Rituximab-associated hypogammaglobulinemia in children with idiopathic nephrotic syndrome: results of an ESPN survey.


Journal

Pediatric nephrology (Berlin, Germany)
ISSN: 1432-198X
Titre abrégé: Pediatr Nephrol
Pays: Germany
ID NLM: 8708728

Informations de publication

Date de publication:
09 2023
Historique:
received: 05 10 2022
accepted: 08 02 2023
revised: 13 01 2023
medline: 17 8 2023
pubmed: 5 4 2023
entrez: 4 4 2023
Statut: ppublish

Résumé

There is paucity of information on rituximab-associated hypogammaglobulinemia (HGG) and its potential infectious consequences in children treated for idiopathic nephrotic syndrome (INS). A survey was distributed by the European Society Pediatric Nephrology to its members. It addressed the screening and management practices of pediatric nephrology units for recognizing and treating RTX-associated HGG and its morbidity and mortality. Eighty-four centers which had treated an overall 1328 INS children with RTX responded. The majority of centers administered several courses of RTX and continued concomitant immunosuppressive therapy. Sixty-five percent of centers routinely screened children for HGG prior to RTX infusion, 59% during, and 52% following RTX treatment. Forty-seven percent had observed HGG prior to RTX administration, 61% during and 47% >9 months following treatment in 121, 210, and 128 subjects respectively. Thirty-three severe infections were reported among the cohort of 1328 RTX-treated subjects, of whom 3 children died. HGG had been recognized in 30/33 (80%) of them. HGG in steroid-dependent/frequently relapsing nephrotic syndrome (SDNS/FRNS) children is probably multifactorial and can be observed prior to RTX administration in children with SDNS/FRNS. Persistent HGG lasting >9 months from RTX infusion is not uncommon and may increase the risk of severe infections in this cohort. We advocate for the obligatory screening for HGG in children with SDNS/FRNS prior to, during, and following RTX treatment. Further research is necessary to identify risk factors for developing both HGG and severe infections before recommendations are made for its optimal management. A higher resolution version of the Graphical abstract is available as Supplementary information.

Sections du résumé

BACKGROUND
There is paucity of information on rituximab-associated hypogammaglobulinemia (HGG) and its potential infectious consequences in children treated for idiopathic nephrotic syndrome (INS).
METHODS
A survey was distributed by the European Society Pediatric Nephrology to its members. It addressed the screening and management practices of pediatric nephrology units for recognizing and treating RTX-associated HGG and its morbidity and mortality. Eighty-four centers which had treated an overall 1328 INS children with RTX responded.
RESULTS
The majority of centers administered several courses of RTX and continued concomitant immunosuppressive therapy. Sixty-five percent of centers routinely screened children for HGG prior to RTX infusion, 59% during, and 52% following RTX treatment. Forty-seven percent had observed HGG prior to RTX administration, 61% during and 47% >9 months following treatment in 121, 210, and 128 subjects respectively. Thirty-three severe infections were reported among the cohort of 1328 RTX-treated subjects, of whom 3 children died. HGG had been recognized in 30/33 (80%) of them.
CONCLUSIONS
HGG in steroid-dependent/frequently relapsing nephrotic syndrome (SDNS/FRNS) children is probably multifactorial and can be observed prior to RTX administration in children with SDNS/FRNS. Persistent HGG lasting >9 months from RTX infusion is not uncommon and may increase the risk of severe infections in this cohort. We advocate for the obligatory screening for HGG in children with SDNS/FRNS prior to, during, and following RTX treatment. Further research is necessary to identify risk factors for developing both HGG and severe infections before recommendations are made for its optimal management. A higher resolution version of the Graphical abstract is available as Supplementary information.

Identifiants

pubmed: 37014530
doi: 10.1007/s00467-023-05913-1
pii: 10.1007/s00467-023-05913-1
pmc: PMC10432325
doi:

Substances chimiques

Rituximab 4F4X42SYQ6
Immunosuppressive Agents 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

3035-3042

Investigateurs

H Alpay (H)
R Andersen (R)
G Ariceta (G)
B Atmış (B)
U S Bayrakçı (US)
B Esrea (B)
V Baudouin (V)
N Bervina (N)
E Benetti (E)
E Berard (E)
A Bjerre (A)
M Christian (M)
A Couderc (A)
J Dehoorne (J)
G Deschenes (G)
C Dossier (C)
R Düşünsel (R)
Z Ekinci (Z)
F Emma (F)
L Espinoza (L)
A Gianviti (A)
M Herrero Goñi (M)
G Guido (G)
L Ghio (L)
J Groothoff (J)
D Ö Hacıhamdioğlu (DÖ)
M Espino Hernández (M)
A Jankauskiene (A)
M Kagan (M)
M Kemper (M)
M Kovacevic (M)
S Kohl (S)
R T Kramar (RT)
M López-González (M)
A M Aris (AM)
S Maringhini (S)
M Marlais (M)
M Melgosa (M)
A Mitsioni (A)
G Montini (G)
A Moczulska (A)
L Murer (L)
V Obukhova (V)
J Oh (J)
P Ortega (P)
Z B Özçakar (ZB)
T Ulinski (T)
A Pasini (A)
T Papalia (T)
S Paunova (S)
A Pena (A)
C Pecoraro (C)
E Petrosyan (E)
L Peruzzi (L)
N Printza (N)
L Prikhodina (L)
C Pietrement (C)
S Rittig (S)
D Rodrigo (D)
N Savenkova (N)
M Saraga (M)
F L Sever (FL)
M Schreuder (M)
M Szczepanska (M)
V Tasic (V)
B Tonshoff (B)
K Tullus (K)
J Vara (J)
J Vande Walle (J)
E Volokhina (E)
J Zieg (J)
A Waters (A)
L T Weber (LT)
N Webbe (N)
M Wasiak (M)
K Endén (K)
A Tsygin (A)
R Grenda (R)
A Raes (A)
J M Van Hoeck Koen (JM)
B Adams (B)
M Aksenova (M)
B Ranchin (B)
N Hooman (N)
I Ogarek (I)
T Seeman (T)
M Fila (M)
L Oni (L)
S Mir (S)
R Novo (R)
S Stabouli (S)
J Vara-Martín (J)
J A E van Wijk (JAE)
R Ehren (R)
Z Bekassy (Z)
M Herthelius (M)
F Becherucci (F)
H Shasha-Lavsky (H)
F Santos (F)
M Feldkötter (M)
M Pańczyk-Tomaszewska (M)
J Harambat (J)
D Grima (D)
I Gökçe (I)
A Teixeira (A)
C Licht (C)
I M Schmidt (IM)
P Brandström (P)
N Dinçel (N)

Informations de copyright

© 2023. The Author(s).

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Auteurs

Aleksandra Zurowska (A)

Department of Pediatrics, Nephrology and Hypertension, Medical University of Gdańsk, ul. Debinki 7, 80-952, Gdańsk, Poland. aleksandra.zurowska@gumed.edu.pl.
Centre for Rare Diseases, Medical University of Gdańsk, Gdańsk, Poland. aleksandra.zurowska@gumed.edu.pl.

Magdalena Drozynska-Duklas (M)

Department of Pediatrics, Nephrology and Hypertension, Medical University of Gdańsk, ul. Debinki 7, 80-952, Gdańsk, Poland.

Rezan Topaloglu (R)

Department of Pediatric Nephrology, Hacettepe University School of Medicine Hacettepe University, Ankara, Turkey.

Antonia Bouts (A)

Department of Pediatric Nephrology, Emma Children's Hospital, Amsterdam University Medical Centers, Amsterdam, The Netherlands.

Olivia Boyer (O)

Department of Pediatric Nephrology, Reference Center for Idiopathic Nephrotic Syndrome in Children and Adults, Necker Hospital, Paris, France.
Laboratory of Hereditary Kidney Diseases, Imagine Institute, Paris Descartes University, Paris, France.

Mohan Shenoy (M)

Department of Paediatric Nephrology, Royal Manchester Children's Hospital, Manchester University Hospitals NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, UK.

Marina Vivarelli (M)

Division of Nephrology and Dialysis, Bambino Gesù Children's Hospital and Research Institute, Rome, Italy.

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