In recent times, some research has focused on the study of potential treatments for cystic fibrosis (CF), such as cystic fibrosis transmembrane conductance regulator (CFTR) modulators. These treatments have been reported to produce neuropsychiatric symptoms in a few patients, even though there is still no clear correlation nor underlying mechanism proposed. We present the case of a 23-year-old woman with CF and no previous psychiatric history who was admitted to our inpatient psychiatric unit presenting a wide range of neuropsychiatric symptoms, such as disorganized speech, bizarre poses or persecutory delusional ideation, after going under CFTR modulators treatment. After several diagnostic tests, other possible organic causes were ruled out. Multiple antipsychotic treatments were tested during her admission, with poor tolerance and scarce response. Finally, symptomatic remission was only observed after electroconvulsive therapy was initiated. The final diagnostic hypothesis was unspecified psychosis. This case highlights the relevance of considering the possibility of neuropsychiatric symptoms appearing in patients under CFTR modulators treatment.

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