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Inferior petrosal sinus sampling (IPSS) plays an important role in the assessment of patients with ACTH-dependent Cushing's syndrome. IPSS has been described as an innocuous, essentially risk-free procedure. Neurological complications associated with IPSS are rare. Previously reported neurological complications include brain stem infraction, pontine hemorrhage and subarachnoid hemorrhage. A 26-year-old woman was admitted to the hospital because of a moon face and a buffalo hump. A MRI showed a suspicious, but uncertain, area on the left side of the sella turcica. IPSS was performed as an appropriate means of providing more information about the side of the ACTH source. Immediately after the procedure, the woman experienced double vision, caused by a complete sixth nerve palsy of the left eye, together with a contralateral hemiparesis. From a clinical point of view, abducens nerve palsy and contralateral hemiparesis corresponded to Raymond-Cestan syndrome. I report a case of Raymond-Cestan syndrome, following petrosal sinus sampling in a female patient with ACTH-dependent Cushing's syndrome. To the best of our knowledge, this complication associated with petrosal sinus sampling has only been described in one previous case report. Although IPSS may provide essential information in the investigation of Cushing's syndrome, the possibility of very occasional but devastating complications should be remembered.