Clinical features and prognosis of late-onset neuromyelitis optica spectrum disorders in a Latin American cohort

Contentti, Edgar Carnero; Marques, Vanessa Daccach; Castillo, Ibis Soto de; Tkachuk, Verónica; Bustos, Ariel; Castillo, M.C.; Cristiano, Edgardo; Serva, Gabriel Braga Diégues; Santos, Antônio Carlos dos; Finkelsteyn, Ana Mariel; López, Pablo A.; Patrucco, Liliana; Molina, Omaira; Pettinicchi, Juan Pablo; Toneguzzo, Vanesa; Caride, Alejandro; Rojas, Juan Ignacio

doi:10.1007/s00415-020-09699-2

Cited by 42 publications

(33 citation statements)

References 27 publications

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“…5,[7][8][9][10][11][12][13] In our LO-NMOSD-ON cohort, the mean age at ON onset was approximately 65 years, which was slightly higher than the ages reported in previous studies (56-59.4 years). 5,[7][8][9][10][11][12][13] However, the mean age at ON onset was 32.6 years in our EO-NMOSD-ON cohort, which is comparable with the typical age at NMOSD onset in previous studies (32-41 years). [2][3][4] Moreover, our previously published article, which studied prognostic factors for visual outcomes following the first episode of ON in each affected eye in overall age group of NMOSD-ON patients, showed that a median age at NMOSD-ON onset was 36 years (range 4-84 years).…”

Section: Demographic Datacontrasting

confidence: 72%

“…This was similar to the positive correlations between age at disease onset and Expanded Disability Status Scale score observed in previous studies. 5,8 This finding implies that age at disease onset may affect disability outcomes; specifically, older age at disease onset may be associated with worse disability outcomes.…”

Section: Long-term Visual Outcomesmentioning

confidence: 98%

“…These findings were comparable with the results in several Asian and Caucasian population-based studies that found worse Expanded Disability Status Scale scores at the last visit, as well as greater motor disability and wheelchair dependence, in patients with LO-NMOSD than in patients with EO-NMOSD. 5,8,13 Additionally, one multicenter study found more frequent severe visual disability (ie, VA worse than or equal to 1.0 logMAR) in patients with LO-NMOSD than in patients with EO-NMOSD. 12 Here, we found a positive correlation between age at ON onset of each eye and final VA (logMAR).…”

Section: Long-term Visual Outcomesmentioning

confidence: 99%

“…[2][3][4] Older age at NMOSD onset is reportedly associated with worse disease prognosis, higher mortality rate, and greater susceptibility to disability. 5 When it occurs in older patients (age at NMOSD onset ≥ 50 years), the disorder is known as late-onset neuromyelitis optica spectrum disorder (LO-NMOSD). Notably, one retrospective study demonstrated a relationship between age at ON onset and final visual outcomes; later onset of neuromyelitis optica spectrum disorder-related optic neuritis (NMOSD-ON) was associated with worse final visual outcome.…”

Section: Introductionmentioning

confidence: 99%

“…5,[7][8][9][10][11][12][13] Some have compared these aspects with early-onset NMOSD (EO-NMOSD; age at NMOSD onset < 50 years). 5,[10][11][12][13] To the best of our knowledge, no studies have been performed to specifically compare clinical characteristics and long-term visual outcomes between patients with late-onset neuromyelitis optica spectrum disorder-related optic neuritis (LO-NMOSD-ON) and those with early-onset neuromyelitis optica spectrum disorder-related optic neuritis (EO-NMOSD-ON). Therefore, this study was performed to comprehensively compare demographic data, clinical and radiological characteristics, treatments, and long-term visual outcomes between patients with LO-NMOSD-ON and patients with EO-NMOSD-ON.…”

Section: Introductionmentioning

confidence: 99%

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Comparison of Early- and Late-Onset NMOSD-Related Optic Neuritis in Thai Patients: Clinical Characteristics and Long-Term Visual Outcomes

Thongmee¹,

Narongkhananukul²,

Padungkiatsagul³

et al. 2021

OPTH

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Objective To compare demographic data, clinical and radiological characteristics, treatments, and long-term visual outcomes between patients with late-onset neuromyelitis optica spectrum disorder-related optic neuritis (LO-NMOSD-ON) (age at onset ≥ 50 years) and patients with early-onset neuromyelitis optica spectrum disorder-related optic neuritis (EO-NMOSD-ON) (age at onset < 50 years). Patients and Methods This retrospective study included 47 patients (69 eyes) who were diagnosed with neuromyelitis optica spectrum disorder-related optic neuritis (NMOSD-ON) over a 12-year period. There were 14 patients (21 eyes) and 33 patients (48 eyes) in the LO-NMOSD-ON and EO-NMOSD-ON groups, respectively. Results LO-NMOSD-ON–affected eyes exhibited significantly worse median nadir visual acuity (VA) at optic neuritis (ON) onset, compared with EO-NMOSD-ON–affected eyes (2.7 logMAR (range 2.6–2.9 logMAR) vs 1.95 logMAR (range 0.4–2.9 logMAR); p = 0.03). Similarly, 100% of LO-NMOSD-ON–affected eyes demonstrated a nadir VA of worse than or equal to 1.0 logMAR, compared with 62.5% of EO-NMOSD-ON–affected eyes ( p = 0.03). LO-NMOSD-ON–affected eyes had a worse median final VA, compared with EO-NMOSD-ON–affected eyes (1.3 logMAR (range 0–2.9 logMAR) vs 0.3 logMAR (range 0–2.9 logMAR); adjusted p = 0.037). LO-NMOSD-ON–affected eyes more frequently exhibited a final VA of worse than or equal to 1.0 logMAR, compared with EO-NMOSD-ON–affected eyes (57.1% vs 27.0%; adjusted p = 0.039). A positive correlation was observed between age at ON onset of each eye and the final VA (logMAR) (Spearman r = 0.34, p = 0.0075). The remaining parameters did not significantly differ between the two groups. Conclusion Patients with LO-NMOSD-ON had significantly worse nadir VA at ON onset and significantly worse final VA, relative to patients with EO-NMOSD-ON. Age at ON onset of each eye was positively correlated with final VA (logMAR). Despite the difference in common age at onset, NMOSD-ON should be included in the differential diagnosis of late-onset acute to subacute optic neuropathy, along with ischemic optic neuropathy.

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Section: Demographic Datacontrasting

confidence: 72%

Section: Long-term Visual Outcomesmentioning

confidence: 98%

Section: Long-term Visual Outcomesmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Comparison of Early- and Late-Onset NMOSD-Related Optic Neuritis in Thai Patients: Clinical Characteristics and Long-Term Visual Outcomes

Thongmee¹,

Narongkhananukul²,

Padungkiatsagul³

et al. 2021

OPTH

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Astrocyte‐Derived Exosomes Contribute to Pathologies of Neuromyelitis Optica Spectrum Disorder in Rodent Model

Xie

Chen

Wang

et al. 2023

Annals of Neurology

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Objective: Neuromyelitis optica spectrum disorder (NMOSD) is an inflammatory demyelinating disease that leads to severe disability. A large proportion of NMOSD patients are seropositive for aquaporin-4 autoantibodies (AQP4-IgG, named as NMO-IgG) targeting AQP4, which is selectively expressed on astrocytes in the central nervous system. This study tests the hypothesis that in response to NMO-IgG, the pathogenic astrocyte-derived exosomes are released and injure the neighboring cells. Methods: IgG purified from serum of either NMOSD patients or healthy controls was used to generate astrocytederived exosomes (AST-Exos NMO vs AST-Exos CON ) in cultured rat astrocytes. The exosomes were respectively delivered to cultured rat oligodendrocytes in vitro, tissue culture of rat optic nerve ex vivo, and rat optic nerve in vivo to evaluate the pathogenic roles of AST-Exos NMO . The microRNA (miRNA) sequencing of AST-Exos and verification were performed to identify the key pathogenic miRNA. The custom-designed adeno-associated virus (AAV) antagonizing the key miRNA was evaluated for its therapeutic effects in vivo. Moreover, the serum levels of the key exosomal miRNA were measured between NMOSD patients and healthy controls. Results: AST-Exos NMO led to notable demyelination in both cultured oligodendrocytes and optic nerve tissue. Exosomal miR-129-2-3p was identified as the key miRNA mediating the demyelinating pathogenesis via downstream target gene SMAD3. AAV antagonizing miR-129-2-3p protected against demyelination in an NMOSD rodent model. The serum exosomal miR-129-2-3p level was significantly elevated in NMOSD patients and correlated with disease severity. Interpretation: Astrocytes targeted by NMO-IgG release pathogenic exosomes that could potentially be used as therapeutic targets or disease monitoring biomarkers in NMOSD.

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Sex ratio and age of onset in AQP4 antibody-associated NMOSD: a review and meta-analysis

Arnett,

Chew,

Leitner

et al. 2024

J Neurol

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Background Aquaporin-4 (AQP4) antibody-associated neuromyelitis optica spectrum disorder (NMOSD) is an antibody-mediated inflammatory disease of the central nervous system. We have undertaken a systematic review and meta-analysis to ascertain the sex ratio and mean age of onset for AQP4 antibody associated NMOSD. We have also explored factors that impact on these demographic data. Methods A systematic search of databases was conducted according to the PRISMA guidelines. Articles reporting sex distribution and age of onset for AQP4 antibody-associated NMSOD were reviewed. An initially inclusive approach involving exploration with regression meta-analysis was followed by an analysis of just AQP4 antibody positive cases. Results A total of 528 articles were screened to yield 89 articles covering 19,415 individuals from 88 population samples. The female:male sex ratio was significantly influenced by the proportion of AQP4 antibody positive cases in the samples studied (p < 0.001). For AQP4 antibody-positive cases the overall estimate of the sex ratio was 8.89 (95% CI 7.78–10.15). For paediatric populations the estimate was 5.68 (95% CI 4.01–8.03) and for late-onset cases, it was 5.48 (95% CI 4.10–7.33). The mean age of onset was significantly associated with the mean life expectancy of the population sampled (p < 0.001). The mean age of onset for AQP4 antibody-positive cases in long-lived populations was 41.7 years versus 33.3 years in the remainder. Conclusions The female:male sex ratio and the mean age of onset of AQP4 antibody-associated NMOSD are significantly higher than MS. The sex ratio increases with the proportion of cases that are positive for AQP4 antibodies and the mean age of onset increases with population life expectancy.

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Clinical features and prognosis of late-onset neuromyelitis optica spectrum disorders in a Latin American cohort

Cited by 42 publications

References 27 publications

Comparison of Early- and Late-Onset NMOSD-Related Optic Neuritis in Thai Patients: Clinical Characteristics and Long-Term Visual Outcomes

Comparison of Early- and Late-Onset NMOSD-Related Optic Neuritis in Thai Patients: Clinical Characteristics and Long-Term Visual Outcomes

Astrocyte‐Derived Exosomes Contribute to Pathologies of Neuromyelitis Optica Spectrum Disorder in Rodent Model

Sex ratio and age of onset in AQP4 antibody-associated NMOSD: a review and meta-analysis

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