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Background Leptospirosis, a zoonotic infection caused by Leptospira species, is a rare cause of neurological complications, including meningitis, encephalitis, and intracranial haemorrhage. Subdural haemorrhage (SDH) in leptospirosis is exceptionally rare, with fewer than ten cases reported worldwide. This case highlights an unusual presentation of neuroleptospirosis with atraumatic, bilateral SDH exhibiting irregular, loculated morphology, and a review of the limited literature shows a unique tendency of neuroleptospirosis (especially icterohaemorrhagic leptospirosis) to cause such morphologically unusual spontaneous subdural haematomas.Results A 60-year-old woman from rural New South Wales presented with fever, hypotension, renal failure, and cholestatic liver failure, necessitating continuous renal replacement therapy and vasopressor support. On hospital day 11, she developed multiple left hemibody focal motor seizures. Imaging revealed acute, spontaneous, bilateral asymmetrical subdural hematomas with an irregular, loculated appearance and midline shift. Serological testing confirmed acute leptospirosis (IgM and IgG positive). She improved well with ceftriaxone/doxycycline, antiepileptic medication treatments and supportive care for renal and hepatic dysfunction, and serial imaging demonstrated gradual improvement of the subdural haematomas. A review of literature revealed similar unusual morphologies in the few reported cases of neuroleptospirosis-associated subdural haemorrhages.Conclusion Neuroleptospirosis should be considered in the differential diagnosis of atraumatic SDH, especially in patients with epidemiological risk factors. Such SDH is often noticeably irregular in shape with internal heterogeneity, reflecting the systemic coagulopathy, endothelial dysfunction, vasculitis and direct bacterial invasion that pathogenetically underlie the haemorrhagic complications of leptospirosis. Early serological testing and multidisciplinary management are critical to optimising outcomes in this rare but severe complication.