Paroxysmal hemicrania is a trigeminal autonomic cephalalgia first described in 1976, characterized by episodic attacks of excruciating unilateral periorbital and temporal stabbing, pulsatile, craw-like, or boring headaches lasting 2 – 30 minutes, accompanied by autonomic features, and alleviated by indomethacin. Paroxysmal hemicrania is divided into an episodic or chronic form, depending on the duration and frequency of the attacks. We describe a case of paroxysmal hemicrania in a patient with a contralateral anterior clinoid meningioma, which resolved after tumor resection. Most cases of autonomic cephalgias are primary headaches and not caused by underlying intracranial structural lesions. Based on our patient and a literature review of secondary causes of trigeminal autonomic cephalalgias, we recommend that all patients with trigeminal autonomic cephalalgias including paroxysmal hemicrania undergo neuroimaging studies. The preferred neuro-radiologic procedure should be a cranial MRI to exclude underlying structural intracranial lesions, particularly in the sellar and parasellar regions.