Central giant cell granuloma: off-label treatment with Denosumab in a patient with Noonan syndrome

• Published in: Journal of stomatology, oral and maxillofacial surgery oral and maxillofacial surgery, 2024-02, Vol.125 (1), p.101640, Article 101640
• Main Authors: Amorós, Carlota Mazo, Bascones, Alejandro Encinas, Leone, Roy Camacho, De la Sen Corcuera, Óscar, Barone, Simona, De Pedro Marina, Manuel
• Format: Article
• Language: English
• Subjects: Denosumab; Giant cell granuloma; immunotherapy; Noonan syndrome
• ISSN: 2468-7855; 2468-7855
• DOI: 10.1016/j.jormas.2023.101640

This study aims to describe the utilization of Denosumabࣨ, a human monoclonal antibody against the RANK-L receptor, in a mandibular giant cell granuloma (GCG) with a significant local aggressiveness component that was unresponsive to surgical treatment. We present a case of a 19-year-old male patient diagnosed with Noonan syndrome, who presented a multifocal giant cell granuloma with aggressive behavior resistant to surgical treatment. Due to the functional and aesthetic implications associated with a surgical procedure, a decision was made to initiate medical treatment using Denosumabࣨ. Throughout the treatment, the patient presented excellent clinical and analytical tolerance, with no reported adverse effects. Surgical intervention remains the preferred approach for GCG. Denosumabࣨ emerges as an alternative, either as neoadjuvant treatment or as definitive therapy for unresectable or resectable tumors associated with significant morbidity. It leads to size stabilization and regression of the tumor stage.