Tocilizumab for massive refractory pleural effusion in an adolescent with systemic lupus erythematosus.


Journal

Pediatric rheumatology online journal
ISSN: 1546-0096
Titre abrégé: Pediatr Rheumatol Online J
Pays: England
ID NLM: 101248897

Informations de publication

Date de publication:
16 Sep 2021
Historique:
received: 24 06 2021
accepted: 30 08 2021
entrez: 17 9 2021
pubmed: 18 9 2021
medline: 11 2 2022
Statut: epublish

Résumé

Pleural effusion in systemic lupus erythematous (SLE) is a common symptom, and recent studies demonstrated that IL-6 has a pivotal role in its pathogenesis. We report a case of a 15 years old Caucasian boy with a history of persistent pleural effusion without lung involvement or fever. Microbiological and neoplastic aetiologies were previously excluded. Based on the presence of pleuritis, malar rash, reduction of C3 and C4 levels and positivity of antinuclear antibody (ANA) and anti-double stranded DNA (dsDNA), the diagnosis of juvenile SLE (JSLE) was performed. Treatment with high dose of intravenous glucocorticoids and mycophenolate mofetil was started with partial improvement of pleural effusion. Based on this and on adults SLE cases with serositis previously reported, therapy with intravenous tocilizumab (800 mg every two weeks) was started with prompt recovery of pleural effusion. To the best of our knowledge, this is the first case of JSLE pleuritis successfully treated with tocilizumab.

Sections du résumé

BACKGROUND BACKGROUND
Pleural effusion in systemic lupus erythematous (SLE) is a common symptom, and recent studies demonstrated that IL-6 has a pivotal role in its pathogenesis.
CASE PRESENTATION METHODS
We report a case of a 15 years old Caucasian boy with a history of persistent pleural effusion without lung involvement or fever. Microbiological and neoplastic aetiologies were previously excluded. Based on the presence of pleuritis, malar rash, reduction of C3 and C4 levels and positivity of antinuclear antibody (ANA) and anti-double stranded DNA (dsDNA), the diagnosis of juvenile SLE (JSLE) was performed. Treatment with high dose of intravenous glucocorticoids and mycophenolate mofetil was started with partial improvement of pleural effusion. Based on this and on adults SLE cases with serositis previously reported, therapy with intravenous tocilizumab (800 mg every two weeks) was started with prompt recovery of pleural effusion.
CONCLUSION CONCLUSIONS
To the best of our knowledge, this is the first case of JSLE pleuritis successfully treated with tocilizumab.

Identifiants

pubmed: 34530845
doi: 10.1186/s12969-021-00635-w
pii: 10.1186/s12969-021-00635-w
pmc: PMC8444491
doi:

Substances chimiques

Antibodies, Monoclonal, Humanized 0
tocilizumab I031V2H011

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

144

Informations de copyright

© 2021. The Author(s).

Références

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pubmed: 27503940
Arthritis Rheum. 2010 Feb;62(2):542-52
pubmed: 20112381
BMJ Case Rep. 2012 Dec 21;2012:
pubmed: 23264273
Arch Dis Child. 2014 Jun;99(6):563-7
pubmed: 24585754
Nat Rev Rheumatol. 2020 Jun;16(6):335-345
pubmed: 32327746
Lupus. 2005;14(10):822-6
pubmed: 16302677
Ann Rheum Dis. 2013 Jan;72(1):118-28
pubmed: 22858586
Clin Rheumatol. 2018 Jun;37(6):1695-1700
pubmed: 29362962

Auteurs

Arianna De Matteis (A)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy.

Emanuela Sacco (E)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy.

Camilla Celani (C)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy.

Andrea Uva (A)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy.

Virginia Messia (V)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy.

Rebecca Nicolai (R)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy.

Manuela Pardeo (M)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy.

Fabrizio De Benedetti (F)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy. fabrizio.debenedetti@opbg.net.

Claudia Bracaglia (C)

Division of Rheumatology, ERN RITA center, IRCCS Ospedale Pediatrico Bambino Gesù, Piazza Sant'Onofrio, 4-00165, Rome, Italy.

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Classifications MeSH