International Journal of Case Reports and Images (IJCRI) is an international, peer reviewed, monthly, open access, online journal, publishing high-quality, articles in all areas of basic medical sciences and clinical specialties.Aim of IJCRI is to encourage the publication of new information by providing a platform for reporting of unique, unusual and rare cases which enhance understanding of disease process, its diagnosis, management and clinico-pathologic correlations. Clozapine is an atypical antipsychotic, which is associated with an increased risk of neutropenia. Given that the signs and symptoms of infection in neutropenic patients are often subtle or absent because of the lack of an appropriate inflammatory response, fever may constitute the sole indicator of a serious underlying infection. Unfortunately, data about management of patients with neutropenic fever secondary to clozapine are scarce. Consequently, the entire management of this syndrome is based on extrapolation of data from the experience with cancer patients with neutropenia secondary to cytotoxic chemotherapy. Case Report: Herein, we describe the management of a neutropenic fever case complicated with septic shock and acute respiratory failure in an 57-year-old Caucasian female with the diagnosis of schizophrenia and type-2 diabetes mellitus, who was being treated with clozapine. The patient rapidly developed cardiorespiratory collapse requiring mechanical ventilation and vasoactive drugs few minutes after arrival at hospital. Profound neutropenia (absolute neutrophil count 60 cells/mm3) and lobar pneumonia were diagnosed. Broad-spectrum antimicrobial therapy with piperacillin-tazobactam plus vancomycin and supportive intensive care were promptly implemented. The clozapine-induced neutropenia was managed with filgrastim. Pseudomonas aeruginosa was isolated from the tracheal aspirate and blood cultures. After a total length of hospital stay of 44 days, the patient was discharged home. Conclusion: Neutropenic fever is a serious complication of clozapine treatment. Prompt administration of empiric broad-spectrum antibiotics and supportive care are required to avoid the high levels of mortality associated with this syndrome.