The emissary veins, like the petrosquamosal sinus (PSS), are residual valveless veins, which connect the intracranial dural venous sinuses and the extracranial venous system. Rarely, they may cause pulsatile tinnitus (PT). A 22-year-old woman developed in the first week of puerperium worsening headaches, vomiting, and diplopia, and the accentuation of a PT in the left ear that she presented for eight years. The clinical examination findings nine days after delivery were unremarkable, with the exception of a left sixth nerve palsy, and a peculiar sensibility of the left temporo-mandibular joint. High-resolution computed tomography (HRCT) revealed an osseous canal in the air cells of the left temporal bone compatible with a PSS. CT and magnetic resonance (MR) imaging÷MR-venogram detected signs of thrombosis of the superior sagital sinus, and of the left lateral sinus. Laboratory tests revealed severe inherited thrombophilia. We used antithrombotic therapy (body weight-adjusted subcutaneous low-molecular weight heparin for three weeks, followed by indefinite therapy with warfarin), and the headaches, vomiting, and diplopia resolved within four days of treatment. A follow-up MR-venogram performed two weeks later indicated complete recanalization of the intracranial dural venous sinuses. The PT was improved after two weeks of medical therapy, so she could adapt to it without intervention on the PSS. The early initialization of an efficient medical therapy had a great impact on her favorable evolution. PSS could be identified in her case on HRCT.