Neuromyelitis optica and NMO-IgG in European pediatric patients

Huppke, Peter; Blüthner, Martin; Bauer, O.; Stark, Wiebke; Reinhardt, Katarina; Huppke, Brenda; Gärtner, Jutta

doi:10.1212/wnl.0b013e3181fc2823

Cited by 53 publications

(52 citation statements)

References 18 publications

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“…Only 1 patient with recurrence, who died at 7 years after disease onset, was seroposi- tive for NMO-IgG. All patients with monophasic NMO were seronegative for NMO-IgG and recovered well, in the similar course to the present patient (4). Other studies have also described low rates of NMO-IgG seropositivity in pediatric or adult patients with monophasic NMO (10,12).…”

Section: Discussionsupporting

confidence: 70%

“…The number of first brain MRI lesions was significantly increased and disability time was prolonged in p-NMO patients compared to adult-onset NMO (3). Another German study addressed contrary aspects of p-NMO patients (4). Of 118 pediatric patients with demyelinating central nervous system disorders, 6 patients (5%) fulfilled the revised NMO diagnostic criteria (9).…”

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

“…Recent studies have suggested neurological features of pediatric-onset NMO (p-NMO) patients (2)(3)(4). A high frequency of parainfectious or postinfectious NMO has been noted in p-NMO patients compared to adult patients (4,5). Influenza infection or vaccine rarely causes acute encephalopathy or encephalitis in children and adults (6)(7)(8).…”

Section: Introductionmentioning

confidence: 99%

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Influenza-associated Monophasic Neuromyelitis Optica

et al. 2011

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Neuromyelitis optica (NMO) is an inflammatory demyelinating disorder characterized by optic neuritis and acute myelitis. A parainfectious pathogenesis may play a partial role in the development of this disorder. Several viral infections are known to cause NMO. Here we report the case of a 15-year-old girl diagnosed with postinfluenza monophasic NMO. The patient developed sudden fever and chills, and the rapid diagnostic test for influenza was positive. She was diagnosed as influenza A and was treated with zanamivir hydrate (10 mg/day, inhalation). Three days later, she complained of dysuria and dysesthesia in the lower extremities. After nine days, she experienced blurred vision bilaterally. Neurological examination revealed visual disturbance, dysuria, dysesthesia and hyperreflexia in the lower extremities. Her visual acuity was counting fingers in OD and 2/100 in OS. Pupillary size was 4.0 mm and light reflexes were sluggish on both sides. Ophthalmoscopy showed marked edema of the optic discs. Serum influenza immunoglobulin M antibodies were elevated and serum anti-aquaporin 4 (AQP4) antibodies were undetectable. Spinal cord magnetic resonance imaging (MRI) displayed longitudinally extensive lesions in the thoracic cord. Brain MRI disclosed three subcortical lesions. The patient fulfilled the revised diagnostic criteria for NMO (2006). After methylprednisolone pulse therapy followed by oral administration of prednisolone, visual dysfunction, dysuria, limb dysesthesia and hyperreflexia were improved. Subsequently, she experienced no attacks for 3 years. This is the first case report of influenza A-associated NMO with such features of postinfectious NMO as a pediatric onset, monophasic course and anti-AQP4 antibody-seronegative status.

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Section: Discussionsupporting

confidence: 70%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Influenza-associated Monophasic Neuromyelitis Optica

et al. 2011

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“…The reasons for sex differences in the prevalence of autoreactive autoantibodies are not fully understood, but they might be of importance as NMO/SD possibly takes a more severe course in seropositive patients. 4,25,26 Sex differences in predisposition and course of auto immune diseases are, first and foremost, assumed to be based on differences in immunocompetence and immune reactivity. Hormonal factors, for example, differences in circulating sex hormones or changes in hormone receptor expression are supposed to be involved in sex differences in autoimmune diseases.…”

Section: Discussionmentioning

confidence: 99%

Influence of female sex and fertile age on neuromyelitis optica spectrum disorders

et al. 2016

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Background: Gender and age at onset are important epidemiological factors influencing prevalence, clinical presentation, and treatment response in autoimmune diseases. Objective: To evaluate the impact of female sex and fertile age on aquaporin-4-antibody (AQP4-ab) status, attack localization, and response to attack treatment in patients with neuromyelitis optica (NMO) and its spectrum disorders (neuromyelitis optica spectrum disorder (NMOSD)). Methods: Female-to-male ratios, diagnosis at last visit (NMO vs NMOSD), attack localization, attack treatment, and outcome were compared according to sex and age at disease or attack onset. Results: A total of 186 NMO/SD patients (82% female) were included. In AQP4-ab-positive patients, female predominance was most pronounced during fertile age (female-to-male ratio 23:1). Female patients were more likely to be positive for AQP4-abs (92% vs 55%; p < 0.001). Interval between onset and diagnosis of NMO/SD was longer in women than in men (mean 54 vs 27 months; p = 0.023). In women, attacks occurring ⩽40 years of age were more likely to show complete remission (p = 0.003) and better response to high-dose intravenous steroids (p = 0.005) compared to woman at >40 years. Conclusion: Our data suggest an influence of sex and age on susceptibility to AQP4-ab-positive NMO/ SD. Genetic and hormonal factors might contribute to pathophysiology of NMO/SD.

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Detection of aquaporin‐4 antibodies for patients with CNS inflammatory demyelinating diseases other than typical MS in Lithuania

Sakalauskaitė-Juodeikienė

Armalienė

Kizlaitienė

et al. 2018

Brain and Behavior

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ObjectivesNeuromyelitis optica (NMO) is frequently associated with aquaporin‐4 autoantibodies (AQP4‐Ab); however, studies of NMO in Lithuania are lacking. Therefore, the main objective of our study is to assess positivity for AQP4‐Ab in patients presenting with inflammatory demyelinating central nervous system (CNS) diseases other than typical multiple sclerosis (MS) in Lithuania.Materials and methodsData were collected from the two largest University hospitals in Lithuania. During the study period, there were 121 newly diagnosed typical MS cases, which were included in the MS registry database. After excluding these typical MS cases, we analyzed the remaining 29 cases of other CNS inflammatory demyelinating diseases, including atypical MS (n = 14), acute transverse myelitis, TM (n = 8), acute disseminated encephalomyelitis, ADEM (n = 3), clinically isolated syndrome, CIS (n = 2), atypical optic neuritis, ON (n = 1), and NMO (n = 1). We assessed positivity for AQP4‐Ab for the 29 patients and evaluated clinical, laboratory, and instrumental differences between AQP4‐Ab seropositive and AQP4‐Ab seronegative patient groups.ResultsAQP4‐Ab test was positive for three (10.3%) patients in our study, with initial diagnoses of atypical MS (n = 2) and ADEM (n = 1). One study patient was AQP4‐Ab negative despite being previously clinically diagnosed with NMO. There were no significant clinical, laboratory, or instrumental differences between the groups of AQP4‐Ab positive (3 [10.3%]) and negative (26 [89.7%]) patients.ConclusionsAQP4‐Ab test was positive for one‐tenth of patients with CNS inflammatory demyelinating diseases other than typical MS in our study. AQP4‐Ab testing is highly recommended for patients presenting with not only TM and ON but also an atypical course of MS and ADEM.

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Neuromyelitis optica and NMO-IgG in European pediatric patients

Cited by 53 publications

References 18 publications

Influenza-associated Monophasic Neuromyelitis Optica

Influenza-associated Monophasic Neuromyelitis Optica

Influence of female sex and fertile age on neuromyelitis optica spectrum disorders

Detection of aquaporin‐4 antibodies for patients with CNS inflammatory demyelinating diseases other than typical MS in Lithuania

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