuc, Ioana Darie, Ioana Enache, Ionut Prunescu, Irenee Djiconkpode, Irina Shahapuni, Isabelle Bouchoule, Isabelle Devriendt, Isabelle Kazes, Isabelle Kolb, Isabelle Landru, Isabelle Poli, Isabelle Rey, Isabelle Segalen, Isabelle Selcer, Isabelle Vernier, Isabelle Vrillon, Ismahane Guenifi, J. Dominique Gheerbrandt, Jacky Potier, Jacques Becart, Jacques Cledes, Jacques Ducros, Jacques Duvic, Jacques Fourcade, Jacques Gaultier, Jacques Jurine, Jacques Lebleu, Jacques Ollier, Jacques Ibsen Charles, Jamal Yazji, Janette Mansour, Jean Arnautou, Jean Brocard, Jean Carolfi, Jean Montoriol, Jean Baptiste Gouin, Jean Bernard Palcoux, Jean Christophe Bendini, Jean Claude Aldigier, Jean Claude Alphonse, Jean Daniel Delbet, Jean Francois Bonne, Jean Francois Cantin, Jean Francois De Fremont, Jean Francois Dessassis, Jean Francois Subra, Jean Francois Valentin, Jean Francois Verdier, Jean Jacques Dion, Jean Jacques Haultier, Jean Jacques Montseny, Jean Louis Bacri, Jean Louis Bouchet, Jean Luc Mahe, Jean Marc Chalopin, Jean Marc Gabriel, Jean Marc Hurot, Jean Marc Lanau, Jean Marie Batho, Jean Marie Coulibaly, Jean Michel Hardin, Jean Michel Marc, Jean Michel Poux, Jean Michel Rebibou, Jean Michel Tivollier, Jean Noel Ottavioli, Jean Paul Faucon, Jean Paul Imiela, Jean Paul Jaulin, Jean Paul Masselot, Jean Paul Ortiz, Jean Philippe Bourdenx, Jean Philippe Devaux, Jean Philippe Hammelin, Jean Pierre Rivory, Jean Pierre Wauquier, Jean Rene Larue, Jean Rene Mondain, Jean Sebastien Borde, Jean Simon Virot, Jean Yves Bosc, Jedjiga Achiche, Jennifer Parasote, Jeremie Diolez, Jerome Harambat, Jerome Potier, Jerome Sampol, Jihad Mustel, Jean Jacques Lefevre, Jocelyne Maurizi, Joel Gamberoni, Joelle Claudeon, Joelle Terzic, Joffrey Rogol, Johnny Sayegh, Jorge Cardozo, Jose Brasseur, Jose Guiserix, Joseph Barsumau, Julie Albaret, Julie Beaume, Julie Sohier Attias, Julien Dehay, Julien Hogan, Julien Journet, Julien Ott, Juliette Baleynaud, Justine Bacchetta, Justine Faucher, Kamel Yousfi, Karim Dardim, Karine Clabault, Karine Moreau, Kedna Thomas, Khaled Sirajedine, Khalil Chedid, Khalil El Kaeoui, Khalil El Karoui, Khedidja Bouachi, Kheira Hue, Khuzama El Nasser, Kodso Akposso, Kristian Kunz, Krzysztof Bijak, Lilia Kihal, L. Rasoloarijaona, Laid Harbouche, Larbi Bencheikh, Larbie Lamriben, Latifa Hanafi, Laura Braun Parvez, Laure Champion, Laure Croze, Laure Eprinchard, Laure Patrier, Laurence Nicolet, Laurence Vrigneaud, Laurent Duflot, Leandre Mackaya, Leila Chenine, Leon Odry, Lili Taghipour Tamiji, Lilia Antri Bouzar, Liliane Ngango Nga Messi, Lionel Le Mouellic, Lise Mandart, Lise Weis, Lise Marie Pouteau, Lora Georgieva, Lorita Vitanova, Lotfi Chalabi, Luc Delvallez, Luc Fromentin, Luc Marty, Luc Monjot, Luciana Spataru, Lucie Bessenay, Lucie Boissinot, Lucie Wajsbrot, Lucien Rakoff, Ludivine Lebourg, Lydie Perez, Lyliane Lafage, Lynda Azzouz, Madeleine Dumoulin, Messaoud Ouziala, Maan Joseph, Mabrouk Brahimi, Maeva Wong Fat, Magalie Fort, Magued Nakhla, Mahdi Abtahi, Mahen Albadawy, Mahmoud Alouach, Mahmoud Mezghani, Maite Daroux, Maklouf Boukelmoune, Malek Dhib, Malik Touam, Malina Dubau, Mamadou Balde, Man Nguyen Khoa, Manfred Ismer, Manolie Mehdi, Manon Laforet, Marc Bouiller, Marc Eugene, Marc Fila, Marc Hazzan, Marc Kribs, Marc Ladriere, Marc Lebot, Marc Padilla, Marc Souid, Marcel Marraoui, Maren Burbach, Maria Manescu, Maria Eugenia Noguera Gonzalez, Mariana Revenco, Marianne Terrasse, Marie Essi, Marie Alice Macher, Marie Beatrice Nogier, Marie Cecile Cazin, Marie Christine Schweitzer Camoin, Marie Christine Thouret, Marie Claude Hannaert, Marie France Servel, Marie Helene Chabannier, Marie Jeanne Coudert Krier, Marie Noelle Catoliquot, Marie Paule Guillodo, Marie Sophie Gavard, Marie Xaviere Vairon Codaccioni, Marina Rabec, Marine Freist, Marion Gauthier, Marion Lemaire, Marion Mehrenberger, Marion Venot, Marios Pongas, Marlene Beaubrun Diant, Martial Levannier, Martine Bertaux, Mathieu Jablonski, Mathieu Sacquepee, Mathilde Dargelos, Mathilde Lemoine, Mathilde Tamain, Matthieu Monge, Matthieu Reberolle, Maud Cousin, Maud Francois, Maurice Baron, Maxime Hoffmann, Maxime Ingwiller, Maxime Touzot, Mederick Mohajer, Mehadji Maaz, Melanie Hanoy, Melanie Marroc, Melodie Cuny, Menno Van Der Straaten, Mf. Serveaux, Michel Basteri, Michel Fen Chong, Michel Hecht, Michel Massad, Michel Normand, Michel Olmer, Michel Tolani, Michel Tsimaratos, Michele Hemery, Michele Kessler, Miguel Esposito, Milad Shenouda, Mimi Kareche, Mina Khalili, Mirella Diaconita, Mohamad Khair Rifard, Mohamed Aladib, Mohamed Belmouaz, Mohamed Brahim, Mohamed Diouani, Mohamed Fodil Cherif, Mohamed Jamali, Mohamed Maghlaoua, Mohamed Meddeb, Mohamed Ramdane, Mohamed Rifaat, Mohamed Sharifull Islam, Mohamed Adnan Abbade, Mokhtar Amrandi, Mokhtar Chawki, Monica Ciobotaru, Monica Indrieis, Monique Chanas, Monique Hoarau, Monzer Tomeh, Moufida Bellou, Mouloud Bouzernidj, Mounia Ammor, Mounir Guergour, Mountassir Benzakour, Mourad Hachicha, Moussa Coulibaly, Mustafa Smati, Mustapha Al Morabiti, Mustapha Amirou, Myriam Isnard, Myriam Pastural, Myriam Pujo, Nourredine Boumendjel, Nabil Majbri, Nabila Goumri, Nadege Mingat, Nader Bassilios, Nadia Kerkeni, Nadia Sedrati, Nadia Soltani, Nadine Maroun, Nadine Neyrat, Nahn Luang, Najeh El Esper, Naji Ammar, Nasredine Ghali, Nasser Hamdini, Natacha Noel, Natacha Potelune, Nathalie Maisonneuve, Nathalie Pertuiset, Nathalie Raynal, Nathalie Vittoz, Nazim Terki, Nelly Castin, Nestor Nankeu, Nicolas Bouvier, Nicolas Keller, Nicolas Legros, Nicolas Peters, Nicolas Quirin, Nicole Lefrancois, Nicole Monnier, Nicole Rance, Niels Bruckmann, Noel Mertens, Nolwenn Lorcy, Olivia Gilbert, Olivier Coldefy, Olivier Drouineau, Olivier Dunand, Olivier Fritz, Olivier Imhoff, Olivier Kourilsky, Olivier Lavelle, Olivier Papin, Olivier Roques, Ophelie Le Maner, Oussamah Fikri Benbrahim, Pablo Antonio Erina Torres, Pablo Antonio Urena Torres, Paolo Malvezzi, Pascal Bindi, Pascal Cluzel, Pascal Fontanier, Pascal Wheatley, Pascale Depraetre, Pascale Dubosq, Pascale Halin, Pascale Sebahoun, Pascale Siohan, Pascale Testevuide, Patrice Deteix, Patrice Nolen, Patricia Hue, Patricia Lemarchand, Patrick Donnadieu, Patrick Fievet, Patrick Fohrer, Patrick Francais, Patrick Giraud, Patrick Hallonet, Patrick Henri, Patrick Michaut, Patrick Niaudet, Patrick Pauly, Patrick Thomas, Patrik Deleaval, Paul Finielz, Paul Stroumza, Paule Hardy Yverneau, Pauline Caillard, Pedro Palacin, Perrine Aubertin, Philippe Attias, Philippe Chauveau, Philippe Coindre, Philippe Coste, Philippe Dubot, Philippe Fournier, Philippe Hiernaux, Philippe Jousset, Philippe Lan Yue Wah, Philippe Lang, Philippe Le Cacheux, Philippe Martin Dupont, Philippe Michel, Philippe Mirgaine, Philippe Moriniere, Philippe Nicoud, Philippe Rieu, Philippe Rousseau, Philippe Sporer, Philippe Thorel, Philippe Vanhille, Philippe Vigeral, Philippe Zaoui, Pierre Bataille, Pierre Brignon, Pierre Filipozzi, Pierre Housset, Pierre Peyronnet, Pierre Ramperez, Pierre Vautrin, Pierre Alexandre Michel, Pierre Francois Westeel, Pierre Louis Carron, Pierre Yves Durand, Pierrot Parent, Piotr Seniuta, François Kuentz, Rabah Fraoui, Rachel Tetaz, Rachid Amaria, Rachid Bourouma, Rachid Djeffal, Rachida Nebbad, Radia Allal, Radu Dimulescu, Rafaat Boustani, Rafik Mesbah, Raifat Makdassi, Raji Diab, Raluca Puslenghea, Raoul Roura, Rateb Khayat, Raymond Azar, Raymond Frayssinet, Regine Monkam, Rehouni Boulahrouz, Remi Boudet, Renato Demontis, Renaud Gansey, Rene Cuvelier, Renee Schmitt, Reschad Noordally, Reynald Binaut, Rezkallah Latif, Richard Dufresne, Richard Montagnac, Richard Reade, Robert Genin, Robert Novo, Rocsana Fickl, Roger Dufresne, Roger Magnol, Roland Issautier, Romain Mortelette, Ronan Delaval, Ronan Lohro, Roseline M'barga, S. Beau, Clémentine Dupuis, Marie Jacques Vidil, Sabria Hacini, Said Dahmoune, Saliha Lekhal, Salima Ahriz Sakso, Salima Saksi, Salvatore Citarda, Samir Boubenider, Samuel Kassis, Sandra Verhille, Sandrine Genestier, Sandrine Muller, Saoussen Krid, Sarah Richter, Sebastien Delbes, Sebastien Mailliez, Sebastien Veillon, Sébastien Nony, Seddick Benarbia, Severine Beaudreuil, Sidi Ali Benyaghla, Simon Duquennoy, Simona Baluta, Simona Boncila, Sonia Mzoughi, Sonia Ribal, Sophie Acamer, Sophie Chauvet, Sophie Girerd, Sophie Ozenne, Sophie Parahy, Sophie Rubens Duval, Sophie Taque, Soraya Menouer, Soumaya Chargui, Stanislas Bataille, Stephane Barbier, Stephane Billion, Stephane Roueff, Stephane Torner, Stephane Jean Martin, Stephanie Coupel, Sylvie Cloarec, Sylvie Lavaud, Sylvie Leou, T. Chatelet, Tania Onesta, Tassadit Benhabib, Tayeb Bensalem, Theodora Dimulescu, Theophile Sawadogo, Thibault Dolley Hitze, Thierry Baranger, Thierry Boudemaghe, Thierry Hannedouche, Thierry Krummel, Thierry Milcent, Thomas Dervaux, Thomas Guincestre, Thomas Kofman, Thomas Raphael, Thomas Sadreux, Tim Ulinski, Tiphaine Guyon Roger, Tomas Serrato, Tomek Kofman, Tony Wong, Toufik Boubia, Ubald Assogba Gbindoun, Usama Khuzaie, Valerie Caudwell, Valerie Chatelet, Valerie Crougneau, Valerie De Precigout, Valerie Drouillat, Valerie Galantine, Valerie Granveau Hugot, Valerie Leroy, Veronique Boubia, Veronique Falque, Veronique Fournier, Veronique Queron, Veronique Viviani, Victor Gueuttin, Victor Panescu, Victorio Menoyo Calonge, Viet Nguyen, Vincent Allot, Vincent Delattre, Vincent Leduc, Vincent Pradier, Violaine Emal Aglae, Viorica Badulescu, Virginia Molina, Virginie Besson, Virginie Chaigne, Waddah Jaber, Wael Boudi, Wael El Haggan, Wen Qin Guillon, Wided Tabbi Aneni, William Hanf, Wladimir Kohn, Xavier Bellenfant, Xavier Moreau Gaudry, Yahsou Delmas, Yannick Knefati, Yannick Saingra, Yannick Tirolien, Youssef Mann, Yvan Brunak, Yves Dimitrov, Yves Doussy, Yves Tanter, Zaid Benabid, Zaara Soltani, Zacharia Boukerroucha, Zafer Takla, Zana Ramanantsialonina, Zara Dickson, Zead Tubail, Zoe Koochaki Pour, Zohra Boukhalfa, Zohra Jacquot, Agence de la biomédecine [Saint-Denis la Plaine], Centre Hospitalier Régional Universitaire de Nancy (CHRU Nancy), Université de Caen Normandie (UNICAEN), Normandie Université (NU), CALYDIAL Vienne, Partenaires INRAE, Centre hospitalier universitaire de Nantes (CHU Nantes), Hôpital Armand Trousseau [AP-HP] (AP-HP), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Centre recherche en CardioVasculaire et Nutrition = Center for CardioVascular and Nutrition research (C2VN), Aix Marseille Université (AMU)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Institut National de Recherche pour l’Agriculture, l’Alimentation et l’Environnement (INRAE), Centre de néphrologie et transplantation rénale [Hôpital de la Conception - APHM], Assistance Publique - Hôpitaux de Marseille (APHM)-Hôpital de la Conception [CHU - APHM] (LA CONCEPTION), Service d'Epidémiologie et Evaluations Cliniques [CHRU Nancy] (Pôle S2R), Centre Universitaire des Maladies Rénales [CHU Caen] (CUMR Caen), Normandie Université (NU)-Normandie Université (NU)-CHU Caen, Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN)-Tumorothèque de Caen Basse-Normandie (TCBN), Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Institut de Génomique Fonctionnelle (IGF), Université de Montpellier (UM)-Université Montpellier 1 (UM1)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Montpellier 2 - Sciences et Techniques (UM2)-Centre National de la Recherche Scientifique (CNRS), Groupe hospitalier de la région de Mulhouse Sud-Alsace (GHRMSA), Centre Hospitalier Universitaire de Nîmes (CHU Nîmes), Aide à la Décision pour une Médecine Personnalisé - Laboratoire de Biostatistique, Epidémiologie et Recherche Clinique - EA 2415 (AIDMP), Université Montpellier 1 (UM1)-Université de Montpellier (UM), Assistance Publique - Hôpitaux de Marseille (APHM), Service de diabétologie [CHU Pitié-Salpétrière], CHU Pitié-Salpêtrière [AP-HP], Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), CHU Caen, Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN)-Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN)-Université de Caen Normandie (UNICAEN), Service de Diabétologie [CHU Pitié-Salpétrière], and Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)

Subjects
0301 basic medicine, Male, medicine.medical_treatment, [SDV]Life Sciences [q-bio], 030232 urology & nephrology, Hemodialysis, Home, Disease, MESH: COVID-19 / therapy, registry, Ambulatory Care Facilities, [SDV.MHEP.UN]Life Sciences [q-bio]/Human health and pathology/Urology and Nephrology, MESH: Aged, 80 and over, 0302 clinical medicine, [SDV.MHEP.MI]Life Sciences [q-bio]/Human health and pathology/Infectious diseases, Risk Factors, 80 and over, Prevalence, covid, dialysis, epidemiology, mortality, Aged, Aged, 80 and over, COVID-19, Case-Control Studies, Critical Care, Female, France, Humans, Incidence, Middle Aged, Patient Acuity, Protective Factors, Registries, Renal Dialysis, SARS-CoV-2, Sex Factors, Hypoalbuminemia, MESH: France / epidemiology, education.field_of_study, Incidence (epidemiology), 3. Good health, MESH: COVID-19 / epidemiology, Nephrology, Hemodialysis, medicine.medical_specialty, Population, Lower risk, Article, 03 medical and health sciences, MESH: Sex Factors, Internal medicine, medicine, MESH: SARS-CoV-2, MESH: Renal Dialysis / statistics & numerical data, education, Dialysis, Vascular disease, business.industry, medicine.disease, Former Smoker, MESH: Critical Care / statistics & numerical data, 030104 developmental biology, [SDV.SPEE]Life Sciences [q-bio]/Santé publique et épidémiologie, business, Home, [SDV.MHEP]Life Sciences [q-bio]/Human health and pathology, MESH: Hemodialysis, Home / statistics & numerical data
Abstract

The aim of this study was to estimate the incidence of COVID-19 disease in the French national population of dialysis patients, their course of illness and to identify the risk factors associated with mortality. Our study included all patients on dialysis recorded in the French REIN Registry in April 2020. Clinical characteristics at last follow-up and the evolution of COVID-19 illness severity over time were recorded for diagnosed cases (either suspicious clinical symptoms, characteristic signs on the chest scan or a positive reverse transcription polymerase chain reaction) for SARS-CoV-2. A total of 1,621 infected patients were reported on the REIN registry from March 16th, 2020 to May 4th, 2020. Of these, 344 died. The prevalence of COVID-19 patients varied from less than 1% to 10% between regions. The probability of being a case was higher in males, patients with diabetes, those in need of assistance for transfer or treated at a self-care unit. Dialysis at home was associated with a lower probability of being infected as was being a smoker, a former smoker, having an active malignancy, or peripheral vascular disease. Mortality in diagnosed cases (21%) was associated with the same causes as in the general population. Higher age, hypoalbuminemia and the presence of an ischemic heart disease were statistically independently associated with a higher risk of death. Being treated at a selfcare unit was associated with a lower risk. Thus, our study showed a relatively low frequency of COVID-19 among dialysis patients contrary to what might have been assumed., Graphical abstract

Published
2020

10. Quality of life in adolescents with chronic kidney disease who initiate haemodialysis treatment

Author

Annie Lahoche, Isabelle Vrillon, Julie Berbis, Françoise Broux, Gwenaelle Roussey, Sylvie Cloarec, Jérôme Harambat, Michel Tsimaratos, Olivier Dunand, Mohamed Boucekine, Rémi Salomon, Soraya Menouer, Stéphanie Clavé, Bruno Ranchin, Arnaud Garnier, Marc Fila, Pascal Auquier, Georges Deschênes, Centre d'études et de recherche sur les services de santé et la qualité de vie (CEReSS), Aix Marseille Université (AMU), Hôpital de la Timone [CHU - APHM] (TIMONE), Service de Néphrologie Rhumatologie Dermatologie, Hôpital Femme Mère Enfant [CHU - HCL] (HFME), Hospices Civils de Lyon (HCL)-Hospices Civils de Lyon (HCL)-HCL Groupement Hospitalier Est-Centre de référence Maladies Rénales Rares, Service de néphrologie 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), Service de Néphrologie, Centre Hospitalier Universitaire de La Réunion (CHU La Réunion), CHU Toulouse [Toulouse], Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Centre hospitalier universitaire de Nantes (CHU Nantes), Hôpital Charles Nicolle [Rouen], Centre de Référence du Sud Ouest des Maladies Rénales Rares, CHU Toulouse [Toulouse]-Hôpital des Enfants, Bordeaux population health (BPH), Université de Bordeaux (UB)-Institut de Santé Publique, d'Épidémiologie et de Développement (ISPED)-Institut National de la Santé et de la Recherche Médicale (INSERM), Centre Hospitalier Régional Universitaire de Tours (CHRU Tours), Hôpital de Hautepierre [Strasbourg], Service de néphrologie et pédiatrie générale [CHU Trousseau], Université Pierre et Marie Curie - Paris 6 (UPMC)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Trousseau [APHP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Sorbonne Université (SU), Centre Hospitalier Régional Universitaire de Nancy (CHRU Nancy), French national research program for clinical research (PHRC), HCL Groupement Hospitalier Est-Hôpital Femme Mère Enfant [CHU - HCL] (HFME), Hospices Civils de Lyon (HCL)-Hospices Civils de Lyon (HCL)-Centre de référence Maladies Rénales Rares, Centre Hospitalier Régional Universitaire de Tours (CHRU TOURS), Centre Hospitalier Universitaire de Toulouse (CHU Toulouse), CHU Rouen, Normandie Université (NU)-Normandie Université (NU), CHU Trousseau [APHP], and Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)

Subjects
Adult, Male, Parents, Quality of life, Gerontology, medicine.medical_specialty, Coping (psychology), Medical staff, Multivariate analysis, Adolescent, Population, 030232 urology & nephrology, 030204 cardiovascular system & hematology, Adolescents, lcsh:RC870-923, End stage renal disease, End-stage renal disease, 03 medical and health sciences, Leisure Activities, 0302 clinical medicine, Renal Dialysis, Internal medicine, Adaptation, Psychological, Health care, medicine, Humans, Prospective Studies, Renal Insufficiency, Chronic, Child, education, Bayesian models, Initiating haemodialysis, Coping strategies, education.field_of_study, business.industry, Middle Aged, lcsh:Diseases of the genitourinary system. Urology, medicine.disease, 3. Good health, Nephrology, [SDV.SPEE]Life Sciences [q-bio]/Santé publique et épidémiologie, Female, France, Self Report, business, Research Article, Kidney disease
Abstract

Background To describe the quality of life of adolescents initiating haemodialysis, to determine the factors associated with quality of life, and to assess coping strategies and their impact on quality of life. Methods All adolescents initiating haemodialysis between September 2013 and July 2015 in French paediatric haemodialysis centres were included. Quality of life data were collected using the “Vécu et Santé Perçue de l’Adolescent et l’Enfant” questionnaire, and coping data were collected using the Kidcope questionnaire. Adolescent’s quality of life was compared with age- and sex-matched French control. Results Thirty-two adolescents were included. Their mean age was 13.9 ± 2.0 years. The quality of life score was lowest in leisure activities and highest in relationships with medical staff. Compared with the French control, index, energy-vitality, relationships with friends, leisure activities and physical well-being scores were significantly lower in haemodialysis population. In multivariate analyses, active coping was positively associated with quality of life and especially with energy-vitality, relationships with parents and teachers, and school performance. In contrast, avoidant and negative coping were negatively associated with energy-vitality, psychological well-being and body image for avoidant coping, and body image and relationships with medical staff for negative coping. Conclusions The quality of life of haemodialysis adolescents, and mainly the dimensions of leisure activities, physical well-being, relationships with friends and energy-vitality, were significantly altered compared to that of the French population. The impact of coping strategies on quality of life seems to be important. Given the importance of quality of life and coping strategies in adolescents with chronic disease, health care professionals should integrate these aspects into care management.

Published
2019

11. Treatment and outcome of congenital nephrotic syndrome

Author

Véronique Baudouin, Ariane Zaloszyc, Maud Dehennault, Sandra Bérody, Vincent Morinière, Elodie Merieau, Bernard Boudaillez, Rémi Salomon, Christine Pietrement, Adrien May, Laurence Heidet, Hugues Flodrops, Marie-Alice Macher, Loïc De Parscau, Rachel Vieux, Françoise Monceaux, Justine Bacchetta, Marc Fila, Arnaud Garnier, Caroline Rousset-Rouvière, Ferielle Louillet, J. Tenenbaum, Jérôme Harambat, Olivier Dunand, Olivier Gribouval, Olivia Boyer, Tim Ulinski, Sophie Taque, Gwenaelle Roussey, Patrick Niaudet, Bordeaux population health (BPH), and Université de Bordeaux (UB)-Institut de Santé Publique, d'Épidémiologie et de Développement (ISPED)-Institut National de la Santé et de la Recherche Médicale (INSERM)

Subjects
Male, Pediatrics, medicine.medical_specialty, Nephrotic Syndrome, medicine.medical_treatment, 030232 urology & nephrology, 030204 cardiovascular system & hematology, Nephrectomy, LEHA, 03 medical and health sciences, 0302 clinical medicine, medicine, Humans, Cumulative incidence, Congenital nephrotic syndrome, Dialysis, Retrospective Studies, Transplantation, business.industry, Incidence, Infant, Newborn, Infant, Membrane Proteins, medicine.disease, 3. Good health, Survival Rate, Treatment Outcome, Nephrology, Mutation, Disease Progression, Female, [SDV.SPEE]Life Sciences [q-bio]/Santé publique et épidémiologie, France, Hemodialysis, business, Nephrotic syndrome, Kidney disease
Abstract

Background Recommendations for management of Finnish-type congenital nephrotic syndrome (CNS) followed by many teams include daily albumin infusions, early bilateral nephrectomy, dialysis and transplantation. We aimed to assess the treatment and outcome of patients with CNS in France. Methods We conducted a nationwide retrospective study on 55 consecutive children born between 2000 and 2014 treated for non-infectious CNS. Results The estimated cumulative incidence of CNS was 0.5/100 000 live births. The underlying defect was biallelic mutations in NPHS1 (36/55, 65%), NPHS2 (5/55, 7%), PLCE1 (1/55, 2%), heterozygous mutation in WT1 (4/55, 7%) and not identified in nine children (16%). Fifty-three patients (96%) received daily albumin infusions from diagnosis (median age 14 days), which were spaced and withdrawn in 10 patients. Twenty children (35%) were managed as outpatients. Thirty-nine patients reached end-stage kidney disease (ESKD) at a median age of 11 months. The overall renal survival was 64% and 45% at 1 and 2 years of age, respectively. Thirteen children died during the study period including four at diagnosis, two of nosocomial catheter-related septic shock, six on dialysis and one after transplantation. The remaining 13 patients were alive with normal renal function at last follow-up [median 32 months (range 9-52)]. Renal and patient survivals were longer in patients with NPHS1 mutations than in other patients. The invasive infection rate was 2.41/patient/year. Conclusions Our study shows: (i) a survival free from ESKD in two-thirds of patients at 1 year and in one-half at 2 years and (ii) a significant reduction or even a discontinuation of albumin infusions allowing ambulatory care in a subset of patients. These results highlight the need for new therapeutic guidelines for CNS patients.

Published
2019

12. Pharmacokinetics of mycophenolate mofetil in children with lupus and clinical findings in favour of therapeutic drug monitoring

Author

Vincent Guigonis, Jean-Baptiste Woillard, J. Tenenbaum, Olivier Dunand, Jérôme Harambat, Stéphane Decramer, Bruno Ranchin, Etienne Bérard, Pierre Marquet, Michel Fischbach, Franck Saint-Marcoux, Rémi Salomon, and Brigitte Bader-Meunier

Subjects
Pharmacology, medicine.medical_specialty, medicine.diagnostic_test, business.industry, Area under the curve, Context (language use), Mycophenolate, Logistic regression, Gastroenterology, Mycophenolic acid, 3. Good health, Pharmacokinetics, Therapeutic drug monitoring, Internal medicine, medicine, Pharmacology (medical), business, PK/PD models, medicine.drug
Abstract

AIMS: The use of mycophenolate mofetil (MMF) in children with Systemic Lupus Erythematosus (SLE) is increasing. However, the clinical benefit of its monitoring has been scarcely studied, and little is known about its pharmacokinetics in this context. The objectives of the present study were: (i) to describe mycophenolic acid (MPA; the active moiety of MMF) pharmacokinetics; (ii) to develop a Bayesian estimator (BE) allowing the determination AUC (Area under the curve) from a limited number of blood samples; and (ii) to explore the relationships between exposure indices to MPA and the clinical status in children with SLE. METHODS: This was a retrospective study including 36 children with SLE, extracted from the expert system ISBA, for whom full- pharmacokinetic profiles of MPA were collected together with clinical data. A pharmacokinetic model and a BE were developed using an iterative two-stage Bayesian approach. ROC curve analyses and logistic regressions was used to investigate the association of exposure and active disease. RESULTS: A pharmacokinetic model and a BE were developed that allowed good AUC estimation performance (bias±SD=-0.02±0.15). ROC curve analyses showed that AUC/dose

Published
2014

13. 06Adverse effects of rituximab used in children with idiopathic nephrotic syndrome. A multicentric retrospective study

Author

Ferielle Louillet, R. Novo, Elodie Merieau, Roman Klifa, François Nobili, Olivier Dunand, Gérard Champion, F. Laliève, Ariane Zaloszyc, Olivia Boyer, V. Lefranc, S. Crepin, Etienne Bérard, N. Jay, Sophie Taque, Justine Bacchetta, Djamal Djeddi, J.D. Delbet, D. Postil, Gwenaelle Roussey, C. Samaille, S. Tellier, L. De Parscaud, E. Cousin, Brigitte Llanas, Christine Pietrement, C. Rousset, M. Cailliez, C. Bahans, L. Ichay, Paloma Maria Parvex, and Vincent Guigonis

Subjects
Pediatrics, medicine.medical_specialty, business.industry, Hemodynamics, Retrospective cohort study, Disease, Idiopathic Nephrotic Syndrome, Alternative treatment, Relative risk, Pediatrics, Perinatology and Child Health, medicine, Rituximab, business, Adverse effect, medicine.drug
Abstract

Introduction Rituximab (RTX) is an effective alternative treatment for severe idiopathic nephrotic syndrome in children. Nevertheless, the actual indication of RTX within the therapeutic strategy remains controversial, given the lack of data on the safety of this treatment in this setting. Objective To retrospectively collect and describe all severe and significant adverse effects that had occurred in children treated with RTX for idiopathic nephrotic syndrome. Patients and methods All participating centres within the SNP network were asked to fill an online database for all their patients treated with RTX for INS. Data were collected from the patients’ medical charts. Severe or significant adverse effects were defined as follow: any event that led to (i) a vital risk, (ii) a hospitalization or a prolongation of hospitalization, (iii) the occurrence of sequelae or (iiii) the interruption of RTX therapy. Results Twenty-three centres participated to the study. In total, 196 cases were collected on October 10th. The mean age at diagnosis was 4.7 (± 3.2) years. The mean duration of disease was 7.5 (± 4.3) years when RTX was first used. The mean follow-up was 3.2 (± 2.5) years and 1.5 (± 1.7) years since the first and the last infusion of RTX respectively. Adverse effects were reported in 6.6% of cases during the infusion (dyspnea = 6, cardiac arrhythmia = 1, hemodynamic instability = 1, others = 7), and in 13.1% of cases after the infusion (in relation with neutropenia = 6, low IgM = 3, low IgG = 6, infectious disorder = 14, cardiac = 2, liver = 1, kidney = 1, digestive = 2, neurological = 2, others = 6, several complications can be present in the same patient). The main consequences of these adverse effects were: non-planned hospitalizations (n = 30), modifications or interruptions of RTX treatment (n = 6), significant invalidity (n = 3). A single adverse effect led to a life-threatening situation. Conclusion Adverse effects were reported in 18.9% of these 196 patients treated by RTX for INS. RTX treatment was interrupted or modified in 6 cases. Only 4 adverse effects led to severe consequences (either life threatening event or significant invalidity). These results will help to evaluate the benefit/risk ratio or RTX for INS in children. This is an ongoing study, therefore results can be different when they will be presented during the meeting.

Published
2017

14. Pre-emptive kidney transplantation is associated with improved graft survival in children: Data from the French renal replacement therapy registry

Author

Sylvie Cloarec, F. Garaix, Olivier Dunand, Karen Leffondré, M. Reydit, Gwenaelle Roussey, Rémi Salomon, Etienne Bérard, A. Lahoche, Bruno Ranchin, Marc Fila, C. Couchoud, Stéphane Decramer, I. Vrillon, Tim Ulinski, Ariane Zaloszyc, Marie-Alice Macher, and Jérôme Harambat

Subjects
medicine.medical_specialty, Kidney, business.industry, medicine.medical_treatment, 030232 urology & nephrology, Disease, 030204 cardiovascular system & hematology, medicine.disease, Lower risk, Surgery, 03 medical and health sciences, 0302 clinical medicine, medicine.anatomical_structure, Pediatrics, Perinatology and Child Health, Medicine, Graft survival, Renal replacement therapy, business, Kidney transplantation, Dialysis, Paediatric patients
Abstract

Introduction Kidney transplantation (KT) is the treatment of choice for end-stage renal disease. Pre-emptive KT is considered to be the most optimal treatment of ESRD particularly in children but reports on the results of paediatric pre-emptive KT are scarce. The objective of this study was to evaluate the impact of pre-emptive KT on the risk of graft failure in children with ESRD. Methods We analyzed all first kidney transplants performed in children Results A total of 1920 paediatric patients were included, of whom 387 (20.2%) received a pre-emptive KT. Median time of follow-up was 7.0 years [IQR (3.0–11.7)]. At 10 years post-transplant, graft survival was 85.2% in pre-emptive KT and 67.1% in non-pre-emptive KT (P Conclusion In France, pre-emptive KT in children is associated with a lower risk of graft failure than KT performed after the initiation of dialysis, and should be encouraged when feasible.

Published
2017

15. Long-term outcome of children treated with rituximab for idiopathic nephrotic syndrome

Author

Stéphanie Tellier, Stéphane Decramer, Arnaud Garnier, Brigitte Llanas, Aurélia Bertholet-Thomas, Sylvie Nathanson, Flavio Bandin, Mathilde Cailliez, Vincent Guigonis, Christine Pietrement, Olivier Dunand, L. Ichay, and Karine Brochard

Subjects
Male, Nephrology, medicine.medical_specialty, Pediatrics, Nephrotic Syndrome, Time Factors, Idiopathic Nephrotic Syndrome, Antibodies, Monoclonal, Murine-Derived, immune system diseases, hemic and lymphatic diseases, Internal medicine, medicine, Humans, Immunologic Factors, Child, Retrospective Studies, business.industry, Infant, Treatment Outcome, Child, Preschool, Pediatrics, Perinatology and Child Health, Female, Rituximab, business, medicine.drug
Abstract

Rituximab (RTX) has recently showed promising results in the treatment of steroid-dependent idiopathic nephrotic syndrome (SDNS).This was a retrospective multicenter study of 18 children treated with RTX for SDNS, with a mean follow-up of 3.2 years. RTX was introduced because of side effects or relapses during therapy with immunosuppressive agents. The children received one to four infusions of RTX during the first course of treatment, and subsequent infusions were given due to CD19-cell recovery (CD19 1 %; 54 % of children) or relapse (41 %), as well as systematically (5 %).Treatment with RTX maintained sustained remission without relapse in 22 % of patients and increased the duration of remission in all other patients. The time between two successive relapses was 9 months in the absence of re-treatment and 24.5 months when infusions were performed at the time of CD19-cell recovery. At the last follow-up, 44.5 % of patients were free of oral drug therapy. Of those still receiving oral drugs, all doses had been decreased. No serious adverse events occurred.The results of this retrospective study confirm the efficacy and very good safety of RTX in the treatment of SDNS. The optimal therapeutic protocol seems to be a repeated single infusion at the time of CD19-cell recovery.

Published
2013

16. Pulmonary thrombosis as the first manifestation of systemic lupus erythematosus in a 14-year-old boy

Author

Fouad Madhi, Solenn Gaubicher, Olivier Dunand, Magali Colombat, Natascha Remus, Annie Harroche, and Christophe Delacourt

Subjects
Male, medicine.medical_specialty, Nephrotic Syndrome, Adolescent, Kidney Glomerulus, Lupus nephritis, Diagnosis, Differential, Membranous nephropathy, immune system diseases, Edema, medicine, Humans, skin and connective tissue diseases, Systemic lupus erythematosus, business.industry, medicine.disease, Lupus Nephritis, Dermatology, Pulmonary embolism, Treatment Outcome, Nephrology, Pediatrics, Perinatology and Child Health, Immunology, Radiography, Thoracic, Differential diagnosis, medicine.symptom, Pulmonary Embolism, business, Nephrotic syndrome, Anti-SSA/Ro autoantibodies
Abstract

We describe the case of a 14-year-old boy who presented with pulmonary embolism and who was subsequently found to have nephrotic syndrome due to lupus membranous nephropathy. He had no other signs of nephrotic syndrome or of systemic lupus erythematosus, such as edema or circulating lupus anticoagulants (antiphospholipid or anticardiolipin antibodies), and no hereditary coagulopathy. This case contributes to our understanding of unusual clinical presentations of systemic lupus erythematosus in children.

Published
2009

17. Maternal uniparental heterodisomy of chromosome 17 in a patient with nephropathic cystinosis

Author

Vincent Morinière, Anne-Sophie Lebre, Olivier Dunand, Nicole Morichon-Delvallez, Corinne Antignac, and Albert Bensman

Subjects
Proband, Genetics, Cystinosis, Haplotype, Uniparental Disomy, Biology, Uniparental Heterodisomy, medicine.disease, Article, Uniparental disomy, Pedigree, Chromosome 17 (human), Amino Acid Transport Systems, Neutral, Nephropathic Cystinosis, Child, Preschool, medicine, Humans, Female, Kidney Diseases, Allele, Genetics (clinical), Chromosomes, Human, Pair 17
Abstract

We report maternal uniparental disomy of chromosome 17 (mat UPD17) in a 2.5-year-old girl presenting infantile cystinosis. This patient was homozygous for the 57 kb deletion encompassing the CTNS gene, frequently found in patients from the European origin. The proband's mother was heterozygous for the deletion and the father did not carry the deletion. We carried out haplotype analysis with polymorphic markers spanning the whole chromosome 17. Informative markers showed the presence of two maternal alleles but no paternal allele for regions spanning the 17q arm and the proximal half of 17p, and only one maternal allele on the distal 17p arm. As deletion of half of 17p is probably not viable, these results suggest mat UPD17 with heterodisomy of 17q and proximal 17p and isodisomy of distal 17p. This is the first demonstration of mat UPD17, in particular of isodisomy 17p, in cystinosis.

Published
2009

18. Clinical characteristics and outcomes of childhood-onset ANCA-associated vasculitis: a French nationwide study

Author

Hubert Nivet, Olivia Boyer, Benoit Florkin, Marie Essig, Tim Ulinski, Jérôme Harambat, Christine Pietrement, T. Chambaraud, Emma Allain-Launay, Anne-Sylvia Sacri, Patrick Le Pogamp, Michel Fischbach, Helène See, Olivier Dunand, Eric Hachulla, Alexandre Belot, Rémi Salomon, Pierre Quartier, Bruno Ranchin, Stéphane Decramer, Hugues Flodrops, Georges Deschênes, François Nobili, Loïc Guillevin, and Jean-Louis Stephan

Subjects
Male, medicine.medical_specialty, Adolescent, Microscopic Polyangiitis, Renal function, Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis, Antibodies, Antineutrophil Cytoplasmic, Recurrence, Internal medicine, Ethnicity, medicine, Humans, Child, Retrospective Studies, Transplantation, Proportional hazards model, business.industry, Incidence, Mortality rate, Incidence (epidemiology), Prognosis, medicine.disease, Surgery, Survival Rate, Nephrology, Female, Kidney Diseases, France, Granulomatosis with polyangiitis, Microscopic polyangiitis, business, Vasculitis, Rheumatism, Glomerular Filtration Rate
Abstract

Background Data on anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis are scarce in children. The current study is aimed at describing the clinical features and outcomes of childhood-onset ANCA-associated vasculitis (AAV). Methods We conducted a retrospective French multicentre study involving patients in whom AAV was diagnosed before the age of 18 years. Inclusion criteria were (i) granulomatosis with polyangiitis (GPA) or microscopic polyangiitis (MPA) according to classification criteria of the European League Against Rheumatism/Paediatric Rheumatology European Society, and (ii) ANCA positivity. Patient and renal survival were analysed. Results Among 66 children included, 80% were female, 42% had GPA and 58% MPA including renal-limited vasculitis, 67% were pANCA+ and 33% cANCA+. The mean incidence of reported cases increased to 0.45 per million children/year in the period 2006-10. Median age at diagnosis was 11.5 years, and median time to diagnosis was 1 month. Initial symptoms included fever and fatigue (79%), skin lesions (41%), arthritis (42%), pulmonary (45%) and renal involvement (88%). Clinical features were similar between GPA and MPA with the exception of upper airway impairment (28%) specific of GPA. Ninety percent of the patients achieved remission after induction treatment. After a median follow-up of 5.2 years, 4 patients (6%) died, corresponding to a mortality rate of 1.2 per 100 person-years, and 22 patients (34%) developed end-stage renal disease (ESRD). Renal survival was 74, 70 and 59% at 1, 5 and 10 years, respectively. In a multivariable Cox regression model, baseline glomerular filtration rate, ethnic origin, histopathological classification and era of treatment were associated with the occurrence of ESRD. Relapse-free survival was 57% at 5 years and 34% at 10 years of follow-up. Patient and renal outcome did not significantly differ between GPA and MPA. Conclusion Childhood-onset AAV is a rare disease characterized by female predominance, delayed diagnosis, frequent renal impairment and a high remission rate. Baseline GFR and new histopathological classification system are strong predictors of ESRD. Renal survival in childhood AAV has improved over time.

Published
2015

19. Are there good reasons for inequalities in access to renal transplantation in children?

Author

Rémi Salomon, Tim Ulinski, Olivier Dunand, Benoît Audry, Arnaud Garnier, Jérôme Harambat, Cécile Couchoud, Marie-Alice Macher, and Julien Hogan

Subjects
Adult, Male, Pediatrics, medicine.medical_specialty, Inequality, Adolescent, Waiting Lists, media_common.quotation_subject, Descriptive survey, Patient characteristics, Logistic regression, Health Services Accessibility, CLINICAL SCIENCE, Second transplant, Young Adult, Residence Characteristics, Risk Factors, medicine, Humans, Registries, Healthcare Disparities, Child, media_common, Transplantation, business.industry, Patient Selection, Kidney Transplantation, Logistic Models, Socioeconomic Factors, Nephrology, Waiting list, Kidney Failure, Chronic, Female, National registry, France, business
Abstract

Background. Studies in the USA and Europe have demonstrated inequalities in adult access to renal transplants. We previously demonstrate that the centre of treatment was impacting the time to be registered on the renal waiting list. In this study, we sought to ascertain the influence of patient and centre characteristics on the probability of transplantation within 1 year after registration on the waiting list for children. Methods. We included patients

Published
2014

20. Efficacité du profil de conductivité sodique personnalisé dans le contrôle de l’équilibre tensionnel chez les enfants anéphriques hémodialysés

Author

V. Leroy, B. Beauval, and Olivier Dunand

Subjects
Pediatrics, Perinatology and Child Health
Abstract

L’hypertension chez les enfants anephriques en hemodialyse chronique est frequente, en depit des strategies mises en place pour estimer le poids de base. L’objectif de l’etude etait d’evaluer l’efficacite d’un profil de conductivite sodique personnalise sur l’equilibre tensionnel. Patients et methodes Cinq patients anephriques en dialyse chronique 6 jours/7 (âge median 6,3 ans) ont ete dialyses selon 2 modalites. Pendant 4 semaines (S-4–S-1), le debit d’ultrafiltration (UF) et la conductivite sodique (14,2 mS/cm, soit une concentration sodique du dialysat [DNa] de 140 mM) etaient constants. Pendant les 4 semaines suivantes (S1–S4), la prescription etait personnalisee : profil decroissant lineaire d’UF et de conductivite (en debut de seance, la DNa est egale a la natremie moyenne predialytique du patient et d’au minimum 140 mM ; en fin de seance, la DNa est de 136 mM). Les donnees cliniques et biologiques ont ete recueillies prospectivement. Pour chaque patient, nous avons compare les resultats observes en modalite « profil » (moyenne des valeurs de S3 et S4) a ceux observes en modalite « constant » (moyenne des valeurs de S-4 a S-1). Resultats Quatre patients recevaient un traitement a visee antihypertensive a S-4. A S4, le traitement anti-HTA avait ete diminue ou arrete chez tous, alors que le poids de base etait stable ou en augmentation ( Tableau 1 ). Le double profil a ete parfaitement tolere, sans augmentation du nombre d’hypotensions perdialytiques. Conclusion L’augmentation de la perte sodee diffusive perdialytique s’est accompagnee d’une amelioration de l’equilibre tensionnel chez les enfants anephriques, sans augmentation des episodes d’hypotension.

Published
2016

22. Commentary to 'Native nephrectomy in pediatric transplantation--less is more!'

Author

Jean-Luc Michel, Luke Harper, and Olivier Dunand

Subjects
Male, Kidney, medicine.medical_specialty, business.industry, Urology, medicine.medical_treatment, Autosomal dominant polycystic kidney disease, medicine.disease, Kidney Transplantation, Nephrectomy, Transplantation, medicine.anatomical_structure, Pediatrics, Perinatology and Child Health, medicine, Polycystic kidney disease, Humans, Female, Hemodialysis, Renal Insufficiency, Chronic, business, Kidney transplantation, Bilateral Nephrectomy
Abstract

[4] Fujisawa M, Iijima, Ishimura T, Higuchi A, Isotani S, Yoshiya K, et al. Long-term outcome of focal segmental glomerulosclerosis after Japanese pediatric transplantation. Pediatr Nephrol 2002;17:165e8. [5] Cavallini M, Di Zazza GD, Giordano U, Pongiglione G, Strologo LD, Capozza N, et al. Long-term cardiovascular effects of pre-transplant native kidney nephrectomy in children. Pediatr Nephrol 2010;25:2523e9. [6] Gundeti M, Taghizaedh A, Mushtaq I. Bilateral synchronous posterior prone retroperitoneoscopic nephrectomy with simultaneous peritoneal dialysis: a new management for end-stage renal disease in children. BJU Int 2007;99:904e6. [7] Ubara Y, Tagami T, Sawa N, Katori H, Yokota M, Takemoto F, et al. Renal contraction therapy for enlarged polycystic kidneys by transcatheter arterial embolization in hemodialysis patients. Am J Kidney Dis 2002;39:571e9. [8] Nunes P, Mota A, Figueiredo A, Parada B, Rolo F. Simultaneous renal transplantation and native nephrectomy in patients with autosomal-dominant polycystic kidney disease. Transplant Proc 2007;39:2483e5. [9] Glassman DT, Nipkow L, Bartlett ST, Jacobs SC. Bilateral nephrectomy with concomitant renal graft transplantation for autosomal dominant polycystic kidney disease. J Urol 2000;164:661e4. [10] Darby CR, Cranston D, Raine AEG, Morris PJ. Bilateral nephrectomy before transplantation: indications, surgical morbidity and mortality. Br J Surg 1991;78:305e7. [11] Chaudhuri A, Salvatierra Jr O, Alexander SR, Sarwal MM. Option of pre-emptive nephrectomy and renal transplantation for Bartter’s syndrome. Pediatr Transplant 2006;10:266e70. [12] Odorico JS, Knechtle SJ, Rayhill SC, Pirsch JD, D’Alessandro AM, Belzer FO, et al. The influence of native

Published
2012

23. Papillary stones with Randall's plaques in children: clinicobiological features and outcome

Author

Olivia Boyer, François Nobili, Gandon Laloum Sylvie, Karim Bouchireb, Michel Daudon, Hubert Nivet, Rémi Salomon, Hélène Martelli, Olivier Dunand, Niaudet P, and Christine Pietrement

Subjects
Male, medicine.medical_specialty, Adolescent, MEDLINE, Kidney Calculi, X ray computed, medicine, Humans, Disease management (health), Child, Retrospective Studies, Transplantation, Kidney Medulla, Calcium Oxalate, business.industry, Follow up studies, Infant, Newborn, Disease Management, Infant, Retrospective cohort study, Prognosis, Multicenter study, Nephrology, Child, Preschool, Female, Radiology, business, Tomography, X-Ray Computed, Follow-Up Studies
Published
2011

24. Therapy-resistant anaemia in congenital nephrotic syndrome of the Finnish type--implication of EPO, transferrin and transcobalamin losses

Author

Julie Toubiana, Tim Ulinski, Renata Vitkevic, Marie-Hélène Schlageter, Olivier Dunand, Albert Bensman, and Bilal Aoun

Subjects
medicine.medical_specialty, Nephrotic Syndrome, Anemia, Nephrectomy, Transcobalamin, Internal medicine, Medicine, Humans, Blood Transfusion, Congenital nephrotic syndrome, Erythropoietin, chemistry.chemical_classification, Transplantation, Transcobalamins, business.industry, Infant, Newborn, Transferrin, Glomerulonephritis, medicine.disease, Endocrinology, chemistry, Nephrology, Hematinics, Erythropoiesis, Female, business, Nephrotic syndrome, medicine.drug
Abstract

Congenital nephrotic syndrome of the Finnish type (CNF) is due to NPHS1 mutation and is responsible for a variety of urinary protein losses. We report the case of a 4-month-old girl with a particularly severe form (proteinuria approximately 150 g/l) of CNF. She developed severe non-regenerative anaemia requiring bi-monthly blood transfusions despite daily EPO (600 UI/kg) and iron supplementation. Epoetin pharmacokinetics revealed a urinary loss of 27% of the given dose within the first 24 h after IV injection. However, plasma levels remained increased after 24 h (228 UI/l). Plasma transferrin and transcobalamin levels were undetectable. Atransferrinaemia and atranscobalaminaemia seem to be responsible for disturbed erythropoiesis.

Published
2009

25. Urinary Tract Infections

Author

Tim Ulinski, Olivier Dunand, and Albert Bensman

Subjects
medicine.medical_specialty, business.industry, Urinary system, Urology, Medicine, business
Published
2009

26. Ocular involvement in hemolytic uremic syndrome due to factor H deficiency--are there therapeutic consequences?

Author

Sandrine Leroy, Olivier Dunand, Marta Montchilova, Georges Deschênes, Albert Bensman, Anis Larakeb, Véronique Frémeaux-Bacchi, Béatrice Pelosse, and Tim Ulinski

Subjects
Nephrology, Choroid Hemorrhage, Male, medicine.medical_specialty, Pathology, Thrombotic microangiopathy, genetic structures, medicine.medical_treatment, Complement Pathway, Alternative, Gastroenterology, Blurred vision, Internal medicine, medicine, Humans, medicine.diagnostic_test, biology, business.industry, Haptoglobin, Homozygote, Infant, Glaucoma, Thrombosis, Fluorescein angiography, medicine.disease, eye diseases, Factor H, Complement Factor H, Pediatrics, Perinatology and Child Health, Hemolytic-Uremic Syndrome, biology.protein, Hemodialysis, Fresh frozen plasma, medicine.symptom, business
Abstract

Factor H deficiency is responsible for thrombotic microangiopathy (TMA) via uncontrolled activation of the alternative pathway of the complement system. Ocular TMA has never been reported in patients with factor H abnormalities. A male patient with congenital homozygote factor H deficiency reached end-stage renal disease at the age of 10 years. Hemodialysis was uneventful for 3 years, when, suddenly, unilateral ocular pain and blurred vision occurred while he had febrile pharyngitis. Ophthalmologic examination found vitreous bleeding, elevated ocular pressure, choroidal hemorrhage (ultrasound biomicroscopy) and retinal ischemia (fluorescein angiography). C-reactive protein concentration was increased, while haptoglobin levels remained normal. We suspected that TMA due to factor H deficiency was responsible for the ocular manifestations and immediately initiated daily plasma exchanges (PEs) with fresh frozen plasma (FFP) for 10 days followed by three sessions per week. Factor H serum level increased from 6% to 82%, and C3 level normalized. Progressively, ocular pain decreased, and visual acuity and ophthalmologic findings showed improvement. When there is permanent activation of the alternative pathway in patients with end-stage renal disease (ESRD), the search for secondary targets might be of interest. In nephrectomized patients, no biological parameter can predict isolated ocular TMA. Early ophthalmologic investigation and substitution of factor H via FFP may avoid irreversible damage.

Published
2007

27. CO-58 – Syndrome néphrotique congénital: une étude multicentrique française

Author

Olivia Boyer, G. Rousset, Sophie Taque, S. Pierrepont, Véronique Baudouin, L de Parscau, Adrien May, Marc Fila, Caroline Rousset-Rouvière, B. Boudaillez, Hugues Flodrops, Tim Ulinski, R. Vieux, Maud Dehennault, Corinne Antignac, Laurence Heidet, Ferielle Louillet, Olivier Dunand, Ariane Zaloszyc, Patrick Niaudet, Christine Pietrement, J. Baccheta, Rémi Salomon, Sandra Bérody, Elodie Merieau, Arnaud Garnier, J. Tenenbaum, and Jérôme Harambat

Subjects
Pediatrics, Perinatology and Child Health
Published
2015

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