Angiolymphoid hyperplasia with eosinophilia (AHE) is a rare skin condition of unknown aetiology. The lesion seems neoplastic in nature, or at least an abnormal vasoproliferative reaction.A 40-year-old man presented with an 18-month history of erythematous papula over the right temporal area without a history of trauma. The patient reported a history of Hodgkin lymphoma at the age of 20, treated by radiochemotherapy. A subcutaneous nodule was found on the superior branch of the right temporal artery. An echocolordoppler revealed a normal temporal artery flow with pariental thickness. An excisional biopsy was performed and the patient remained asymptomatic at 24 months. The histological diagnosis was angiolymphoid hyperplasia with eosinophilia of the temporal artery.More appropriate studies are necessary to assess whether AHE is a manifestation of an unknown immunological disorder. If a correlation could be found between an altered immunological system and AHE, an intensive follow-up could be applied to patients. We report this case to encourage further studies to highlight potential challenges in the diagnosis and management of variants of vascular processes, such as AHE.