Rituximab and improved nodular regenerative hyperplasia-associated non-cirrhotic liver disease in common variable immunodeficiency: a case report and literature study.
common variable immune deficiency (CVID)
inborn errors of immunity
nodular regenerative hyperplasia
non-cirrhotic portal hypertension
non-infectious complications
primary immunodeficiency
rituximab
Journal
Frontiers in immunology
ISSN: 1664-3224
Titre abrégé: Front Immunol
Pays: Switzerland
ID NLM: 101560960
Informations de publication
Date de publication:
2023
2023
Historique:
received:
20
07
2023
accepted:
29
08
2023
medline:
1
11
2023
pubmed:
5
10
2023
entrez:
5
10
2023
Statut:
epublish
Résumé
Common variable immunodeficiency (CVID) associated liver disease is an underrecognized and poorly studied non-infectious complication that lacks an established treatment. We describe a CVID patient with severe multiorgan complications, including non-cirrhotic portal hypertension secondary to nodular regenerative hyperplasia leading to diuretic-refractory ascites. Remarkably, treatment with rituximab, administered for concomitant immune thrombocytopenia, resulted in the complete and sustained resolution of portal hypertension and ascites. Our case, complemented with a literature review, suggests a beneficial effect of rituximab that warrants further research.
Identifiants
pubmed: 37795099
doi: 10.3389/fimmu.2023.1264482
pmc: PMC10546204
doi:
Substances chimiques
Rituximab
4F4X42SYQ6
Types de publication
Review
Case Reports
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
1264482Informations de copyright
Copyright © 2023 Roosens, Staels, Van Loo, Humblet-Baron, Meyts, De Samblanx, Verslype, van Malenstein, van der Merwe, Laleman and Schrijvers.
Déclaration de conflit d'intérêts
The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. The author(s) declared that they were an editorial board member of Frontiers, at the time of submission. This had no impact on the peer review process and the final decision.
Références
Expert Rev Clin Immunol. 2023 Jun;19(6):627-638
pubmed: 36996348
Front Immunol. 2020 Feb 28;11:338
pubmed: 32184784
J Allergy Clin Immunol Pract. 2019 Sep - Oct;7(7):2484-2486.e3
pubmed: 31026542
J Clin Immunol. 2013 Jan;33(1):84-95
pubmed: 22986767
Front Immunol. 2020 Feb 07;11:149
pubmed: 32117289
Eur Ann Allergy Clin Immunol. 2022 Mar 18;:
pubmed: 35301845
BMJ Case Rep. 2016 Jun 22;2016:
pubmed: 27335365
Epidemiol Mikrobiol Imunol. 2018 Winter;67(3):142-148
pubmed: 30602282
Clin Mol Allergy. 2019 May 6;17:9
pubmed: 31080365