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Problem: The association of pediatric‐onset multiple sclerosis (POMS) and psychosis remains rare and unclear in the literature. We aim to elucidate the association between POMS and psychosis across case reports. Methods: We report a case of schizophrenia (SCZ) revealing POMS. We conducted a systematic review of case reports adhered to PRISMA 2020. We searched PubMed, Google Scholar, Web of Science, and Cochrane for case reports describing psychoses in POMS cases. Findings: We presented a 21‐year‐old male who developed psychotic symptoms at 16 and was diagnosed with SCZ at 18. Regarding the treatment‐resistant SCZ, neurological assessment led to POMS. The patient had a favorable outcome after POMS management. Moreover, our systematic review of nine cases revealed that most psychoses began before or concomitant to the POMS diagnosis, highlighting a delay in identifying POMS. SCZ and BP were the most common types of psychosis observed. We observed a predominance of males with a median age at psychosis onset was 15 years (12.5–16.5 years). Treatment‐resistant SCZ was noted in one study along aside with our case. All cases had a good evolution after POMS management. Conclusion: Our study suggested a critical link between POMS and psychosis emphasizing the need for thorough evaluation, early diagnosis, and prompt management. This association highlights the importance of recognizing psychotic symptoms as potential indicators of POMS, particularly in males. [ABSTRACT FROM AUTHOR]