Tehran University of Medical Sciences Frontiers in Dentistry 2676-296X 19 0 2022 08 10 3897 3897 Hoorieh Bashizadehfakhar Dental Research Center, Dentistry Research Institute, Tehran University of Medical Sciences, Tehran, Iran AND Department of Dentomaxillofacial Radiology, Faculty of Dentistry, Tehran University of Medical Sciences, Tehran, Iran. Mehrdad Panjnoush Department of Dentomaxillofacial Radiology, Faculty of Dentistry, Tehran University of Medical Sciences, Tehran, Iran. Yasaman Kheirandish Department of Dentomaxillofacial Radiology, Faculty of Dentistry, Tehran University of Medical Sciences, Tehran, Iran. Azin Sedaghati Department of Dentomaxillofacial Radiology, Faculty of Dentistry, Ilam University of Medical Sciences, Ilam, Iran. Faeze Mirjalili Department of Dentomaxillofacial Radiology, Faculty of Dentistry, Kashan University of Medical Sciences, Kashan, Iran Sara Aliasghari Abandansari Dental Research Center, Dentistry Research Institute, Tehran University of Medical Sciences, Tehran, Iran 2020 12 25 2021 05 01 Congenital diffuse infiltrating lipomatosis of the face (CDIL-F) is a rare clinicopathological entity with an unknown etiology, in which mature adipose tissue infiltrates the soft tissue structures in one side, and causes considerable asymmetry. Herein, we report a case of CDIL-F who underwent many surgical procedures without definite diagnosis during 12 years.  CDIL-F presents symptoms with various severity levels due to infiltration of adipose tissue that makes removal difficult. Thus, it is recommended to inform patients about the high rate of recurrence. https://fid.tums.ac.ir/index.php/fid/article/view/3897