Cerebral venous thrombosis (CVT) is a rare but potentially life-threatening cerebrovascular disorder that frequently presents with headache as a dominant or even sole symptom. This chapter provides a comprehensive overview of CVT-related headache, integrating clinical presentation, pathophysiology, diagnosis, and treatment through the lens of a detailed postpartum case. Headache in CVT is highly variable, ranging from diffuse subacute pain to thunderclap-like onset, often mimicking primary headache disorders and delaying diagnosis. We present the case of a young postpartum female whose only initial symptom was a progressive occipital headache, ultimately culminating in seizures and leading to the diagnosis of CVT confirmed by magnetic resonance imaging (MRI) and MR venography. This chapter explores the multifactorial pathophysiological mechanisms—including venous congestion, intracranial hypertension, cortical infarction, neuroinflammation, and hemorrhagic transformation—that underlie headache in CVT. Pain generation is mediated by activation of the trigeminovascular system, sensitization of central pain pathways, and mechanical stress on pain-sensitive intracranial structures. Diagnostic approaches emphasize the importance of high clinical suspicion, neuroimaging, and exclusion of prothrombotic conditions. Treatment involves anticoagulation as the mainstay, supplemented by intracranial pressure management, seizure control, and long-term headache strategies. This case exemplifies the critical role of early recognition of atypical headache patterns in CVT and underscores the need for multidisciplinary vigilance in postpartum patients. This chapter concludes with current gaps in knowledge and future directions, particularly in individualized headache management and post-thrombotic care.