Rytkönen Eelis, Ottavainen Vuokko, Rytkönen Aleksi, Uusitalo Sanna, Lehenkari Petri, Sándor George K
Department of Oral and Maxillofacial Surgery, Oulu University Hospital, Oulu, Finland.
Department of Pathology, Oulu University Hospital, Oulu, Finland.
Ann Maxillofac Surg. 2018 Jul-Dec;8(2):265-269. doi: 10.4103/ams.ams_192_18.
Aggressive familial giant-cell granulomas of the jaws can be severely deforming. Surgical and nonsurgical treatments may be associated with multiple recurrences. Denosumab, a new generation antiresorptive drug, is an osteoclast inhibitor, which may be particularly useful to manage such potentially disfiguring lesions.
Two sisters, both with a history of multiple recurrent aggressive central giant-cell granuloma (CGCG)-like lesions in both jaws, were referred for management. All lesions were histologically consistent with the diagnosis of CGCG. The lesions were treated surgically with curettage and perilesional injection of triamcinolone. In particular, the older sister had four separate anatomic sites where some of her lesions had multiple recurrences necessitating three repeat procedures. A course of subcutaneous denosumab was administered following the last giant-cell granuloma removal in both sisters.
Bony healing was normal. No further recurrences were observed over 3.5 years of follow-up after denosumab therapy in either sister.
In this small cohort comprising two sisters with multiple aggressive recurrent giant-cell granuloma lesions at multiple sites in the mouth, subcutaneous denosumab administration was associated with success over 3.5 years of follow-up. This report cautiously adds to the clinical experience in the use of denosumab for the treatment of recurrent aggressive familial CGCG lesions.
侵袭性颌骨家族性巨细胞肉芽肿可导致严重畸形。手术和非手术治疗可能会多次复发。地诺单抗是一种新一代抗吸收药物，是一种破骨细胞抑制剂，可能对处理此类潜在毁容性病变特别有用。
两姐妹均有双侧颌骨多处复发性侵袭性中央巨细胞肉芽肿（CGCG）样病变病史，前来就诊。所有病变的组织学表现均符合CGCG的诊断。病变采用刮除术和病变周围注射曲安奈德进行手术治疗。特别是，姐姐有四个不同的解剖部位，其中一些病变多次复发，需要进行三次重复手术。在两姐妹最后一次切除巨细胞肉芽肿后，给予一个疗程的皮下注射地诺单抗。
骨愈合正常。在接受地诺单抗治疗后的3.5年随访中，两姐妹均未观察到进一步复发。
在这个由两姐妹组成的小队列中，她们口腔内多个部位有多处侵袭性复发性巨细胞肉芽肿病变，皮下注射地诺单抗在3.5年的随访中取得了成功。本报告谨慎地增加了使用地诺单抗治疗复发性侵袭性家族性CGCG病变的临床经验。
