Juvenile Neuropsychiatric Systemic Lupus Erythematosus: Identification of Novel Central Neuroinflammation Biomarkers
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Published:2022-12-05
Issue:3
Volume:43
Page:615-624
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ISSN:0271-9142
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Container-title:Journal of Clinical Immunology
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language:en
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Short-container-title:J Clin Immunol
Author:
Labouret Mathilde, Costi Stefania, Bondet Vincent, Trebossen Vincent, Le Roux Enora, Ntorkou Alexandra, Bartoli Sophie, Auvin Stéphane, Bader-Meunier Brigitte, Baudouin Véronique, Corseri Olivier, Dingulu Glory, Ducrocq Camille, Dumaine Cécile, Elmaleh Monique, Fabien Nicole, Faye Albert, Hau Isabelle, Hentgen Véronique, Kwon Théresa, Meinzer Ulrich, Ouldali Naim, Parmentier Cyrielle, Pouletty Marie, Renaldo Florence, Savioz Isabelle, Rozenberg Flore, Frémond Marie-Louise, Lepelley Alice, Rice Gillian I., Seabra Luis, Benoist Jean-François, Duffy Darragh, Crow Yanick J., Ellul Pierre, Melki IsabelleORCID
Abstract
Abstract
Introduction
Juvenile systemic lupus erythematosus (j-SLE) is a rare chronic autoimmune disease affecting multiple organs. Ranging from minor features, such as headache or mild cognitive impairment, to serious and life-threatening presentations, j-neuropsychiatric SLE (j-NPSLE) is a therapeutic challenge. Thus, the diagnosis of NPSLE remains difficult, especially in pediatrics, with no specific biomarker of the disease yet validated.
Objectives
To identify central nervous system (CNS) disease biomarkers of j-NPSLE.
Methods
A 5-year retrospective tertiary reference monocentric j-SLE study. A combination of standardized diagnostic criteria and multidisciplinary pediatric clinical expertise was combined to attribute NP involvement in the context of j-SLE. Neopterin and interferon-alpha (IFN-α) protein levels in cerebrospinal fluid (CSF) were assessed, together with routine biological and radiological investigations.
Results
Among 51 patients with j-SLE included, 39% presented with j-NPSLE. J-NPSLE was diagnosed at onset of j-SLE in 65% of patients. No specific routine biological or radiological marker of j-NPSLE was identified. However, CSF neopterin levels were significantly higher in active j-NPSLE with CNS involvement than in j-SLE alone (p = 0.0008). Neopterin and IFN-α protein levels in CSF were significantly higher at diagnosis of j-NPSLE with CNS involvement than after resolution of NP features (respectively p = 0.0015 and p = 0.0010) upon immunosuppressive treatment in all patients tested (n = 10). Both biomarkers correlated strongly with each other (Rs = 0.832, p < 0.0001, n = 23 paired samples).
Conclusion
CSF IFN-α and neopterin constitute promising biomarkers useful in the diagnosis and monitoring of activity in j-NPSLE.
Funder
H2020 European Research Council Agence Nationale de la Recherche
Publisher
Springer Science and Business Media LLC
Subject
Immunology,Immunology and Allergy
Reference38 articles.
1. Arnaud L, Fagot J-P, Mathian A, Paita M, Fagot-Campagna A, Amoura Z. Prevalence and incidence of systemic lupus erythematosus in France: a 2010 nation-wide population-based study. Autoimmun Rev. 2014;13(11):1082–9. 2. Sibbitt WL, Brandt JR, Johnson CR, Maldonado ME, Patel SR, Ford CC, et al. The incidence and prevalence of neuropsychiatric syndromes in pediatric onset systemic lupus erythematosus. J Rheumatol. 2002;29(7):1536–42. 3. Olfat MO, Al-Mayouf SM, Muzaffer MA. Pattern of neuropsychiatric manifestations and outcome in juvenile systemic lupus erythematosus. Clin Rheumatol. 2004;23(5):395–9. 4. Giani T, Smith EM, Al-Abadi E, Armon K, Bailey K, Ciurtin C, et al. Neuropsychiatric involvement in juvenile-onset systemic lupus erythematosus: data from the UK juvenile-onset systemic lupus erythematosus cohort study. Lupus. 2021;2:9612033211045050. 5. Hanly JG, Urowitz MB, Su L, Bae SC, Gordon C, Wallace DJ, et al. Prospective analysis of neuropsychiatric events in an international disease inception cohort of patients with systemic lupus erythematosus. Ann Rheum Dis. 2010;69(3):529–35.
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