An association between granulocyte colony-stimulating factor therapy (G-CSFT) in patients with glycogen storage disease type Ib (GSDIb) and the development of giant cell lesions of the maxillofacial complex has emerged. We have reported, to the best of our knowledge, the fourth case of giant cell granuloma (GCG) in a patient with GSDIb undergoing G-CSFT. GSDIb can present with hypoglycemia, hypertriglyceridemia, and neutropenia. G-CSFT has often been used in the treatment of recurrent infections or sepsis caused by neutropenia and to treat inflammatory bowel disease and diarrhea. The current reported data are lacking in both the association and the potential causation of G-CSFT and the development of giant cell tumors. Given the prevalence of GSDIb and its therapy, oral and maxillofacial surgeons should be aware of the tumorigenic potential of G-CSFT in patients with GSDIb. In the present report, we have described the case of a 17-year-old patient with GSDIb undergoing GCSFT who presented with a peripheral and central GCG. She was treated but presented again 13 months later with concerns for a new primary lesion. We have also discussed GSDIb, G-CSFT, and the current data, highlighting the association between G-CSFT for GSDIb, the potential mechanism of GCG development, the use of adjuvant therapy, and the need for close follow-up of this population. The purpose of the present case report is to highlight the presentation, management, and follow-up of giant cell lesions in patients with GSDIb treated with G-CSFT.
ASJC Scopus subject areas
- Oral Surgery