RATIONALE: Brucella neuropathy is a rare clinical condition, particularly in nonendemic areas, where it often presents with nonspecific symptoms such as fever and headaches, leading to frequent misdiagnoses. In these regions, Brucella antibodies are not routinely tested, and the positive rate of blood cultures is relatively low during the early stage of the disease. In addition, the low culture-positive rate of cerebrospinal fluid (CSF) means that many neurobrucellosis diagnoses rely on peripheral blood cultures or Brucella antibodies, which is not rigorous. This is problematic because the treatment of central nervous system brucellosis differs significantly from that of other types of brucellosis. The purpose of this study is to provide a method for the early diagnosis of Brucella neuropathy in nonendemic areas. We present a case of early-stage neurobrucellosis diagnosed using metagenomic next-generation sequencing (mNGS) of CSF. This approach helps avoid delaying diagnosis in the early stage in nonepidemic areas, potentially reducing the duration before diagnosis, which is of great significance for timely and appropriate treatment. PATIENT CONCERNS: The patient had fever, mild headache, and a slight increase in CSF leukocytes. DIAGNOSES: CSF mNGS detected Brucella, which was later confirmed by serum Brucella antibody testing. INTERVENTIONS: The patient was treated with rifampicin, doxycycline, and ceftriaxone. OUTCOMES: The patient experienced significant relief from the headache, and the fever did not recur. Subsequent examinations revealed no abnormalities in the CSF leukocytes or mNGS results. LESSONS: We reviewed 10 articles on brucellosis diagnosed using mNGS, including 7 articles on neurobrucellosis (10 cases). Our review highlights the sensitivity of mNGS as a powerful tool for early detection of neurobrucellosis in the CSF. The common symptoms include fever and headache, with brain magnetic resonance imaging detecting lesions in most cases. CSF mNGS results varied, with only a few positive Brucella cultures or antibody tests.