%0 Case Reports %T Unilateral Hyperpigmented Facial Lesion Since Birth. %A Dalave K %A Deoghare S %A Buccha Y %J Cureus %V 15 %N 10 %D 2023 Oct %M 38034188 暂无%R 10.7759/cureus.47901 %X Facial hyperpigmented lesions that are unilateral are a rare and challenging dermatological anomaly since birth which can be genetic and non-genetic. This paper seeks to provide an exhaustive overview of the etiology, clinical presentation, differential diagnosis, and management strategies for these congenital lesions. Unilateral facial hyperpigmented lesions can be caused by a number of conditions, such as congenital melanocytic nevi, Becker's nevus, nevus of Ota, linear epidermal nevi, and café-au-lait macules. Accurate diagnosis requires meticulous examination, dermoscopy, and histopathological evaluation. Observation, topical therapies, surgical excision, and laser therapy are among the available treatment options. Treatment decisions should be influenced by factors such as lesion characteristics, aesthetic concerns, and patient preferences. Long-term supervision and psychosocial support are indispensable elements of comprehensive management. We present a case of a 12-year-old patient with a progressively growing hyperpigmented lesion on the right side of the face, present since birth with an intermittent area of normal skin in between. Dermoscopy unveiled an irregular, dark brown pigment network, and histopathological evaluation showed an increased number of melanocytes in the dermis. This case highlights the diagnostic challenges of such lesions and underscores the significance of a multidisciplinary approach for accurate evaluation and management. This paper aims to cover existing knowledge gaps regarding unilateral facial hyperpigmented lesions since birth and direct future research efforts for diagnostic and therapeutic interventions.