(42) Treatment Modalities Utilized in Managing Hybrid Central Giant Cell Granuloma and Fibro-osseous Lesions: A Case Study and Systematic Review

Abstract: Synchronous presentation of lesions involving central giant cell granuloma and fibro-osseous lesion (CGCG-FOL) components present a unique challenge in clinical management due to their rarity and complex pathologic nature.1 By examining reported cases, this paper aims to systematically review the treatment modalities utilized to manage hybrid CGCG-FOLs of the jaw. This study also introduces the case of a 12-year-old male with a hybrid lesion of CGCG and fibrous dysplasia (FD). A systematic review was performed based on the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) utilizing PubMed, Embase, and Google Scholar databases. The included studies focused on CGCG tumors that simultaneously presented with FOLs. Studies were excluded if they did not identify hybrid lesions, presented hybrid lesions of CGCG unrelated to FOLs, did not mention treatment conducted, or only reported treatment as surgical debulking. All statistical analyses were performed using GraphPad Prism (version 10.1.2).Among the 1,177 publications screened, 17 were included, describing 19 patients with hybrid CGCG and a FOL. There were 7 males (36.8%) and 12 females (63.2%) with mean age of 28.6 ± 16.9 years. Fourteen of the hybrid lesions involved ossifying fibroma (73.7%), 4 with fibrous dysplasia (21.1%), and 1 with cemento-osseous dysplasia (5.3%). The predominant treatment modality utilized was surgical excision (FD: 50%, OF: 42.9%, COD: 100%). Only 1 patient in our study had a recurrent lesion (5.3%) who was initially treated with surgical excision, specifically curettage-only, for hybrid CGCG-ossifying fibroma. When the CGCG-FOLs were characterized by age (pediatric, < 21 vs. adult, >21 years old), the predominant treatment modality was resection at 54.5% for adults and only 33.3% for pediatric patients. In the adult age group, only one study described using pharmacologic treatment followed by a surgical resection. In the pediatric age group, 3 studies utilized pharmacology-only or pharmacology along with a type of surgical excision. In the pediatric population, it was more common to treat hybrid lesions with excision-only or excision with pharmacology (55.6%), rather than resection-only (33.3%). This systematic review found that surgical excision, including surgical enucleation or curettage, was the predominant choice for treatment of CGCG-FOLs lesions of the jaw. When divided by age between adult (>21 years old) and pediatric populations ( < 21 years old), adult patients were more likely to undergo resection and pediatric patients were more likely to undergo excision procedures, regardless of which hybrid CGCG was encountered. Of the 3 studies that used pharmacology, CGCG-ossifying fibroma responded best to pre-surgical pharmacology. This treatment modality should be heavily considered in pediatric patients for its benefits in preserving growing structures and decreasing surgical morbidity.2 Although there was a lack of consensus on the type of surgical intervention, only one patient showed recurrence of a hybrid lesion. Of the 19 patient cases examined, only 4 demonstrated the same CGCG-FD hybrid encountered in the case study portion of this systematic review. We encourage the continued reporting of these hybrid cases to compile data and enable future providers to provide the most effective treatment possible given the variability and complexity of these cases.1. Kurra, S., et al. (2013). Fibrous dysplasia and central giant cell granuloma: a report of hybrid lesion with its review and hypotheticated pathogenesis. Journal of clinical and diagnostic research: JCDR, 7(5), 954–958. https://doi.org/10.7860/JCDR/2013/5533.29872. Choe, M., et al. (2020). Treatment of central giant cell granuloma in children with denosumab. Pediatric Blood & Cancer, 68(3). https://doi.org/10.1002/pbc.28778

CGCG-FOLs Hybrid Lesions Study Prisma Flowchart
Demographics, Radiographic Appearance, Treatment and Recurrence of hybrid CGCG- FOLs
Pharmacologic Interventions in Hybrid CGCG-FOLs