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PURPOSE Alert intensivists about the diagnostic pitfalls arising from hyperammonemia due to Ureaplasma infections in post-transplant patients. MATERIALS AND METHODS Clinical observation of one patient. CASE REPORT A 65-year-old female with a medical history of semi-recent kidney transplant was admitted to the Intensive Care Unit for refractory status epilepticus. There were no lesions on brain imaging. Bacterial cultures and viral PCR of cerebrospinal fluid were negative. The first blood ammonia level measured on day 2 was 13 times the normal level, but biological liver tests were normal. The persistence of elevated ammonia levels led to the initiation of symptomatic ammonia lowering-treatments and continuous renal replacement therapy, which led to its decrease without normalization. An Ureaplasma spp infection was then diagnosed. Levofloxacin and doxycyline were administered resulting in normalization of ammonia levels within 48 h. However repeat MRI showed diffuse cortical cytotoxic edema and the patient remained in a minimally conscious state. She eventually died 4 months later from a recurrent infection. CONCLUSION Ureaplasma infection must be suspected in cases of neurological symptoms associated with hyperammonemia without liver failure, following an organ transplant. Only urgent treatment could improve the prognosis and prevent severe neurological damage or death.