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Though substance\u2010induced psychotic symptoms have been mentioned in the literature, the potential long\u2010term negative neuropsychiatric effects related to its use have not been well established.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods and Results<\/jats:title><jats:p>This is a patient case report of a young man (N\u2009=\u20091) who initially presented with acute neurological symptoms requiring hospitalization due to heavy nitrous oxide inhalant use, and went on to present with symptoms concerning for a primary psychotic illness over multiple inpatient admissions. He provided both verbal and written consent to share his story for this case report.<\/jats:p><\/jats:sec><jats:sec><jats:title>Discussion and Conclusions<\/jats:title><jats:p>It is important to consider nitrous oxide use as a possible contributing factor to the development of primary psychotic illness.<\/jats:p><\/jats:sec><jats:sec><jats:title>Scientific Significance<\/jats:title><jats:p>Current literature suggests that psychosis associated with nitrous oxide use is transient and resolves upon cessation and treatment of vitamin B<jats:sub>12<\/jats:sub> deficiency. Here, we present a patient with risk factors for psychotic illness developing psychotic illness following extensive nitrous oxide use. This report offers a unique perspective of longitudinal follow\u2010up (often not provided with reports on this topic), and illustrates the importance of healthcare providers inquiring about nitrous oxide abuse in patients presenting with early psychotic symptoms. 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