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<p>Abstract</p> <p>Plexiform neurofibroma (PN) is a rare benign tumor and a special subtype of neurofibromatosis type 1 (NF1). Though the incidence is low, giant PN of the craniomaxillofacial region could result in severe hemifacial hypertrophy which is known as a typical manifestation of NF1 in young children. Here, we retrospectively reported a giant plexiform neurofibroma with hemorrhage in the cranio-maxillofacial region detected by CT and MRI. In addition, a brief review of the relevant literature is presented.</p>