Vincent Audard1,2, Hélène Mellerio3,4,5, Marie-Sophie Meuleman6,7, Sophie Guilmin-Crépon3,8, Aurélie Hummel9, Eric Daugas10, Agnès Dumas3, Fallou Leye8, Jacques Dantal11, Claire Rigothier12, François Provot13, Dominique Chauveau14, Stéphane Burtey15, Alexandre Hertig16, Karine Dahan17, Antoine Durrbach1, Claire Dossier18, Alexandre Karras19, Dominique Guerrot20, Vincent Esnault21, Philippe Rémy1,2, Ziad A Massy22,23, Isabelle Tostivint24, Marie-Pascale Morin25, Philippe Zaoui26, Olivier Fritz27, Moglie Le Quintrec28, Alain Wynckel29, Aurélie Bourmaud3,8, Olivia Boyer30, Dil Sahali1,2, Corinne Alberti3,8. 1. Service de Néphrologie et Transplantation, Centre de Référence Maladie Rare "Syndrome Néphrotique Idiopathique", Assistance Publique des Hôpitaux de Paris (AP-HP), Hôpitaux Universitaires Henri Mondor, Fédération Hospitalo-Universitaire "Innovative Therapy for Immune Disorders", 51 Avenue du Marechal-de-Lattre-de-Tassigny, Créteil Cedex, 94010, Creteil, France. 2. Institut National de la Santé et de la Recherche Médicale (INSERM) U955, Institut Mondor de Recherche Biomédicale (IMRB), Université Paris Est Créteil, Equipe 21, Creteil, France. 3. Université de Paris, ECEVE UMR 1123, INSERM, 75010, Paris, France. 4. Service de médecine d'adolescent, Plateforme de Transition AD'venir, AP-HP, Hôpital Robert Debré, Paris, France. 5. Groupe Français de Recherche en Médecine et Santé de l'Adolescent, Paris, France. 6. Service de Néphrologie et Transplantation, Centre de Référence Maladie Rare "Syndrome Néphrotique Idiopathique", Assistance Publique des Hôpitaux de Paris (AP-HP), Hôpitaux Universitaires Henri Mondor, Fédération Hospitalo-Universitaire "Innovative Therapy for Immune Disorders", 51 Avenue du Marechal-de-Lattre-de-Tassigny, Créteil Cedex, 94010, Creteil, France. marie.meuleman@aphp.fr. 7. Institut National de la Santé et de la Recherche Médicale (INSERM) U955, Institut Mondor de Recherche Biomédicale (IMRB), Université Paris Est Créteil, Equipe 21, Creteil, France. marie.meuleman@aphp.fr. 8. Unité d'Épidémiologie Clinique, CIC 1426, AP-HP.Nord, Hôpital Universitaire Robert Debré, Inserm, 75019, Paris, France. 9. Service de Néphrologie, AP-HP, Hôpital Necker, Paris, France. 10. Service de Néphrologie, AP-HP, Hôpital Bichat, Université de Paris, INSERM U1149, Paris, France. 11. Service de Néphrologie Immunologie Clinique Transplantation, Centre de Recherche en Transplantation et Immunologie (CRTI), Centre Hospitalier Universitaire (CHU) de Nantes, Nantes, France. 12. Service de Néphrologie Transplantation, Dialyse et Aphérèses, CHU de Bordeaux, Bordeaux, France. 13. Service de Néphrologie, Hôpital Huriez, CHU de Lille, Lille, France. 14. Service de Néphrologie et Transplantation d'Organes, Hôpital de Rangueil et Centre de Référence Maladies Rénales Rares, CHU de Toulouse, Toulouse, France. 15. APHM, INSERM, INRAe, C2VN, Centre de Néphrologie et Transplantation Rénale, Hôpital de la Conception, Aix Marseille University, Marseille, France. 16. Service de Néphrologie et Transplantation Rénale, AP-HP, Hôpital Tenon, Paris, France. 17. Service de Néphrologie et Dialyse, AP-HP, Hôpital Tenon, Paris, France. 18. Service de Néphrologie Pédiatrique, AP-HP, Hôpital Robert Debré, Paris, France. 19. Service de Néphrologie, AP-HP, Hôpital Européen Georges Pompidou, Université de Paris, Paris, France. 20. Service de Néphrologie, CHU de Rouen, Rouen, France. 21. Service de Néphrologie, Hôpital Pasteur, CHU de Nice, Nice, France. 22. Service de Néphrologie, AP-HP, Hôpital Ambroise Paré, Boulogne-Billancourt, France. 23. INSERM U1018 CESP, UVSQ, UPS Villejuif, Villejuif, France. 24. Service de Néphrologie et Transplantation, AP-HP, Hôpital Pitié Salpêtrière, Paris, France. 25. Service de Néphrologie, Hôpital de Pontchaillou, CHU de Rennes, Rennes, France. 26. Service de Néphrologie, Hémodialyse, Aphérèse et Transplantation Rénale, CHU de Grenoble Alpes, Université Grenoble-Alpes, Grenoble, France. 27. Service de Néphrologie, Centre Hospitalier (CH) La Rochelle, La Rochelle, France. 28. Service de Néphrologie Dialyse et Transplantation Rénale, Hôpital Lapeyronie, CHU de Montpellier, Montpellier, France. 29. Service de Néphrologie, CHU de Reims, Reims, France. 30. Service de Néphrologie Pédiatrique, Centre de Référence Maladie Rare "Syndrome Néphrotique Idiopathique", Institut Imagine, AP-HP, Hôpital Necker-Enfants Malades, Université de Paris, Paris, France.
Abstract
BACKGROUND: Long-term psychosocial outcomes and health-related quality of life (HRQOL) in adults with pediatric onset of frequently relapsing or steroid-dependent idiopathic nephrotic syndrome (FRNS or SDNS) remain to be determined. METHODS: In this prospective cohort study, 59 adults with pediatric onset of FRNS/SDNS and persistent active glomerular disease in adulthood completed the GEDEPAC-2 questionnaire exploring 11 well-being domains. Data were compared to the French general population (FGP) with standardized incidence ratio ([SIR]; adjusted for period, age, gender). Regression models were performed to identify predictive factors of psychosocial well-being. RESULTS: In 82% of cases, the questionnaire was completed while the participants (n = 59; 47 men; median age = 32 years; median number of relapses = 13) were in complete remission (under specific therapy in 76% of cases). Participants had higher educational degree than in the FGP (SIR = 6.3; p < 0.01) and more frequently a managerial occupation (SIR = 3.1; p < 0.01). Social integration was acceptable with regard to marital status and experience of sexual intercourse, but experiences of discrimination were far more frequent (SIR = 12.5; p < 0.01). The SF-12 mental component summary (MCS) score was altered (Z-score = - 0.6; p < 0.01) and mean multidimensional fatigue inventory (MFI-20) global fatigue score appeared high (12). Transfer from pediatric to adult healthcare was followed by a period of discontinued care for 33% of participants. Multivariate analysis revealed a close relationship between MFI-20, physical health, and MCS. CONCLUSIONS: This study shows that pediatric onset FRNS and SDNS may have a long-term negative impact on mental HRQOL and highlights the impact of fatigue, which is often not adequately considered in routine care.
BACKGROUND: Long-term psychosocial outcomes and health-related quality of life (HRQOL) in adults with pediatric onset of frequently relapsing or steroid-dependent idiopathic nephrotic syndrome (FRNS or SDNS) remain to be determined. METHODS: In this prospective cohort study, 59 adults with pediatric onset of FRNS/SDNS and persistent active glomerular disease in adulthood completed the GEDEPAC-2 questionnaire exploring 11 well-being domains. Data were compared to the French general population (FGP) with standardized incidence ratio ([SIR]; adjusted for period, age, gender). Regression models were performed to identify predictive factors of psychosocial well-being. RESULTS: In 82% of cases, the questionnaire was completed while the participants (n = 59; 47 men; median age = 32 years; median number of relapses = 13) were in complete remission (under specific therapy in 76% of cases). Participants had higher educational degree than in the FGP (SIR = 6.3; p < 0.01) and more frequently a managerial occupation (SIR = 3.1; p < 0.01). Social integration was acceptable with regard to marital status and experience of sexual intercourse, but experiences of discrimination were far more frequent (SIR = 12.5; p < 0.01). The SF-12 mental component summary (MCS) score was altered (Z-score = - 0.6; p < 0.01) and mean multidimensional fatigue inventory (MFI-20) global fatigue score appeared high (12). Transfer from pediatric to adult healthcare was followed by a period of discontinued care for 33% of participants. Multivariate analysis revealed a close relationship between MFI-20, physical health, and MCS. CONCLUSIONS: This study shows that pediatric onset FRNS and SDNS may have a long-term negative impact on mental HRQOL and highlights the impact of fatigue, which is often not adequately considered in routine care.
Authors: David T Selewski; Jonathan P Troost; Susan F Massengill; Rasheed A Gbadegesin; Larry A Greenbaum; Ibrahim F Shatat; Yi Cai; Gaurav Kapur; Diane Hebert; Michael J Somers; Howard Trachtman; Priya Pais; Michael E Seifert; Jens Goebel; Christine B Sethna; John D Mahan; Heather E Gross; Emily Herreshoff; Yang Liu; Peter X Song; Bryce B Reeve; Darren A DeWalt; Debbie S Gipson Journal: Pediatr Nephrol Date: 2015-03-18 Impact factor: 3.714
Authors: Brad H Rovin; Dawn J Caster; Daniel C Cattran; Keisha L Gibson; Jonathan J Hogan; Marcus J Moeller; Dario Roccatello; Michael Cheung; David C Wheeler; Wolfgang C Winkelmayer; Jürgen Floege Journal: Kidney Int Date: 2019-02 Impact factor: 10.612
Authors: Debbie S Gipson; David T Selewski; Susan F Massengill; Larysa Wickman; Kassandra L Messer; Emily Herreshoff; Corinna Bowers; Maria E Ferris; John D Mahan; Larry A Greenbaum; Jackie MacHardy; Gaurav Kapur; Deepa H Chand; Jens Goebel; Gina Marie Barletta; Denis Geary; David B Kershaw; Cynthia G Pan; Rasheed Gbadegesin; Guillermo Hidalgo; Jerome C Lane; Jeffrey D Leiser; Brett W Plattner; Peter X Song; David Thissen; Yang Liu; Heather E Gross; Darren A DeWalt Journal: Health Qual Life Outcomes Date: 2013-03-04 Impact factor: 3.186