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

Prognostic relevance of MOG antibodies in children with an acquired demyelinating syndrome
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DOI:
10.1212/wnl.0000000000004312
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发表时间:
2017-08-29
期刊:
影响因子:
9.9
通讯作者:
Rostasy, Kevin
Rostasy, Kevin
中科院分区:
医学1区
文献类型:
--
作者:
Hennes, Eva-Maria;Baumann, Matthias;Rostasy, Kevin

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目的:评估MOG抗体(abs)在获得性脱髓鞘综合征(ADS)鉴别诊断中的预后价值。方法:在这项观察性、前瞻性、多中心、以医院为基础的研究中,回顾了ADS患儿的临床病程、MRI、MOG-abs、AQP 4-abs、CSF细胞和寡克隆带(OCB)以及24个月的随访。210例ADS患儿被纳入并诊断为急性播散性脑脊髓炎(ADEM)(n = 60)、视神经肌萎缩谱系障碍(NMOSD)(n = 12)、临床孤立综合征(CIS)(n = 101)、多发性硬化(MS)37例。MOG-abs主要见于ADEM(57%),较少见于NMOSD(25%)、CIS(25%)或MS(8%)。MOG-ab滴度增加与年龄较小(p = 0.0001)、ADEM诊断(p = 0.005)、CSF细胞计数增加(p = 0.011)和OCB阴性(p = 0.012)相关。在24个月的随访中,96名儿童没有进一步复发。35名儿童发生复发性非MS发作(63% MOG-,17% AQP 4-abs发作)。79名儿童发展为MS(发病时MOG-abs为4%)。复发性非MS发作与高MOG-ab滴度(p = 0.0003)和发病年龄较大(p = 0.024)相关。MS通过MS样MRI(p < 0.0001)和OCB(p = 0.007)预测。MOG-ab临界滴度≥ 1:1,280预测非MS病程的敏感性为47%,特异性为100%,预测复发性非MS病程的敏感性为46%,特异性为86%.Conclusions:我们的研究结果表明,MOG-ab的存在强烈依赖于疾病发作时的年龄,高MOG-ab滴度与复发性非MS病程相关。
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.