The diagnostic workup of children with the radiologically isolated syndrome differs by age and by sex
Naila Makhani
(1)
,
Christine Lebrun-Frenay
(2, 3, 4)
,
Aksel Siva
(5)
,
Veronika Shabanova
(1)
,
Evangeline Wassmer
(6)
,
Jonathan Santoro
(7, 8)
,
Sona Narula
(9)
,
J. Nicholas Brenton
(10)
,
Soe Mar
(11)
,
Francoise Durand-Dubief
(12)
,
Helene Zephir
(13, 14)
,
Guillaume Mathey
(15)
,
Juan Rojas
(16)
,
Jerome de Seze
(17, 18)
,
Silvia Tenembaum
(19)
,
Robert Thompson Stone
(20)
,
Olivier Casez
(21, 22, 23)
,
Clarisse Carra-Dallière
(24, 25)
,
Rinze Neuteboom
(26)
,
Nusrat Ahsan
(7, 8)
,
Hugo Arroyo
(27)
,
Philippe Cabre
(28)
,
Grace Gombolay
(29, 30)
,
Matilde Inglese
(31, 32)
,
Celine Louapre
(33, 34, 35)
,
Monica Margoni
(36)
,
Filipe Palavra
(37)
,
Daniela Pohl
(38)
,
Daniel Reich
(39)
,
Aurélie Ruet
(40, 41)
,
Eric Thouvenot
(42, 43)
,
Niklas Timby
(44)
,
Mar Tintore
(45, 46, 47)
,
Ugur Uygunoglu
(5)
,
Wendy Vargas
(48)
,
Sunita Venkateswaran
(38)
,
Helene Verhelst
(49)
,
Ronny Wickstrom
(50, 51)
,
Christina Azevedo
(7)
,
Orhun Kantarci
(52)
,
Eugene Shapiro
(1)
,
Darin Okuda
(53)
,
Daniel Pelletier
(7)
1
Yale University [New Haven]
2 CHU Nice - Centre Hospitalier Universitaire de Nice
3 URRIS UR2CA - Unité de Recherche Clinique de la Côte d’Azur
4 UniCA - Université Côte d'Azur
5 Cerrahpasa Faculty of Medicine
6 Aston University [Birmingham]
7 Keck School of Medicine [Los Angeles]
8 Children’s Hospital Los Angeles [Los Angeles]
9 CHOP - Children’s Hospital of Philadelphia
10 University of Virginia
11 Washington University School of Medicine [Saint Louis, MO]
12 GHE - Groupement Hospitalier Lyon-Est
13 LilNCog - Lille Neurosciences & Cognition - U 1172
14 CHRU Lille - Centre Hospitalier Régional Universitaire [CHU Lille]
15 Service de neurologie [CHRU Nancy]
16 CEMIC - Centro de Educación Médica e Investigaciones Clínicas
17 Hôpital de Hautepierre [Strasbourg]
18 Centre d’Investigation Clinique Plurithématique (CIC - P) - CIC Strasbourg
19 Hospital Nacional de Pediatría Prof. Dr Juan P. Garrahan [Buenos Aires]
20 URMC - University of Rochester Medical Center
21 CHUGA - Centre Hospitalier Universitaire [CHU Grenoble]
22 TIMC-T-RAIG - Translational Research in Autoimmunity and Inflammation Group
23 UGA - Université Grenoble Alpes
24 CHRU Montpellier - Centre Hospitalier Régional Universitaire [Montpellier]
25 UM - Université de Montpellier
26 Erasmus MC - Erasmus University Medical Center [Rotterdam]
27 Hospital Nacional de Pediatría J.P. Garrahan
28 CHU de Martinique - Centre Hospitalier Universitaire de Martinique [Fort-de-France, Martinique]
29 Emory University [Atlanta, GA]
30 Children's Healthcare of Atlanta [Atlanta, GA, USA]
31 DINOGMI - Dipartimento di Neuroscienze, riabilitazione, oftalmologia, genetica e scienze materno-infantili [Genova]
32 Ospedale Policlinico San Martino [Genoa]
33 CHU Pitié-Salpêtrière [AP-HP]
34 ICM - Institut du Cerveau = Paris Brain Institute
35 CIC Neurosciences - Centre d'investigation clinique Neurosciences [CHU Pitié Salpêtrière]
36 AOUP - Azienda Ospedale Università di Padova = Hospital-University of Padua
37 Centro Hospitalar e Universitário [Coimbra]
38 The Ottawa Hospital
39 NIH - National Institutes of Health [Bethesda, MD, USA]
40 CHU Bordeaux - Centre Hospitalier Universitaire de Bordeaux
41 U1215 Inserm - UB - Neurocentre Magendie : Physiopathologie de la Plasticité Neuronale
42 CHU Nîmes - Centre Hospitalier Universitaire de Nîmes
43 IGF - Institut de Génomique Fonctionnelle
44 Umeå University = Umeå Universitet
45 CemCat - Centre d'Esclerosi Múltiple de Catalunya
46 UAB - Universitat Autònoma de Barcelona = Autonomous University of Barcelona = Universidad Autónoma de Barcelona
47 UIC - Universitat Internacional de Catalunya [Barcelona]
48 CUMC - Columbia University Medical Center
49 Ghent University Hospital
50 Karolinska Institutet [Stockholm]
51 Karolinska University Hospital [Stockholm]
52 Mayo Clinic [Rochester]
53 University of Texas Southwestern Medical Center
2 CHU Nice - Centre Hospitalier Universitaire de Nice
3 URRIS UR2CA - Unité de Recherche Clinique de la Côte d’Azur
4 UniCA - Université Côte d'Azur
5 Cerrahpasa Faculty of Medicine
6 Aston University [Birmingham]
7 Keck School of Medicine [Los Angeles]
8 Children’s Hospital Los Angeles [Los Angeles]
9 CHOP - Children’s Hospital of Philadelphia
10 University of Virginia
11 Washington University School of Medicine [Saint Louis, MO]
12 GHE - Groupement Hospitalier Lyon-Est
13 LilNCog - Lille Neurosciences & Cognition - U 1172
14 CHRU Lille - Centre Hospitalier Régional Universitaire [CHU Lille]
15 Service de neurologie [CHRU Nancy]
16 CEMIC - Centro de Educación Médica e Investigaciones Clínicas
17 Hôpital de Hautepierre [Strasbourg]
18 Centre d’Investigation Clinique Plurithématique (CIC - P) - CIC Strasbourg
19 Hospital Nacional de Pediatría Prof. Dr Juan P. Garrahan [Buenos Aires]
20 URMC - University of Rochester Medical Center
21 CHUGA - Centre Hospitalier Universitaire [CHU Grenoble]
22 TIMC-T-RAIG - Translational Research in Autoimmunity and Inflammation Group
23 UGA - Université Grenoble Alpes
24 CHRU Montpellier - Centre Hospitalier Régional Universitaire [Montpellier]
25 UM - Université de Montpellier
26 Erasmus MC - Erasmus University Medical Center [Rotterdam]
27 Hospital Nacional de Pediatría J.P. Garrahan
28 CHU de Martinique - Centre Hospitalier Universitaire de Martinique [Fort-de-France, Martinique]
29 Emory University [Atlanta, GA]
30 Children's Healthcare of Atlanta [Atlanta, GA, USA]
31 DINOGMI - Dipartimento di Neuroscienze, riabilitazione, oftalmologia, genetica e scienze materno-infantili [Genova]
32 Ospedale Policlinico San Martino [Genoa]
33 CHU Pitié-Salpêtrière [AP-HP]
34 ICM - Institut du Cerveau = Paris Brain Institute
35 CIC Neurosciences - Centre d'investigation clinique Neurosciences [CHU Pitié Salpêtrière]
36 AOUP - Azienda Ospedale Università di Padova = Hospital-University of Padua
37 Centro Hospitalar e Universitário [Coimbra]
38 The Ottawa Hospital
39 NIH - National Institutes of Health [Bethesda, MD, USA]
40 CHU Bordeaux - Centre Hospitalier Universitaire de Bordeaux
41 U1215 Inserm - UB - Neurocentre Magendie : Physiopathologie de la Plasticité Neuronale
42 CHU Nîmes - Centre Hospitalier Universitaire de Nîmes
43 IGF - Institut de Génomique Fonctionnelle
44 Umeå University = Umeå Universitet
45 CemCat - Centre d'Esclerosi Múltiple de Catalunya
46 UAB - Universitat Autònoma de Barcelona = Autonomous University of Barcelona = Universidad Autónoma de Barcelona
47 UIC - Universitat Internacional de Catalunya [Barcelona]
48 CUMC - Columbia University Medical Center
49 Ghent University Hospital
50 Karolinska Institutet [Stockholm]
51 Karolinska University Hospital [Stockholm]
52 Mayo Clinic [Rochester]
53 University of Texas Southwestern Medical Center
Naila Makhani
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- PersonId : 786682
- ORCID : 0000-0002-2325-130X
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Christine Lebrun-Frenay
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- PersonId : 770285
- ORCID : 0000-0002-3713-2416
- IdRef : 074517929
Francoise Durand-Dubief
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- IdHAL : francoise-durand-dubief
- ORCID : 0000-0002-2860-1700
- IdRef : 15778911X
Helene Zephir
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- PersonId : 1297076
- ORCID : 0000-0003-4029-2012
- IdRef : 081106475
Jerome de Seze
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- PersonId : 962597
- ORCID : 0000-0002-7197-7578
Résumé
Background: Cerebrospinal fluid (CSF) and spinal MRIs are often obtained in children with the radiologically isolated syndrome (RIS) for diagnosis and prognosis. Factors affecting the frequency and timing of these tests are unknown.
Objective: To determine whether age or sex were associated with (1) having CSF or spinal MRI obtained or (2) the timing of these tests.
Methods: We analyzed children (≤ 18 y) with RIS enrolled in an international longitudinal study. Index scans met 2010/2017 multiple sclerosis (MS) MRI criteria for dissemination in space (DIS). We used Fisher's exact test and multivariable logistic regression (covariates = age, sex, MRI date, MRI indication, 2005 MRI DIS criteria met, and race).
Results: We included 103 children with RIS (67% girls, median age = 14.9 y). Children ≥ 12 y were more likely than children < 12 y to have CSF obtained (58% vs. 21%, adjusted odds ratio [AOR] = 4.9, p = 0.03). Pre-2017, girls were more likely than boys to have CSF obtained (n = 70, 79% vs. 52%, AOR = 4.6, p = 0.01), but not more recently (n = 30, 75% vs. 80%, AOR = 0.2, p = 0.1; p = 0.004 for interaction). Spinal MRIs were obtained sooner in children ≥ 12 y (median 11d vs. 159d, p = 0.03).
Conclusions: Younger children with RIS may be at continued risk for misdiagnosis and misclassification of MS risk. Consensus guidelines are needed.