Prognosis of metastatic giant cell tumor of bone in the pre-denosumab era. A systematic review and a meta-analysis

Itkin, Boris; Straminsky, Samanta; Ronato, Gabriela De; Lewi, Daniel; Marantz, Adolfo; Bardach, Ariel

doi:10.1093/jjco/hyy067

Cited by 17 publications

(27 citation statements)

References 66 publications

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“…In the present study, with the large cohort of malignant GCTB, the 5-year OS was 82.3% (95% CI: 77.9-86.6). This rate was consistent with the reported rate of 86.9% by Itkin et al [10] and 80% by Domovitov et al [12]. The prognostic factors were revealed, and poor survival was correlated with the following factors including age older than 34 years, higher T stage, distant tumor and the extraskeletal sites.…”

Section: Discussionsupporting

confidence: 92%

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Survival and prognostic factors analyses in malignant giant cell tumor of bone

Zhang¹,

Wang

Li³

et al. 2019

Preprint

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Background The characteristics and survival in patients with malignant giant tumor cancer of bone (GCTB) were not investigated thoroughly due to the limited population. We evaluated the issues based on a large cohort in the National Cancer Institute’s Surveillance, Epidemiology, and End Results (SEER) database. Methods Patients who were diagnosed with malignant GCTB from 1975 to 2016 were extracted from the SEER database. The overall survival (OS) was calculated by Kaplan–Meier analysis and the inter-group difference was tested by log-rank test. Univariate and multivariate Cox proportional hazard regression were conducted to identify the independent survival factors. Results A total of 325 patients with malignant GCTB were included. The overall 1-, 5-, and 10-year survival rates were 94.3% (95% CI: 91.7-96.8), 82.3% (95% CI: 77.9-86.6), and 80.1% (95% CI: 75.4-84.7), respectively. In the univariate analysis, age older than 34 years, grade IV, T2/3 stage, M1, distant and surgery of the primary site. Multivariate Cox regression showed the poor survival in patients with age older than 34 years (hazard ratio (HR) =3.68, 95% CI: 2.06-6.57, P<0.001), T2 stage (HR=4.96, 95% CI: 1.57-15.63, P=0.006), distant tumor (HR=3.02, 95% CI: 1.32-6.92, P=0.009), and the extra-skeletal sites (HR=8.84, 95% CI: 2.89-27.07, P<0.001), respectively. Conclusions This large population-based series described the clinical characteristics of the malignant GCTB. Age, T2, distant tumor and extra-skeletal sites were determinant survival factors of the patients with malignant GCTB.

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Section: Discussionsupporting

confidence: 92%

“…But the issues were not intensively studied due to the lack of long-term followup clinical data [9]. Itkin and his colleagues retrospectively retrieved the data of 242 patients from 26 studies with a median follow-up of 6.9 years [10]. They reported an estimated overall survival rate of 86.9%.…”

Section: Introductionmentioning

confidence: 99%

Survival and prognostic factors analyses in malignant giant cell tumor of bone

Zhang¹,

Wang

Li³

et al. 2019

Preprint

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“…(95% CI: 77.9-86.6). This rate was consistent with the rate of 86.9% reported by Itkin et al [10] and 80% reported by Domovitov et al [13]. Prognostic factors were identi ed, and poor survival was correlated with the following factors: age older than 34 years, high T stage, distant tumour stage and extraskeletal sites.…”

Section: Discussionsupporting

confidence: 91%

Survival and Prognostic Factors Analyses in Malignant Giant Cell Tumor of Bone

Zhang¹,

Wang²,

Lin³

et al. 2020

Preprint

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Background: The characteristics and survival of patients with malignant giant cell tumour of bone (GCTB) have not been investigated thoroughly due to the rarity of the disease. We evaluated these factors in a large cohort in the National Cancer Institute’s Surveillance, Epidemiology, and End Results (SEER) database.Methods: Data from patients who were diagnosed with malignant GCTB from 1975 to 2016 were extracted from the SEER database. The overall survival was calculated by Kaplan–Meier analysis, and intergroup differences were tested by the log-rank test. Univariate and multivariate Cox proportional hazard regression analyses were conducted to identify the independent survival factors.Results: A total of 325 patients with malignant GCTB were included. The overall 1-, 5-, and 10-year survival rates were 94.3% (95% CI: 91.7-96.8), 82.3% (95% CI: 77.9-86.6), and 80.1% (95% CI: 75.4-84.7), respectively. A potential non-linear J-shaped dose–response relationship between the age or diagnosis year and survival were found. Multivariate Cox regression showed poor survival in patients with age from 35 to 60 years (HR=9.99, 95% CI: 1.34-74.80, P=0.025), age older than 60 years (HR=62.03, 95% CI: 7.94-484.38, P<0.001), with stage T2 disease (HR=4.85, 95% CI: 1.52-15.47, P=0.008), with stage T3 disease (HR=6.09, 95% CI: 1.03-36.23, P=0.047), and with distant tumours (HR=2.76, 95% CI: 1.14-6.65, P=0.024), and extraskeletal sites (HR=3.33, 95% CI: 1.02-10.85, P=0.046).Conclusions: This large population-based series described the clinical characteristics of malignant GCTB. Patients with stage T2/3 disease, distant disease and extra-skeletal sites had more odds to be with worse survival. The elder age than 34 years had a gradually increased risk for survival.

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“…Pulmonary metastases are very rare and typically slow-growing. These are thought to represent pulmonary implants that result from embolization of intravascular growths of GCTB [33].…”

Section: Giant Cell Tumour Of Bonementioning

confidence: 99%

Mutation-driven epigenetic alterations as a defining hallmark of central cartilaginous tumours, giant cell tumour of bone and chondroblastoma

et al. 2019

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Recently, specific driver mutations were identified in chondroblastoma, giant cell tumour of bone and central cartilaginous tumours (specifically enchondroma and central chondrosarcoma), sharing the ability to induce genome-wide epigenetic alterations. In chondroblastoma and giant cell tumour of bone, the neoplastic mononuclear stromal-like cells frequently harbour specific point mutations in the genes encoding for histone H3.3 (H3F3A and H3F3B). The identification of these driver mutations has led to development of novel diagnostic tools to distinguish between chondroblastoma, giant cell tumour of bone and other giant cell containing tumours. From a biological perspective, these mutations induce several global and local alterations of the histone modification marks. Similar observations are made for central cartilaginous tumours, which frequently harbour specific point mutations in the metabolic enzymes IDH1 or IDH2. Besides an altered methylation pattern on histones, IDH mutations also induce a global DNA hypermethylation phenotype. In all of these tumour types, the mutation-driven epigenetic alterations lead to a highly altered transcriptome, resulting for instance in alterations in differentiation. These genomic alterations have diagnostic impact. Further research is needed to identify the genes and signalling pathways that are affected by the epigenetic alterations, which will hopefully lead to a better understanding of the biological mechanism underlying tumourigenesis.

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Prognosis of metastatic giant cell tumor of bone in the pre-denosumab era. A systematic review and a meta-analysis

Abstract: To our knowledge this is the first study to derive estimated pooled OS and DSS of mGCT based on a large dataset. SRs were not exceptional phenomena. In a long run the disease could impact in a significant way on the life expectancy of affected subjects.

Cited by 17 publications

References 66 publications

Survival and prognostic factors analyses in malignant giant cell tumor of bone

Survival and prognostic factors analyses in malignant giant cell tumor of bone

Survival and Prognostic Factors Analyses in Malignant Giant Cell Tumor of Bone

Mutation-driven epigenetic alterations as a defining hallmark of central cartilaginous tumours, giant cell tumour of bone and chondroblastoma

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