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27例地诺单抗治疗后骨巨细胞瘤的临床病理特征:印度一家三级癌症转诊中心的单机构经验

Clinicopathological Features of a Series of 27 Cases of Post-Denosumab Treated Giant Cell Tumors of Bones: A Single Institutional Experience at a Tertiary Cancer Referral Centre, India.

作者信息

Rekhi Bharat, Verma Vivek, Gulia Ashish, Jambhekar Nirmala A, Desai Subhash, Juvekar Shashikant L, Bajpai Jyoti, Puri Ajay

机构信息

Department of Surgical Pathology, Tata Memorial Hospital, 8th Floor, Annex Building, Dr E.B. Road, Parel, Mumbai, 400012, India.

Department of Surgical Oncology (Bone and Soft Tissues), Tata Memorial Hospital, Parel, Mumbai, 400012, India.

出版信息

Pathol Oncol Res. 2017 Jan;23(1):157-164. doi: 10.1007/s12253-016-0123-0. Epub 2016 Oct 8.

DOI:10.1007/s12253-016-0123-0
PMID:27722984
Abstract

Giant cell tumor of bone (GCTB) is mostly a benign tumor, but associated with recurrences and metastasis. Lately, denosumab is being utilized in the treatment of certain GCTBs. Twenty-seven tumors, analyzed in the present study, occurred in 16 males and 11 females (M: F = 1.45:1), in the age-range of 16 to 47 years (mean = 29.5, median = 29). Most tumors were identified in the tibia(6) and femur(6), followed by the humerus(3), radius(3), pelvis(3), fibula(3), sacrum(1), metacarpal(1) and metatarsal(1) bones. There were 18(66.6 %) primary and 9(33.3 %) recurrent tumors. Exact tumor size (19 cases) varied from 3.7 to 15 cm (mean = 7.8, median = 6.4). Eight of the 19 tumors (42.1 %) had size more than or equal to 8 cm. On histopathologic examination of post-denosumab treated specimens, more than half cases (15)(55.5 %) revealed complete absence of osteoclast-like giant cells (OCLGs) and 12 cases revealed residual OCLGCs. In addition, there was replacement by fibro-osseous tissue, including reactive woven bone or osteoid in most cases, followed by variable amount of spindle cells, hyalinisation, fibrosis and chronic inflammatory cells, including lymphocytes, macrophages and plasma cells. Post-treatment follow-up (25 cases, 92.5 %), over 7-27 months duration (median = 18), revealed 20 cases continuously disease-free. Five patients developed recurrences at 9, 12, 13, 14 and 18 months, respectively. Out of these, who underwent repeat surgical intervention, 4 patients are alive with no evidence of disease and a single patient, planned for a second surgery, is alive-with-disease. Denosumab was mostly offered to patients with large sized, borderline salvageable tumors, in order to decrease the morbidity of index surgical procedure, that led to disappearance of OCLGCs in most cases. Post-denosumab treated GCT cases appear as low grade osteosarcomas on histopathologic examination, but lack the clinical behaviour of an osteosarcoma, therefore may be considered as pseudo malignant bony lesions.

摘要

骨巨细胞瘤(GCTB)大多为良性肿瘤,但易复发和转移。近来,地诺单抗被用于某些骨巨细胞瘤的治疗。本研究分析的27例肿瘤发生于16例男性和11例女性(男∶女 = 1.45∶1),年龄在16至47岁之间(平均29.5岁,中位数29岁)。大多数肿瘤位于胫骨(6例)和股骨(6例),其次是肱骨(3例)、桡骨(3例)、骨盆(3例)、腓骨(3例)、骶骨(1例)、掌骨(1例)和跖骨(1例)。有18例(66.6%)为原发性肿瘤,9例(33.3%)为复发性肿瘤。确切的肿瘤大小(19例)在3.7至15厘米之间(平均7.8厘米,中位数6.4厘米)。19例肿瘤中有8例(42.1%)大小大于或等于8厘米。在地诺单抗治疗后的标本组织病理学检查中,超过半数病例(15例,55.5%)显示完全没有破骨细胞样巨细胞(OCLGs),12例显示有残余的破骨细胞样巨细胞。此外,大多数病例被纤维骨组织替代,包括反应性编织骨或类骨质,其次是不同数量的梭形细胞、玻璃样变、纤维化和慢性炎症细胞,包括淋巴细胞、巨噬细胞和浆细胞。治疗后随访(25例,92.5%),持续7至27个月(中位数18个月),显示20例持续无病。5例患者分别在9、12、13、14和18个月时复发。其中,接受再次手术干预的患者中,4例存活且无疾病证据,1例计划进行二次手术的患者仍患有疾病。地诺单抗主要用于治疗大型、临界可挽救的肿瘤患者,以降低初次手术的发病率,这在大多数情况下导致破骨细胞样巨细胞消失。地诺单抗治疗后的骨巨细胞瘤病例在组织病理学检查中表现为低级别骨肉瘤,但缺乏骨肉瘤的临床行为,因此可被视为假性恶性骨病变。

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