Long-term health-related quality of life outcomes of adults with pediatric onset of frequently relapsing or steroid-dependent nephrotic syndrome
Marie-Sophie Meuleman
(1, 2)
,
Sophie Guilmin-Crépon
(3, 4)
,
Aurélie Hummel
(5)
,
Eric Daugas
(6, 7)
,
Agnès Dumas
(4)
,
Fallou Leye
(3, 8)
,
Jacques Dantal
(9)
,
Claire Rigothier
(10)
,
François Provot
(11)
,
Dominique Chauveau
(12)
,
Stéphane Burtey
(13, 14)
,
Alexandre Hertig
(15)
,
Karine Dahan
(15)
,
Antoine Durrbach
(1)
,
Claire Dossier
(8)
,
Alexandre Karras
(16)
,
Dominique Guerrot
(17)
,
Vincent Esnault
(18)
,
Philippe Rémy
(1, 2)
,
Ziad Massy
(19, 20)
,
Isabelle Tostivint
(21)
,
Marie-Pascale Morin
(22)
,
Philippe Zaoui
(23)
,
Olivier Fritz
(24)
,
Moglie Le Quintrec
(25, 26)
,
Alain Wynckel
(27)
,
Aurélie Bourmaud
(3, 4)
,
Olivia Boyer
(28)
,
Dil Sahali
(1, 2)
,
Corinne Alberti
(3, 4)
,
Vincent Audard
(1, 2)
,
Hélène Mellerio
(4, 8)
1
CHU Henri Mondor [Créteil]
2 IMRB - Institut Mondor de Recherche Biomédicale
3 CIC 1426 - Centre d'Investigation Clinique 1426
4 ECEVE (U1123 / UMR_S_1123) - Epidémiologie Clinique et Evaluation Economique Appliquées aux Populations Vulnérables
5 Département de Néphrologie [CHU Necker]
6 AP-HP - Hôpital Bichat - Claude Bernard [Paris]
7 CRI (UMR_S_1149 / ERL_8252 / U1149) - Centre de recherche sur l'Inflammation
8 AP-HP Hôpital universitaire Robert-Debré [Paris]
9 CHU Nantes - Centre Hospitalier Universitaire de Nantes
10 CHU Bordeaux
11 CHRU Lille - Centre Hospitalier Régional Universitaire [CHU Lille]
12 CHU Toulouse - Centre Hospitalier Universitaire de Toulouse
13 C2VN - Centre recherche en CardioVasculaire et Nutrition = Center for CardioVascular and Nutrition research
14 LA CONCEPTION - Hôpital de la Conception [CHU - APHM]
15 CHU Tenon [AP-HP]
16 HEGP - Hôpital Européen Georges Pompidou [APHP]
17 Service de Néphrologie [Rouen]
18 CHU Nice - Centre Hospitalier Universitaire de Nice
19 Hôpital Ambroise Paré [AP-HP]
20 CESP - Centre de recherche en épidémiologie et santé des populations
21 CHU Pitié-Salpêtrière [AP-HP]
22 Centre Hospitalier Universitaire de Rennes [CHU Rennes] = Rennes University Hospital [Ponchaillou]
23 CHUGA - Centre Hospitalier Universitaire [CHU Grenoble]
24 Centre Hospitalier de La Rochelle (CHR)
25 CHRU Montpellier - Centre Hospitalier Régional Universitaire [Montpellier]
26 Cellules Souches, Plasticité Cellulaire, Médecine Régénératrice et Immunothérapies (IRMB)
27 Hôpital universitaire Robert Debré [Reims]
28 Hôpital Necker - Enfants Malades [AP-HP]
2 IMRB - Institut Mondor de Recherche Biomédicale
3 CIC 1426 - Centre d'Investigation Clinique 1426
4 ECEVE (U1123 / UMR_S_1123) - Epidémiologie Clinique et Evaluation Economique Appliquées aux Populations Vulnérables
5 Département de Néphrologie [CHU Necker]
6 AP-HP - Hôpital Bichat - Claude Bernard [Paris]
7 CRI (UMR_S_1149 / ERL_8252 / U1149) - Centre de recherche sur l'Inflammation
8 AP-HP Hôpital universitaire Robert-Debré [Paris]
9 CHU Nantes - Centre Hospitalier Universitaire de Nantes
10 CHU Bordeaux
11 CHRU Lille - Centre Hospitalier Régional Universitaire [CHU Lille]
12 CHU Toulouse - Centre Hospitalier Universitaire de Toulouse
13 C2VN - Centre recherche en CardioVasculaire et Nutrition = Center for CardioVascular and Nutrition research
14 LA CONCEPTION - Hôpital de la Conception [CHU - APHM]
15 CHU Tenon [AP-HP]
16 HEGP - Hôpital Européen Georges Pompidou [APHP]
17 Service de Néphrologie [Rouen]
18 CHU Nice - Centre Hospitalier Universitaire de Nice
19 Hôpital Ambroise Paré [AP-HP]
20 CESP - Centre de recherche en épidémiologie et santé des populations
21 CHU Pitié-Salpêtrière [AP-HP]
22 Centre Hospitalier Universitaire de Rennes [CHU Rennes] = Rennes University Hospital [Ponchaillou]
23 CHUGA - Centre Hospitalier Universitaire [CHU Grenoble]
24 Centre Hospitalier de La Rochelle (CHR)
25 CHRU Montpellier - Centre Hospitalier Régional Universitaire [Montpellier]
26 Cellules Souches, Plasticité Cellulaire, Médecine Régénératrice et Immunothérapies (IRMB)
27 Hôpital universitaire Robert Debré [Reims]
28 Hôpital Necker - Enfants Malades [AP-HP]
Stéphane Burtey
- Fonction : Auteur
- PersonId : 807084
- ORCID : 0000-0003-1813-7884
Alexandre Hertig
- Fonction : Auteur
- PersonId : 771905
- ORCID : 0000-0002-2903-3303
- IdRef : 112040470
Antoine Durrbach
- Fonction : Auteur
- PersonId : 768839
- ORCID : 0000-0003-0385-8726
- IdRef : 061677949
Alexandre Karras
- Fonction : Auteur
- PersonId : 762481
- ORCID : 0000-0002-3075-7739
- IdRef : 077765036
Ziad Massy
- Fonction : Auteur
- PersonId : 1003368
- ORCID : 0000-0001-5771-5996
Olivia Boyer
- Fonction : Auteur
- PersonId : 889775
- ORCID : 0000-0002-3957-1359
- IdRef : 11556134X
Corinne Alberti
- Fonction : Auteur
- PersonId : 9314
- IdHAL : corinne-alberti
- ORCID : 0000-0002-9336-1395
- IdRef : 078801877
Vincent Audard
- Fonction : Auteur
Hélène Mellerio
- Fonction : Auteur
- PersonId : 823620
- ORCID : 0000-0001-6586-8677
Résumé
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.