ObjectiveTo assess the prevalence of dual positive NMOSD and outline its clinical phenotype.BackgroundNeuromyelitis Optica Spectrum Disorders (NMOSD) is an autoimmune syndrome that is frequently positive for Aquaporin 4 (AQP4) IgG or Myelin Oligodendrocyte Glycoproteins (MOG) IgG. However, dual positivity to both is rare.Design/MethodsThis is a retrospective cross-sectional study conducted at a tertiary healthcare center in South Asia between August 2018 and November 2021. The serum and/or CSF samples of suspected cases of NMOSD were tested for both AQP4-IgG and MOG-IgG using an Indirect immunofluorescence test on transfected cells.ResultsDuring the study period, 1935 cases of NMOSD were tested for both antibodies- 65 patients (3.36%; 57 females and 8 males) tested positive for AQP4-IgG, 217 patients (11.23%; 122 females and 95 males) tested positive for MOG-IgG and 3 patients (0.15%; 2 females and 1 male) showed dual positivity. There was a strong female preponderance in all three groups (87.69%, 56.22% and 66.66% respectively). This study identified 3 patients with dual positivity. The first patient (42 years, Male) presented with area postrema syndrome initially and subsequently relapsed by developing right-sided numbness of the temporal area and limbs during which he tested dual positive. The second patient (27 years, Female) presented with bilateral optic neuritis (left >right) initially and subsequently relapsed following an episode of a seizure with left-sided hemiplegia and right-sided facial deviation. The third patient (25 years, Female) initially presented with acute bilateral optic neuritis and later developed left-sided hemiplegia post-recovery at which point she tested dual positive. Management using methylprednisolone was ineffective for all three patients, however, plasmapheresis and/or periodic rituximab injections produced an excellent response.ConclusionsOur study reports that the prevalence of dual positive NMOSD is 0.15% and its clinical phenotype is more similar to NMO rather than MOG- associated disease.

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Steroids have been used for a wide spectrum of indications in clinical practice. The potential benefits of steroids outweigh the complications in most settings. Steroids have been used over the counter in our country for allergy and arthritis. Here we present a case of a young girl who presented with headache, tinnitus, vomiting, and papilledema, visual field defect in perimetry with elevated IOP, presumed secondary to intranasal steroid use for allergic rhinitis. There are case reports suggesting a causal relationship between steroids and benign intracranial hypertension. Here patient on follow-up had significant improvement on stopping steroid spray. This case report is to highlight the need for awareness of the possibility of benign intracranial hypertension in patients who are on inhalational steroids.

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