Prognostic relevance of MOG antibodies in children with an acquired demyelinating syndrome

被引:277
作者
Hennes, Eva-Maria [1 ,29 ]
Baumann, Matthias [2 ]
Schanda, Kathrin [3 ]
Anlar, Banu [5 ]
Bajer-Kornek, Barbara [6 ]
Blaschek, Astrid [7 ]
Brantner-Inthaler, Sigrid [8 ]
Diepold, Katharina [9 ]
Eisenkolbl, Astrid [10 ]
Gotwald, Thaddaeus [4 ]
Kuchukhidze, Georgi [3 ]
Gruber-Sedlmayr, Ursula [11 ]
Haeusler, Martin [12 ]
Hoeftberger, Romana [6 ]
Karenfort, Michael [13 ]
Klein, Andrea [14 ]
Koch, Johannes [15 ]
Kraus, Verena [16 ]
Lechner, Christian [2 ]
Leiz, Steffen [17 ]
Leypoldt, Frank [18 ]
Mader, Simone [19 ]
Marquard, Klaus [1 ]
Poggenburg, Imke [20 ]
Pohl, Daniela [21 ]
Pritsch, Martin [22 ]
Raucherzauner, Markus [23 ]
Schimmel, Mareike [24 ]
Thiels, Charlotte [25 ]
Tibussek, Daniel [26 ]
Vieker, Silvia [27 ]
Zeches, Carolin [28 ]
Berger, Thomas [3 ]
Reindl, Markus [3 ]
Rostasy, Kevin [29 ]
机构
[1] Olga Hosp, Dept Pediat, Stuttgart, Germany
[2] Med Univ Innsbruck, Dept Pediat 1, Div Pediat Neurol, Innsbruck, Austria
[3] Med Univ Innsbruck, Dept Clin Neurol, Innsbruck, Austria
[4] Med Univ Innsbruck, Dept Neuroradiol, Innsbruck, Austria
[5] Hacettepe Univ, Fac Med, Dept Pediat Neurol, Ankara, Turkey
[6] Med Univ Vienna, Inst Neurol, Vienna, Austria
[7] Univ Munich, Dr von Hauners Childrens Hosp, Dept Pediat Neurol & Dev Med, Munich, Germany
[8] Donauspital, Dept Pediat, Vienna, Austria
[9] Klinikum Kassel, Dept Pediat, Div Pediat Neurol, Kassel, Germany
[10] Womens & Childrens Hosp, Dept Pediat, Linz, Austria
[11] LKH Med Univ Graz, Dept Pediat, Graz, Austria
[12] Med Univ RWTH Aachen, Dept Pediat, Div Neuropediat & Social Pediat, Aachen, Germany
[13] Heinrich Heine Univ, Univ Childrens Hosp, Dept Gen Pediat Neonatol & Pediat Cardiol, Dusseldorf, Germany
[14] Univ Childrens Hosp Basel & Bern, Dept Neuropediat, Basel, Switzerland
[15] Salzburger Landeskliniken SALK, Dept Pediat, Salzburg, Austria
[16] Tech Univ Munich, Childrens Hosp Schwabing, Munich, Germany
[17] Klinikum Dritter Orden, Dept Pediat, Div Pediat Neurol, Munich, Germany
[18] Med Univ Schleswig Hostein, Inst Clin Chem & Dept, Neuroimmunol, Kiel, Germany
[19] Feinstein Inst Med Res, Ctr Autoimmune & Musculoskeletal Dis, Manhasset, NY USA
[20] Med Univ Oldenburg, Elisabeth Childrens Hosp, Dept Pediat Neurol, Oldenburg, Germany
[21] Univ Ottawa, Childrens Hosp Eastern Ontario, Dept Neurol, Ottawa, ON, Canada
[22] Childrens Hosp DRK Siegen, Dept Neuropediat, Siegen, Germany
[23] Kliniken Ostallgau Kaufbeuren, Dept Pediat, Kaufbeuren, Germany
[24] Klinikum Augsburg, Childrens Hosp, Div Pediat Neurol, Augsburg, Germany
[25] Ruhr Univ Bochum, Univ Childrens Hosp, Dept Neuropediat, Bochum, Germany
[26] Heinrich Heine Univ Dusseldorf, ZSE, Dept Pediat, Dusseldorf, Germany
[27] Klinikum Bayreuth, Dept Pediat, Div Pediat Neurol, Bayreuth, Germany
[28] Schoen Klinikum Vogtareuth, Dept Pediat, Div Pediat Neurol, Vogtareuth, Germany
[29] Univ Witten Herdecke, Vest Kinder & Jugendklin Datteln, Dept Pediat Neurol, Witten, Germany
关键词
MYELIN OLIGODENDROCYTE GLYCOPROTEIN; ACUTE DISSEMINATED ENCEPHALOMYELITIS; PEDIATRIC MULTIPLE-SCLEROSIS; NEUROMYELITIS-OPTICA; SPECTRUM DISORDERS; DISEASES; NMO; AUTOIMMUNITY; MULTICENTER; DISTINCTION;
D O I
10.1212/WNL.0000000000004312
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Objective: To assess the prognostic value of MOG antibodies (abs) in the differential diagnosis of acquired demyelinating syndromes (ADS). Methods: Clinical course, MRI, MOG-abs, AQP4-abs, and CSF cells and oligoclonal bands (OCB) in children with ADS and 24 months of follow-up were reviewed in this observational prospective multicenter hospital-based study. Results: Two hundred ten children with ADS were included and diagnosed with acute disseminated encephalomyelitis (ADEM) (n = 60), neuromyelitis optica spectrum disorder (NMOSD) (n = 12), clinically isolated syndrome (CIS) (n = 101), and multiple sclerosis (MS) (n = 37) after the first episode. MOG-abs were predominantly found in ADEM (57%) and less frequently in NMOSD (25%), CIS (25%), or MS (8%). Increased MOG-ab titers were associated with younger age (p = 0.0001), diagnosis of ADEM (p = 0.005), increased CSF cell counts (p = 0.011), and negative OCB (p = 0.012). At 24-month follow-up, 96 children had no further relapses. Thirtyfive children developed recurrent non-MS episodes (63% MOG-, 17% AQP4-abs at onset). Seventy-nine children developed MS (4% MOG-abs at onset). Recurrent non-MS episodes were associated with high MOG-ab titers (p = 0.0003) and older age at onset (p = 0.024). MS was predicted by MS-like MRI (p < 0.0001) and OCB (p = 0.007). An MOG-ab cutoff titer >= 1:1,280 predicted a non-MS course with a sensitivity of 47% and a specificity of 100% and a recurrent non-MS course with a sensitivity of 46% and a specificity of 86%. Conclusions: Our results show that the presence of MOG-abs strongly depends on the age at disease onset and that high MOG-ab titers were associated with a recurrent non-MS disease course.
引用
收藏
页码:900 / 908
页数:9
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