Abstract: OBJECTIVE To improve the clinical understanding of suspected intracranial infection of Mycobacterium chelonae. METHODS The clinical data of one case of suspected intracranial infection caused by Mycobacterium chelonae were retrospectively analyzed, and reviewed the literature. RESULTS The patient suffered from surgical incision infection and secondary intracranial infection after cerebral hemorrhage operation. The detection of cerebrospinal fluid by the metagenomics next-generation sequencing showed that the suspected pathogen was Mycobacterium chelonae. Combined with the etiological results and pharmacokinetic characteristics, the condition was improved after treatment with linezolid. CONCLUSION Mycobacterium chelonae has become an important nosocomial infection pathogen secondary to skin invasive operation. It should pay more attention to clinical and infection control, take early identification and control measures to avoid causing disseminated infection, especially intracranial infection which is difficult to treat, and should be alert to potential hospital transmission risk.