Introduction Only 12 cases of gallbladder (GB) cancer associated with leptomeningeal carcinomatosis (LMC) have been reported so far. Herein, we report the first known case of LMC originating from GB cancer after curative resection and discuss the risk factors of LMC associated with GB cancer. Case Presentation An 85-year-old Japanese woman presented with vomiting and impaired awareness 2 years after curative extended cholecystectomy for GB cancer . Computed tomography showed hydronephrosis of the right kidney and ureteral thickening. Magnetic resonance imaging revealed areas of hyperintense reflecting lesions along the cerebral sulci, suggesting meningitis. A spinal tap showed an elevated cerebrospinal fluid pressure of > 270 mmH 2 O, and cytological examination of the spinal fluid revealed the presence of adenocarcinoma cells. The patient was diagnosed with retroperitoneal metastasis and LMC originating from GB cancer. The patient was given palliative care and died 4 weeks after the onset of symptoms. Conclusion The findings of this study show that LMC could occur even after curative resection of GB cancer and should be considered when patients present with neurological symptoms. Retroperitoneal metastases and poorly differentiated tumors are possible risk factors of LMC originating from GB cancer.

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