Introduction and importance: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign vascular tumor characterized by abnormal endothelial proliferation and inflammatory cell infiltration, primarily affecting the head and neck region. The diverse clinical presentations of ALHE pose significant diagnostic challenges, often leading to misdiagnosis. Accurate histopathological examination is crucial for differentiating ALHE from other vascular lesions and guiding appropriate treatment.
Case presentation: We report the case of a 27-year-old male who presented with a solitary, asymptomatic, dome-shaped lesion on his right cheek. Initial clinical examination and MRI suggested a diagnosis of atypical hemangioma. Surgical excision of the lesion was performed, and histopathological analysis revealed features consistent with ALHE, including vascular hyperplasia, epithelioid endothelial cells, and a mixed inflammatory infiltrate with eosinophils. The patient remained asymptomatic with no recurrence during a two-year follow-up period.
Clinical discussion: This case highlights the diagnostic complexities associated with ALHE due to its varied clinical and radiological presentation, often mimicking other benign or malignant vascular lesions. Although MRI findings initially suggested hemangioma, histopathological confirmation was pivotal in establishing the correct diagnosis. The involvement of the cheek in ALHE is rare, with most cases affecting other regions of the head and neck. The standard treatment for ALHE remains surgical excision, with our patient showing no recurrence over a two-year period.
Conclusion: Given the potential for misdiagnosis due to the overlapping features of ALHE with other vascular lesions, clinicians should maintain a high index of suspicion and consider histopathological examination in atypical cases.
Keywords: Angiolymphoid hyperplasia with eosinophilia; Eosinophilia; Surgical excision; Vascular neoplasms.
Copyright © 2025 The Authors. Published by Elsevier Ltd.. All rights reserved.