We describe the case of a patient with symptomatic strictly unilateral paroxysmal headache mimicking cluster headache related to an ipsilateral forehead lipoma. Interestingly, immediately after the surgical excision of the lipoma pain attacks disappeared with no recurrence during a follow-up period of 18 months. Like other descriptions of cluster-like headaches secondary to extracranial lesions, this case report focuses on the hypothetical role of a peripheral trigger factor for trigeminal autonomic cephalgias (TACs). To our knowledge, this is the first well-described cluster-like headache case secondary to an extracerebral lipoma. This case offered the opportunity to discuss the possible pathophysiological mechanisms underlying probable TACs and the relationship with peripheral extracerebral activation of the trigeminal-autonomic reflex.