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Clinical course, treatment responses and outcomes in Chinese paediatric neuromyelitis optica spectrum disorder.

Abstract：

BACKGROUND:Few data exists on paediatric neuromyelitis optica spectrum disorder (NMOSD). Here, we investigated the clinical presentation, treatment responses and long-term prognoses in a large cohort of patients with NMOSD and compared between children and adults with aquaporin-4 antibody (AQP4-IgG). METHODS:A retrospective chart review of patients followed in multiple centres identified 127 patients with NMOSD (31 children; 96 adults). Data were collected through medical records and structured questionnaire. RESULTS:In the paediatric cohort, median age at onset was 14 (range 7-17) years; 87% were female. AQP4 and myelin oligodendrocyte glycoprotein antibodies were detected in 82% and 16%, respectively. During a median disease duration of 48 months, 23% developed visual acuity of <6/60 Snellen, 3% were wheelchair-dependent. The frequency of brain/brainstem phenotype (18% vs 9%, p = 0.018) was more common in AQP4-IgG-positive children, while AQP4-IgG-positive adults were more likely to present transverse myelitis (TM) (44% vs 29%, p = 0.005) of all 452 episodes. Multivariable analyses showed that sustained disability was independently associated with the presence of TM (p = 0.030), brain/brainstem symptoms (p = 0.010), annualized relapse rate (p < 0.001) and possibly age of onset (p = 0.069). The reduction of ARR after azathioprine was more prominent in adults (79%) than in children (48%). Mycophenolate mofetil and rituximab decreased the relapse frequency of children, with a reduction of 94% and 100%, respectively. CONCLUSIONS:paediatric NMOSD is a severely disabling disorder characterized by repeated brain attacks and early disability accrual. Prompt therapy including mycophenolate mofetil and rituximab should be considered to improve paediatric care.

journal_name

Mult Scler Relat Disord

journal_title

Multiple sclerosis and related disorders

authors

Zhou Y,Zhong X,Shu Y,Cui C,Wang J,Wang Y,Li X,Chen Z,Peng L,Kermode A,Qiu W

doi

10.1016/j.msard.2018.12.038

subject

Has Abstract

pub_date

2019-02-01 00:00:00

pages

213-220

eissn

2211-0348

issn

2211-0356

pii

S2211-0348(18)30566-2

journal_volume

28

pub_type

杂志文章,多中心研究

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