Dados do Trabalho


Título

Prediction model for the differential diagnosis of MOG-IgG associated disease and Multiple Sclerosis at first Central Nervous System demyelinating episode in paediatric patients

Resumo

Introduction: Anti-myelin oligodendrocyte associated disease (MOGAD) is more frequent in pediatric patients than in adults. In international cohorts about 30% of pediatric patients with central nervous system inflammatory diseases have MOGAD. Since MOG-IgG testing is not yet widely available worldwide, the identification of patients that might benefit from testing at first presentation is recommended specially in low-income countries. Also, the correct differential diagnosis of MOGAD and multiple sclerosis (MS) prevents unnecessary and potentially harmful treatments.
Objective: Our aim is to propose a predictive score based on the clinical characteristics at first clinical attack for the differential diagnosis between MOGAD and MS in pediatric patients.
Methods: This is a nested case-control study of patients ≤18 years of a Brazilian pediatric cohort of 6 reference centres in neuroimmunology in 5 states. We selected patients that tested positive for MOG-IgG at the incidental inflammatory attack and those diagnosed with MS at the first year of follow-up in the study. The clinical characteristics at first presentation were evaluated identifying those more strongly associated with MOGAD. Maximum likelihood or exact logistic regressions were used to investigate associations of clinical characteristics and the diagnosis of MOGAD or MS. The cut-off for the predictive score was estimated based on the receiver operating characteristic (ROC) analysis.
Results: We found that younger age at presentation (1 point), male sex (1 point), bilateral optic neuritis (1 point) and multifocal presentation with encephalopathy or isolated optic neuritis (1 point) were associated with MOGAD and contributed for the composite score. Having ≥ 2 points in our proposed clinical composite score has 80% sensitivity (95% CI, 0.56-0.93) and 66% specificity (95% CI, 0.35-0.89) for the diagnosis of MOGAD.
Conclusions: Combined clinical and demographic characteristics at first attack might be used to guide serologic testing for MOG-IgG and help to differentiate pediatric MS from MOGAD. The score might support treatment decisions and optimize the use of health resources.

Palavras Chave

MOG-IgG associated disease, pediatric multiple sclerosis, optic neuritis

Área

Neuroimunologia

Autores

Bruna Klein da Costa, Rafael Canani Sommer, Fernanda Silveira de Quadros, Marlise de Castro Ribeiro, Renata Barbosa Paolilo, José Albino da Paz, Manuela de Oliveira Fragomeni, Vanessa Fragoso, Maria Lucia Brito, Douglas Kazutoshi Sato