Objective: To report a case of inferior petrosal sinus thrombosisassociated with malposition of central venous catheterization (CVC). Clinical Presentation and Intervention: A 5-month-old boy was admitted to Şifa University Hospital because of pneumonia. When exophthalmos occurred in the right eye, he was referred to Ege University Hospital. Cranial magnetic resonance imaging and magnetic resonance venography confirmed that the catheter in the right inferior petrosal sinus caused the thrombosis. The catheter was extracted and anticoagulant treatment was started. Conclusion: In this case report, malpositioning of the CVC was the cause of the thrombosis. To minimize such complications, catheterization should be done with the supervision of an expert and postprocedure radiography should also be performed.