Neurosurgical devices provide clinical benefits that are undermined by the incidence of device-related infections. This study examined the microbiological and clinical characteristics of central nervous system (CNS) infections associated with cerebrospinal fluid (CSF) shunts and drains in neurosurgery patients during August 2022–July 2024. Upon suspicion of CNS infection, CSF samples were submitted for cytology, biochemical analysis, and culture. Identification and susceptibility testing were performed by VITEK-2 and VITEK-MS PRIME (bioMérieux); susceptibility to colistin was determined by broth microdilution (Bruker). The results were interpreted according to EUCAST guidelines. An infection definition was based on IDSA’s guidelines, and the outcome was considered positive upon successful therapeutic intervention. Out of 825 CSFs, 178 showed microbial growth in culture; they corresponded to 102 patients. Neurosurgical devices were implanted in 68 patients and, among them, 29 presented with a CNS infection. The infections attributed to drains and shunts were 72% versus 28%, respectively. Polymicrobial or consecutive infections occurred in 6/29 patients. Overall, 40 pathogens were isolated. The predominant causative agents were Acinetobacter baumannii (40%), Klebsiella pneumoniae (12.5%), coagulase-negative-Staphylococci (12.5%), and Pseudomonas aeruginosa (10%). Neurosurgical devices are easily contaminated and may cause CNS infections, regardless of the device type. The infections’ outcome is multifactorial; timely diagnosis and intervention are of utmost importance.

While primarily described in children, adult-onset Langerhans cell histiocytosis (LCH) has been reported, albeit infrequently. In the present scenario, we unveil a unique case of adult-onset LCH in an HIV-infected individual. After the diagnosis was made, the patient was successfully treated and demonstrated total disease remission. This case illustrates the diagnostic challenge that rare clinical entities such as LCH pose, especially in the context of an untreated HIV infection. Furthermore, the complexity of treating adult-onset Langerhans cell histiocytosis in an HIV-positive patient is highlighted, with emphasis given on a multidisciplinary approach.