Further, he complained of persistent hiccough, vomiting and nausea that began a couple of days prior to the progressive weakness of his limbs made an appearance. yet been reported. We herein report a novel case of a previously healthy man who presented with a clinical picture of bouts of vomiting and hiccoughs (area postrema syndrome), which rapidly evolved to acute LETM, all following SARS-CoV-2 infection. He was finally diagnosed to be a case of seropositive NMOSD which presented as area postrema syndrome. The response to immunomodulatory drugs was excellent. strong class=”kwd-title” Keywords: Neuromyelitis optica, NMOSD, COVID-19, SARS-CoV-2, Immune-mediated, Longitudinally extensive transverse myelitis Graphical abstract Open in a separate window 1.?Introduction Neurological manifestations of coronavirus infectious disease of 2019 (COVID-19) have been ever evolving and the spectrum of neuraxial involvement is broadening.(Roy et al., 2020) Albeit it may affect any area of the neural axis, the involvement of the spinal cord is rare compared to that of the brain and of the peripheral nervous system.(Ghosh et al., 2020a; Ghosh et al., 2020c; Ghosh et al., 2020d; Gutirrez-Ortiz et al., 2020; Rbano-Surez et al., 2020; Roy Azelaic acid et al., 2020) SARS-CoV-2 has immense propensity to dysregulate the host immune system resulting in generation of various autoantibodies, which are turning out to be extremely pathogenic (Finsterer et al., 2020; Gutirrez-Ortiz et al., 2020; Zhang et al., 2020; Zhou et al., 2020). Neuromyelitis optica spectrum disorder (NMOSD) is a disabling autoimmune astrocytopathic channelopathy, characterized by the presence of pathogenic antibodies SERPINA3 to aquaporin-4 (AQP-4) water channels, expressed on astrocytic foot processes at blood brain barrier, subpial and subependymal regions.(Fujihara, 2019; Ghosh et al., 2020b; Lucchinetti et al., 2014) Long segments of spinal cord inflammation (myelitis), severe optic neuritis, and/or bouts of intractable vomiting and hiccoughs (area postrema syndrome) are the classic forms of presentation.(Fujihara, 2019; Lucchinetti et al., 2014) From a pathological point of view, NMOSD mainly encompasses AQP-4-antibody-positive astrocytopathy and myelin oligodendrocyte glycoprotein (MOG)-antibody-positive inflammatory demyelinating disease.(Fujihara, 2019, Lucchinetti Azelaic acid et al., 2014) Several viral infections including HIV, influenza virus, varicella zoster virus, and Epstein Barr virus, among others, have been alleged to trigger NMOSD in both immunocompetent and immunocompromised individuals.(Lana-Peixoto et al., 2018; Machado et al., 2015; Mathew et al., 2019; Sellner et al., 2010; Tran et al., 2007) It has recently reported a case of a young man presenting with bilateral severe optic neuritis and myelitis, determined to be simultaneously SARS-CoV-2 and MOG IgG antibody positive, i.e. a variant of NMOSD.(Zhou et al., 2020) However, AQP-4-antibody-seropositive NMOSD following SARS-CoV-2 infection had not yet been reported. We herein report a novel case of a previously healthy man who presented with a clinical picture of bouts of vomiting and hiccoughs (area postrema syndrome), which rapidly evolved to acute LETM, all following SARS-CoV-2 infection. He was finally diagnosed to be a case of seropositive NMOSD which presented as area postrema syndrome. The response to immunomodulatory drugs was excellent. 2.?Case report A previously healthy Asian-Indian 20-year-old man was admitted to the emergency department with rapidly progressive weakness and decreased sensation of all four Azelaic acid limbs, urinary retention, and constipation. His past medical history was unremarkable. Ten days prior to his admission, he had developed a mild fever and non-productive cough for 3?days, which resolved spontaneously. Five days after this mild clinical picture, he developed rapidly progressive weakness of his lower limbs, followed by weakness of both upper limbs within 12?h. Further, he complained of persistent hiccough, nausea and vomiting that started a few days before the progressive weakness of his limbs appeared. On the day of admission, on getting up from sleep, he found himself unable to get up from lying down position on his own. He also noticed reduced sensation in all 4 limbs and over his torso to all modalities for the same duration. He also complained of dull aching, deep seated, non-radiating pain over his nape of neck, that appeared the day before he developed weakness. Weakness was also associated with difficulty in voiding, pain and fullness in his lower abdomen, and constipation. Azelaic acid There was no history suggestive of any similar previous episode, cranial nerve involvement, headache, eye pain, visual symptoms, diplopia, swallowing difficulty, seizures, loss of consciousness, vaccination, trauma, skin rash, joint pain, photosensitivity, orogenital ulcers, dry eyes, dry mouth, band/girdle-like sensation, root pain, thinning or twitching of muscles, flexor spasms, and respiratory distress. Family history was non-contributory. On physical examination, he was fully awake, conscious, afebrile, and normotensive (recorded supine blood pressure was 120/70?mm of Hg). His respiratory rate was 16/min and oxygen saturation of 96% in room air. Neurological examination revealed intact cognitive and cranial nerve functions. Motor system examination revealed asymmetric flaccid hypo-to-areflexic quadriparesis (muscle power.