Juvenile Neuropsychiatric Systemic Lupus Erythematosus: Identification of Novel Central Neuroinflammation Biomarkers
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 chal...
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| Veröffentlicht in: | Journal of clinical immunology 2023-04, Vol.43 (3), p.615-624 |
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| creator | 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, Isabelle |
| description | 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 (
R
s
= 0.832,
p
|
| doi_str_mv | 10.1007/s10875-022-01407-1 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_pubme</sourceid><recordid>TN_cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_9957825</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2747003994</sourcerecordid><originalsourceid>FETCH-LOGICAL-c512t-a475f90918ac76a41892423332005d77ad2c515450cdf22ea8f33583c20b7ec3</originalsourceid><addsrcrecordid>eNp9ksFu1DAQhi1ERZfCC3BAkbhwCYzHcRxzQCqrQlut2gO9W67jdF2SONjxSvv2uKQU6KEnW55v_vk9M4S8ofCBAoiPkUIjeAmIJdAKREmfkRXlgpXIJT4nK0BBS0krPCQvY7wFAFYjf0EOWV3Vkkq6IvN52tnR9ba4sCn4Ke7N1uk5OFN838fZDvmySVOKxUnYz1s76NnHFD8VZ60dZ9c5o2fnx8J3xYXf2b5Y5-eg-0XOjV2vh2FBvjg_6PDDhviKHHS6j_b1_XlErr6eXK1Py83lt7P18aY0nOJc6krwToKkjTai1hVtJFbIGEMA3gqhW8wgrziYtkO0uukY4w0zCNfCGnZEPi-yU7oebGsWZ2oKLvvYK6-d-j8yuq268TslJRcN8ixQLgLbR2mnxxs16dyeFBSwppE1pTua-ff3BYP_mWyc1eCisX2vR-tTVCgqkWcgZZXRd4_QW5_CmLuRKSEFYMMgU7hQJvgYg-0eXFBQdzuglh1QeQfU7x1Qdy7e_vvth5Q_Q88AW4CYQ-ONDX9rPyH7C7wSvpQ</addsrcrecordid><sourcetype>Open Access Repository</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2779702830</pqid></control><display><type>article</type><title>Juvenile Neuropsychiatric Systemic Lupus Erythematosus: Identification of Novel Central Neuroinflammation Biomarkers</title><source>MEDLINE</source><source>SpringerLink Journals - AutoHoldings</source><creator>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, Isabelle</creator><creatorcontrib>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, Isabelle</creatorcontrib><description>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 (
R
s
= 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.</description><identifier>ISSN: 0271-9142</identifier><identifier>EISSN: 1573-2592</identifier><identifier>DOI: 10.1007/s10875-022-01407-1</identifier><identifier>PMID: 36469191</identifier><language>eng</language><publisher>New York: Springer US</publisher><subject>Autoimmune diseases ; Biomarkers ; Biomedical and Life Sciences ; Biomedicine ; Central nervous system ; Cerebrospinal fluid ; Child ; Cognitive ability ; Diagnosis ; Humans ; Immunology ; Infectious Diseases ; Inflammation ; Internal Medicine ; Life Sciences ; Lupus ; Lupus Erythematosus, Systemic - diagnosis ; Lupus Vasculitis, Central Nervous System ; Medical Microbiology ; Neopterin ; Neuroinflammatory Diseases ; Neurological complications ; Original ; Original Article ; Patients ; Pediatrics ; Retrospective Studies ; Systemic lupus erythematosus ; α-Interferon</subject><ispartof>Journal of clinical immunology, 2023-04, Vol.43 (3), p.615-624</ispartof><rights>The Author(s) 2022</rights><rights>2022. The Author(s).</rights><rights>The Author(s) 2022. This work is published under http://creativecommons.org/licenses/by/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><rights>Attribution</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c512t-a475f90918ac76a41892423332005d77ad2c515450cdf22ea8f33583c20b7ec3</citedby><cites>FETCH-LOGICAL-c512t-a475f90918ac76a41892423332005d77ad2c515450cdf22ea8f33583c20b7ec3</cites><orcidid>0000-0002-8057-333X ; 0000-0002-8875-2308 ; 0000-0001-8476-8196 ; 0000-0001-8539-6507</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s10875-022-01407-1$$EPDF$$P50$$Gspringer$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s10875-022-01407-1$$EHTML$$P50$$Gspringer$$Hfree_for_read</linktohtml><link.rule.ids>230,314,776,780,881,27901,27902,41464,42533,51294</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/36469191$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink><backlink>$$Uhttps://pasteur.hal.science/pasteur-03889611$$DView record in HAL$$Hfree_for_read</backlink></links><search><creatorcontrib>Labouret, Mathilde</creatorcontrib><creatorcontrib>Costi, Stefania</creatorcontrib><creatorcontrib>Bondet, Vincent</creatorcontrib><creatorcontrib>Trebossen, Vincent</creatorcontrib><creatorcontrib>Le Roux, Enora</creatorcontrib><creatorcontrib>Ntorkou, Alexandra</creatorcontrib><creatorcontrib>Bartoli, Sophie</creatorcontrib><creatorcontrib>Auvin, Stéphane</creatorcontrib><creatorcontrib>Bader-Meunier, Brigitte</creatorcontrib><creatorcontrib>Baudouin, Véronique</creatorcontrib><creatorcontrib>Corseri, Olivier</creatorcontrib><creatorcontrib>Dingulu, Glory</creatorcontrib><creatorcontrib>Ducrocq, Camille</creatorcontrib><creatorcontrib>Dumaine, Cécile</creatorcontrib><creatorcontrib>Elmaleh, Monique</creatorcontrib><creatorcontrib>Fabien, Nicole</creatorcontrib><creatorcontrib>Faye, Albert</creatorcontrib><creatorcontrib>Hau, Isabelle</creatorcontrib><creatorcontrib>Hentgen, Véronique</creatorcontrib><creatorcontrib>Kwon, Théresa</creatorcontrib><creatorcontrib>Meinzer, Ulrich</creatorcontrib><creatorcontrib>Ouldali, Naim</creatorcontrib><creatorcontrib>Parmentier, Cyrielle</creatorcontrib><creatorcontrib>Pouletty, Marie</creatorcontrib><creatorcontrib>Renaldo, Florence</creatorcontrib><creatorcontrib>Savioz, Isabelle</creatorcontrib><creatorcontrib>Rozenberg, Flore</creatorcontrib><creatorcontrib>Frémond, Marie-Louise</creatorcontrib><creatorcontrib>Lepelley, Alice</creatorcontrib><creatorcontrib>Rice, Gillian I.</creatorcontrib><creatorcontrib>Seabra, Luis</creatorcontrib><creatorcontrib>Benoist, Jean-François</creatorcontrib><creatorcontrib>Duffy, Darragh</creatorcontrib><creatorcontrib>Crow, Yanick J.</creatorcontrib><creatorcontrib>Ellul, Pierre</creatorcontrib><creatorcontrib>Melki, Isabelle</creatorcontrib><title>Juvenile Neuropsychiatric Systemic Lupus Erythematosus: Identification of Novel Central Neuroinflammation Biomarkers</title><title>Journal of clinical immunology</title><addtitle>J Clin Immunol</addtitle><addtitle>J Clin Immunol</addtitle><description>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 (
R
s
= 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.</description><subject>Autoimmune diseases</subject><subject>Biomarkers</subject><subject>Biomedical and Life Sciences</subject><subject>Biomedicine</subject><subject>Central nervous system</subject><subject>Cerebrospinal fluid</subject><subject>Child</subject><subject>Cognitive ability</subject><subject>Diagnosis</subject><subject>Humans</subject><subject>Immunology</subject><subject>Infectious Diseases</subject><subject>Inflammation</subject><subject>Internal Medicine</subject><subject>Life Sciences</subject><subject>Lupus</subject><subject>Lupus Erythematosus, Systemic - diagnosis</subject><subject>Lupus Vasculitis, Central Nervous System</subject><subject>Medical Microbiology</subject><subject>Neopterin</subject><subject>Neuroinflammatory Diseases</subject><subject>Neurological complications</subject><subject>Original</subject><subject>Original Article</subject><subject>Patients</subject><subject>Pediatrics</subject><subject>Retrospective Studies</subject><subject>Systemic lupus erythematosus</subject><subject>α-Interferon</subject><issn>0271-9142</issn><issn>1573-2592</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2023</creationdate><recordtype>article</recordtype><sourceid>C6C</sourceid><sourceid>EIF</sourceid><sourceid>BENPR</sourceid><recordid>eNp9ksFu1DAQhi1ERZfCC3BAkbhwCYzHcRxzQCqrQlut2gO9W67jdF2SONjxSvv2uKQU6KEnW55v_vk9M4S8ofCBAoiPkUIjeAmIJdAKREmfkRXlgpXIJT4nK0BBS0krPCQvY7wFAFYjf0EOWV3Vkkq6IvN52tnR9ba4sCn4Ke7N1uk5OFN838fZDvmySVOKxUnYz1s76NnHFD8VZ60dZ9c5o2fnx8J3xYXf2b5Y5-eg-0XOjV2vh2FBvjg_6PDDhviKHHS6j_b1_XlErr6eXK1Py83lt7P18aY0nOJc6krwToKkjTai1hVtJFbIGEMA3gqhW8wgrziYtkO0uukY4w0zCNfCGnZEPi-yU7oebGsWZ2oKLvvYK6-d-j8yuq268TslJRcN8ixQLgLbR2mnxxs16dyeFBSwppE1pTua-ff3BYP_mWyc1eCisX2vR-tTVCgqkWcgZZXRd4_QW5_CmLuRKSEFYMMgU7hQJvgYg-0eXFBQdzuglh1QeQfU7x1Qdy7e_vvth5Q_Q88AW4CYQ-ONDX9rPyH7C7wSvpQ</recordid><startdate>20230401</startdate><enddate>20230401</enddate><creator>Labouret, Mathilde</creator><creator>Costi, Stefania</creator><creator>Bondet, Vincent</creator><creator>Trebossen, Vincent</creator><creator>Le Roux, Enora</creator><creator>Ntorkou, Alexandra</creator><creator>Bartoli, Sophie</creator><creator>Auvin, Stéphane</creator><creator>Bader-Meunier, Brigitte</creator><creator>Baudouin, Véronique</creator><creator>Corseri, Olivier</creator><creator>Dingulu, Glory</creator><creator>Ducrocq, Camille</creator><creator>Dumaine, Cécile</creator><creator>Elmaleh, Monique</creator><creator>Fabien, Nicole</creator><creator>Faye, Albert</creator><creator>Hau, Isabelle</creator><creator>Hentgen, Véronique</creator><creator>Kwon, Théresa</creator><creator>Meinzer, Ulrich</creator><creator>Ouldali, Naim</creator><creator>Parmentier, Cyrielle</creator><creator>Pouletty, Marie</creator><creator>Renaldo, Florence</creator><creator>Savioz, Isabelle</creator><creator>Rozenberg, Flore</creator><creator>Frémond, Marie-Louise</creator><creator>Lepelley, Alice</creator><creator>Rice, Gillian I.</creator><creator>Seabra, Luis</creator><creator>Benoist, Jean-François</creator><creator>Duffy, Darragh</creator><creator>Crow, Yanick J.</creator><creator>Ellul, Pierre</creator><creator>Melki, Isabelle</creator><general>Springer US</general><general>Springer Nature B.V</general><general>Springer 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Neuropsychiatric Systemic Lupus Erythematosus: Identification of Novel Central Neuroinflammation Biomarkers</title><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, Isabelle</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c512t-a475f90918ac76a41892423332005d77ad2c515450cdf22ea8f33583c20b7ec3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2023</creationdate><topic>Autoimmune diseases</topic><topic>Biomarkers</topic><topic>Biomedical and Life Sciences</topic><topic>Biomedicine</topic><topic>Central nervous system</topic><topic>Cerebrospinal fluid</topic><topic>Child</topic><topic>Cognitive ability</topic><topic>Diagnosis</topic><topic>Humans</topic><topic>Immunology</topic><topic>Infectious Diseases</topic><topic>Inflammation</topic><topic>Internal Medicine</topic><topic>Life Sciences</topic><topic>Lupus</topic><topic>Lupus Erythematosus, Systemic - diagnosis</topic><topic>Lupus Vasculitis, Central Nervous System</topic><topic>Medical Microbiology</topic><topic>Neopterin</topic><topic>Neuroinflammatory Diseases</topic><topic>Neurological complications</topic><topic>Original</topic><topic>Original Article</topic><topic>Patients</topic><topic>Pediatrics</topic><topic>Retrospective Studies</topic><topic>Systemic lupus erythematosus</topic><topic>α-Interferon</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Labouret, Mathilde</creatorcontrib><creatorcontrib>Costi, Stefania</creatorcontrib><creatorcontrib>Bondet, Vincent</creatorcontrib><creatorcontrib>Trebossen, Vincent</creatorcontrib><creatorcontrib>Le Roux, Enora</creatorcontrib><creatorcontrib>Ntorkou, Alexandra</creatorcontrib><creatorcontrib>Bartoli, Sophie</creatorcontrib><creatorcontrib>Auvin, Stéphane</creatorcontrib><creatorcontrib>Bader-Meunier, Brigitte</creatorcontrib><creatorcontrib>Baudouin, Véronique</creatorcontrib><creatorcontrib>Corseri, Olivier</creatorcontrib><creatorcontrib>Dingulu, Glory</creatorcontrib><creatorcontrib>Ducrocq, Camille</creatorcontrib><creatorcontrib>Dumaine, 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Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>Biological Science Collection</collection><collection>ProQuest Central</collection><collection>Natural Science Collection</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>SciTech Premium Collection</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>ProQuest Biological Science Collection</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Biological Science Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><collection>Hyper Article en Ligne (HAL)</collection><collection>Hyper Article en Ligne (HAL) (Open Access)</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Journal of clinical immunology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Labouret, Mathilde</au><au>Costi, Stefania</au><au>Bondet, Vincent</au><au>Trebossen, Vincent</au><au>Le Roux, Enora</au><au>Ntorkou, Alexandra</au><au>Bartoli, Sophie</au><au>Auvin, Stéphane</au><au>Bader-Meunier, Brigitte</au><au>Baudouin, Véronique</au><au>Corseri, Olivier</au><au>Dingulu, Glory</au><au>Ducrocq, Camille</au><au>Dumaine, Cécile</au><au>Elmaleh, Monique</au><au>Fabien, Nicole</au><au>Faye, Albert</au><au>Hau, Isabelle</au><au>Hentgen, Véronique</au><au>Kwon, Théresa</au><au>Meinzer, Ulrich</au><au>Ouldali, Naim</au><au>Parmentier, Cyrielle</au><au>Pouletty, Marie</au><au>Renaldo, Florence</au><au>Savioz, Isabelle</au><au>Rozenberg, Flore</au><au>Frémond, Marie-Louise</au><au>Lepelley, Alice</au><au>Rice, Gillian I.</au><au>Seabra, Luis</au><au>Benoist, Jean-François</au><au>Duffy, Darragh</au><au>Crow, Yanick J.</au><au>Ellul, Pierre</au><au>Melki, Isabelle</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Juvenile Neuropsychiatric Systemic Lupus Erythematosus: Identification of Novel Central Neuroinflammation Biomarkers</atitle><jtitle>Journal of clinical immunology</jtitle><stitle>J Clin Immunol</stitle><addtitle>J Clin Immunol</addtitle><date>2023-04-01</date><risdate>2023</risdate><volume>43</volume><issue>3</issue><spage>615</spage><epage>624</epage><pages>615-624</pages><issn>0271-9142</issn><eissn>1573-2592</eissn><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 (
R
s
= 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.</abstract><cop>New York</cop><pub>Springer US</pub><pmid>36469191</pmid><doi>10.1007/s10875-022-01407-1</doi><tpages>10</tpages><orcidid>https://orcid.org/0000-0002-8057-333X</orcidid><orcidid>https://orcid.org/0000-0002-8875-2308</orcidid><orcidid>https://orcid.org/0000-0001-8476-8196</orcidid><orcidid>https://orcid.org/0000-0001-8539-6507</orcidid><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 0271-9142 |
| ispartof | Journal of clinical immunology, 2023-04, Vol.43 (3), p.615-624 |
| issn | 0271-9142 1573-2592 |
| language | eng |
| recordid | cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_9957825 |
| source | MEDLINE; SpringerLink Journals - AutoHoldings |
| subjects | Autoimmune diseases Biomarkers Biomedical and Life Sciences Biomedicine Central nervous system Cerebrospinal fluid Child Cognitive ability Diagnosis Humans Immunology Infectious Diseases Inflammation Internal Medicine Life Sciences Lupus Lupus Erythematosus, Systemic - diagnosis Lupus Vasculitis, Central Nervous System Medical Microbiology Neopterin Neuroinflammatory Diseases Neurological complications Original Original Article Patients Pediatrics Retrospective Studies Systemic lupus erythematosus α-Interferon |
| title | Juvenile Neuropsychiatric Systemic Lupus Erythematosus: Identification of Novel Central Neuroinflammation Biomarkers |
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