Intractable nausea, vomiting, and hiccups are characteristic symptoms associated with neuromyelitis optica spectrum disorder (NMOSD) as the result of immune-mediated lesions affecting the area postrema and medullary floor of the fourth ventricle. Reports of paroxysmal sneezing as part of the NMOSD phenotype are rare. We present a case of a patient who developed symptoms of NMOSD associated with prominent paroxysmal sneezing as a heralding symptom. The precise location of the human sneeze center has not been identified; this case provides further evidence for its location in the dorsolateral medulla as previously proposed.1
Case report.
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A 55-year-old woman with a medical history of hypothyroidism developed hypersomnolence (sleeping up to 22 hours daily). Soon after, she began to experience severe nausea and vomiting, intractable hiccups, and then violent paroxysmal sneezing lasting several minutes that would awaken her from sleep. The sneezing spells lasted several days. During each paroxysm, she sneezed several times, sometimes in a stereotypic cyclic fashion alternating with bouts of hiccups. She then developed urinary retention, vertigo, and diplopia, followed by new-onset atrial fibrillation and ataxia.
The patient had no history of allergies, pulmonary disease, or sinusitis and was a lifelong nonsmoker. Otolaryngologic and pulmonary examinations were unremarkable. Neurologic examination showed oscillopsia on primary gaze, multidirectional nystagmus, and gait ataxia. Laboratory analysis showed normal blood count and liver, kidney, and thyroid function; vitamin levels, autoimmune screens, and paraneoplastic antibodies were negative. CSF analysis revealed a white blood cell count of 19 cells/μL (93% lymphocytes) with normal flow cytometry, glucose, and protein and no oligoclonal bands. MRI (figure) showed fluid-attenuated inversion recovery hyperintensities in the hypothalamus, fourth ventricle, periependymal, periventricular, and periaqueductal regions. Prominent involvement of the dorsolateral aspect of the medulla was noted bilaterally.
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Methylprednisolone 1 g was administered for 5 days; significant improvement of the nausea, vomiting, hiccups, and paroxysmal sneezing was noted. Given that the ataxia, oscillopsia, and diplopia were still prominent, she underwent plasmapheresis with good clinical response.
Aquaporin-4 immunoglobulin G (AQP4-IgG) was identified via cell-based assay in the serum obtained on admission; it was negative in the CSF.
She had no further spells of paroxysmal sneezing and has remained clinically stable on rituximab monotherapy for 16 months.
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