A 19-year-old woman was referred to the Oral and Maxillofacial Pathology Resident Clinic at Northwell Health Long Island Jewish Medical Center with a primary symptom of “dark spots on my tongue and lips.” She initially reported a noncontributory medical and social history and was not taking any medication at that time nor had any allergies to report. On further questioning, the patient revealed a prior diagnosis of a condition involving multiple bones, including her femur, humerus, maxilla, and mandible. This condition caused expansion of these bones and created a greater risk of developing a fracture. Previous radiographs of the humerus and femur were reviewed (Figure 1). At 10 years of age, she underwent mandibular recontouring for mandibular expansion and obvious facial asymmetry. The tissue was submitted for histologic examination, which revealed a benign fibro-osseous lesion consisting of irregularly shaped, lamellar trabeculae with minimal osteoblastic rimming in a fibrous stroma exhibiting clefting around the trabeculae (Figure 2). She also reported multiple pigmented cutaneous macules on her right shoulder, right buttock, and right inner thigh that had not changed in size and were of unknown duration. She reported an incident of vaginal bleeding at the age of 7, which on further evaluation was revealed to be a sequela from an ovarian cyst. A search of our institution’s archives revealed that the histologic examination of the tissue showed an ovarian follicular cyst with luteinization, which is not an age-appropriate finding in a prepubescent 7-year-old girl. No other family member had exhibited these signs or symptoms. Figure 2Histologic presentation of tissue submitted after recontouring of expansile mandibular lesion. A. A low-power (hematoxylin-eosin, original magnification ×40) image of the tissue specimen shows irregular trabeculae of woven bone in a somewhat parallel arrangement within a fibrous connective tissue stroma. There is a marked clefting artifact separating the connective tissue stroma from the bony trabeculae. B. A higher power (hematoxylin-eosin, original magnification ×100) view of the tissue shows the minimal osteoblastic rimming around the trabeculae in scattered areas but absent entirely in others. The fibrous tissue stroma, parallel trabeculae arrangement, and cleft artifact can also be seen at this power. View Large Image Figure Viewer Download Hi-res image Dr Roth is an oral and maxillofacial pathology resident, Department of Dental Medicine, Donald and Barbara Zucker School of Medicine, Hofstra/Northwell Health, New Hyde Park, NY. Dr Kelsch is an associate professor, Donald and Barbara Zucker School of Medicine, Hofstra/Northwell Health, New Hyde Park, NY.