Head and Neck Pathol (2009) 3:174–178 DOI 10.1007/s12105-009-0119-0 C A S E R E P O R T A Case of Recurrent Multifocal Central Giant Cell Granulomas Elizabeth Bilodeau Æ Khalid Chowdhury Æ Bobby Collins Received: 10 November 2008 / Accepted: 8 May 2009 / Published online: 11 June 2009 Humana 2009 Abstract One case of recurrent multifocal central giant cell granulomas (CGCG) is presented. Initially, the lesions presented concurrently in the maxilla and mandible with subsequent recurrence in the mandible. Now, two recur- rences are seen in the maxillary sinus and ethmoid region. The literature regarding multifocal CGCG is reviewed. Keywords Central giant cell granulomas (CGCG) Reparative giant cell granuloma Craniofacial giant cell dysplasia Introduction The central giant cell granuloma (CGCG) affects females more often than males, in a 2:1 ratio and is seen most fre- quently under the age of 30 years [1]. One study of 38 patients shows 74% to be less than 30 years of age and 61% to be less than 20 years of age [2]. The lesion commonly presents as a solitary radiolucency with a multilocular appearance or less commonly, a unilocular appearance [2–4]. It is more prevalent in the anterior than the posterior jaws, often crossing the midline, and the mandible is more commonly affected than the maxilla [2, 4]. This lesion has E. Bilodeau (&) B. Collins Department of Diagnostic Sciences, University of Pittsburgh School of Dental Medicine, Pittsburgh, PA, USA e-mail: rodeea@upmc.edu B. Collins e-mail: bcollins@dental.pitt.edu K. Chowdhury Center for Craniofacial & Skull Base Disorders/Department of Otolaryngology, University of Colorado, Denver, CO, USA e-mail: craniofacial@qwestofﬁce.net also been reported in the small bones of the hands and feet [5, 6]. The behavior of CGCG is variable, most commonly producing asymptomatic expansion of the jaws [7]. How- ever, it can be clinically aggressive, associated with pain, osseous destruction, cortical perforation, root resorption, and recurrence [8]. Cases of CGCG occurring with neuro- ﬁbromatosis (type 1) [7, 9–11], Noonan-like syndrome [12, 13], or both [7, 14] have been reported. The treatment of CGCG includes simple curettage or curettage with peripheral ostectomy; Resection for lesions of the maxilla or paranasal sinuses has been advocated as the thin bony cortices and sinuses do not provide a good anatomic barrier [15]. Corticosteroids and calcitonin are used for non-surgical management [16, 17]. Interferon alpha therapy has also been used as a postoperative adju- vant and to prevent tumor progression [18, 19]. Report of a Case This patient’s histological slides were referred to the Uni- versity of Pittsburgh Medical Center for pathology consul- tation. Her medical records were subsequently obtained. The patient was a 42-year-old female with a history of multifocal giant cell lesions in the left mandible and left maxilla. A year after her initial diagnosis and treatment, a recurrent lesion was noted in the left mandible and subsequently curetted. Two and a half years after the initial lesions, this was the second recurrent episode. This recurrence, the patient pre- sented with two distinct lesions the larger, inferior lesion ﬁlling much of the maxillary sinus and the smaller, superior lesion adjacent to the ethmoid region. A comprehensive laboratory assessment to rule out hyperparathyroidism revealed: calcium (serum) 9.8 (normal 8.5–10.4 mg/dl), PTH (intact) 58 pg/ml (normal 12–65 pg/ml), phosphorus