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Kartagener’s syndrome is a rare genetic disorder of primary ciliary dyskinesia characterized by the triad of bronchiectasis, sinusitis, and situs inversus. There is limited data on the role of Kartagener’s syndrome leading to psychotic manifestations. We would like to discuss the case of a 26-year-old male, who presented to us with psychotic symptoms of 4-year duration. Upon detailed physical examination and pertinent investigations, he was diagnosed to be having Kartagener’s syndrome. He was treated with antipsychotics and benzodiazepines. This case highlights the importance of a thorough physical examination and eliciting medical history, as it may lead to the finding of various syndromes that have been said to be associated with schizophrenia, thereby contributing to its genetic hypothesis. The neurological, biochemical, and structural malformations seen in Kartagener’s syndrome may also lead to psychotic manifestations. They could be genetically linked to loci contributing to an increased risk of psychosis. [ABSTRACT FROM AUTHOR]