Denosumab Treatment for Aggressive Multiple Recurrent Familial Central Giant Cell Granulomas

Vuokko Ottavainen DDS, MD, Oral and Maxillofacial Surgery, Chief Resident, Oulu, Finland
Eelis Rytkönen DDS, Department of Oral and Maxillofacial Surgery, Resident, Oulu, Finland
Sanna Uusitalo DDS, Oral and Maxillofacial Surgery, Resident, Oulu, Finland
Aleksi Rytkönen DDS, Oral and Maxillofacial Surgery, Staff Oral Pathologist, Oulu, Finland
Petri Lehenkari MD, PhD, Anatomy, Professor of Anatomy, Oulu, Finland
George K. Sŕndor MD. DDS, PhD, Oral and Maxillofacial Surgery and Tissue Engineering, Professor, Oulu, Finland
Aggressive familial giant cell granulomas (AFCGCGs) of the jaws can be severely deforming and present with multiple recurrences. Denosumab is a newer generation osteoclast inhibitor which radically differs from bisphosphonate medications and may be particularly useful to treat patients with AFCGCGs.

Two sisters with a history of multiple recurrent central giant cell granuloma-like lesions were treated at our university hospital after clinical presentation and comprehensive work-up for intra-oral tumor presentation. Tests included panoramic radiographs, CBCT examination, serum PTH, calcium, phosphorous and alkaline phosphatase levels, along with PET scans, genetic work-up and biopsy. All lesions were histologically consistent with the diagnosis of central giant cell granulomas and were treated surgically with curettage or local resection and peri-lesional injection of triamcynalone. In particular, the older sister had 4 separate lesion sites with multiple recurrences necessitating 3 repeat procedures. Denosumab treatment was given as a one-time adjuvant dose of 4 mg, 8 months following the last AFCGCG resection.

No further recurrences were noted in 3 years of follow-up after the Denosumab therapy. Denosumab is a potent osteoclast inhibitor and unlike bisphosphonates it is a monoclonal antibody specific to RANK Ligand receptors. Since AFCGCG are rich in osteoclast-like cells, the potential of Denosumab to treat such lesions is great.

In this small cohort of two sisters with aggressive recurrent lesions, this approach using a RANK Ligand inhibitor was successful over 3 years of follow-up, adding to the clinical experience of Denosumab treatment for AFCGCG lesions.

1. Malmquist M, Schow SR. Treatment of Central Giant Cell Granuloma with Denosumab Therapy in 2 Patients. J Oral Maxfac Surg 2013; 71(supplement): e75 poster 59.

2. Schreuder W, Coumou A, Kessler P, De Lange J. Alterenative Pharmacxological Therapy for the Aggressive Giant Cell Granuloma: Denosumab. J Oral Maxfac Surg. 2014; doi.org/10.1016/j.joms.2014.02.017

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