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两例骨巨细胞瘤继发肉瘤接受地诺单抗治疗的病例

Two Cases of Sarcoma Arising in Giant Cell Tumor of Bone Treated with Denosumab.

作者信息

Broehm Cory Julian, Garbrecht Erika L, Wood Jeff, Bocklage Therese

机构信息

Department of Pathology, University of New Mexico School of Medicine, MSC08 4640, 1 University of New Mexico, Albuquerque, NM 87131, USA.

Department of Orthopaedics, University of New Mexico School of Medicine, MSC10 5600, Albuquerque, NM 87131, USA.

出版信息

Case Rep Med. 2015;2015:767198. doi: 10.1155/2015/767198. Epub 2015 Dec 22.

DOI:10.1155/2015/767198
PMID:26798348
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC4700156/
Abstract

Giant cell tumor (GCT) of bone is a generally benign, but often locally aggressive, neoplasm of bone, with a propensity for recurrence. Sarcomatous transformation is rare and typically occurs with a history of recurrences and radiation treatment. Denosumab, an inhibitor of the RANK ligand involved in bone resorption in GCT, is increasingly used in treatment of recurrent or unresectable giant cell tumor of bone. We report two cases of sarcomatous transformation of GCT to osteosarcoma in patients receiving denosumab. One was a 59-year-old male with a 12-year history of GCT and multiple recurrences taking denosumab for 2.5 years. The second case was in a 56-year-old male with a seven-year history of GCT taking denosumab for six months. Review of the literature shows one case report of malignant transformation of GCT in a patient being treated with denosumab. As the use of denosumab for treatment of GCT will likely increase, larger, controlled studies are needed to ascertain whether denosumab may play a role in malignant transformation of giant cell tumor of bone.

摘要

骨巨细胞瘤(GCT)通常是一种良性但常具有局部侵袭性的骨肿瘤,容易复发。肉瘤样变罕见,通常发生在有复发和放疗史的情况下。地诺单抗是一种参与骨巨细胞瘤骨吸收的RANK配体抑制剂,越来越多地用于治疗复发性或不可切除的骨巨细胞瘤。我们报告了两例接受地诺单抗治疗的骨巨细胞瘤肉瘤样变至骨肉瘤的病例。一例是一名59岁男性,有12年骨巨细胞瘤病史且多次复发,接受地诺单抗治疗2.5年。第二例是一名56岁男性,有7年骨巨细胞瘤病史,接受地诺单抗治疗6个月。文献回顾显示有一例关于接受地诺单抗治疗的骨巨细胞瘤恶性变的病例报告。由于地诺单抗用于治疗骨巨细胞瘤的情况可能会增加,因此需要进行更大规模的对照研究,以确定地诺单抗是否可能在骨巨细胞瘤的恶性变中起作用。

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A High-grade Sarcoma Arising in a Patient With Recurrent Benign Giant Cell Tumor of the Proximal Tibia While Receiving Treatment With Denosumab.一名接受地诺单抗治疗的复发性胫骨近端良性骨巨细胞瘤患者发生了高级别肉瘤。
Clin Orthop Relat Res. 2015 Sep;473(9):3050-5. doi: 10.1007/s11999-015-4249-2. Epub 2015 Mar 11.
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单细胞 RNA 测序推动骨骼健康和疾病研究。
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Current Concepts in the Treatment of Giant Cell Tumor of Bone: An Update.当前骨巨细胞瘤治疗的新理念:更新。
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