Delay in Diagnosis and Its Effect on Clinical Outcome in High‐grade Sarcoma of Bone: A Referral Oncological Centre Study

Goedhart, Louren M; Gerbers, Jasper G; Ploegmakers, Joris J. W.; Jutte, Paul C.

doi:10.1111/os.12239

Cited by 35 publications

(44 citation statements)

References 32 publications

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“…This study focused on the potential impact of two time intervals on the survival of children diagnosed with osteosarcoma who were treated in an LMIC. Our findings reveal that the median LD was longer than that observed in HIC and was comparable to reported times in other LMIC . Initial metastatic disease was found in 42.4% of patients; however, we failed to associate this finding with a delayed LD, which is similar to published data .…”

Section: Discussionsupporting

confidence: 87%

Prognostic impact of diagnostic and treatment delays in children with osteosarcoma

Vásquez

Silva

Chávez

et al. 2020

Pediatric Blood & Cancer

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Background The aim of this study is to evaluate the relationship between the latency to diagnosis (LD) and the time to completion of chemotherapy (TCC) with clinical outcomes in children with osteosarcoma. Methods We performed a retrospective analysis of all patients who received treatment for osteosarcoma in two tertiary centers in Peru from 2008 to 2015. All causes of delayed LD or TCC were evaluated. Overall survival (OS) and event‐free‐survival (EFS) were estimated and compared according to LD, TCC, and established clinical prognostic factors. Results One hundred and thirteen patients were included in the study. The median LD was 13.5 weeks (interquartile range, 10‐18.5 weeks). No association was observed among clinical stage, tumor size, and LD. Delayed LD was not associated with a worse clinical outcome. Multivariate analysis confirmed that OS and EFS were significantly worse in cases of a delayed TCC (≥4 weeks), with hazard ratios of 2.70 (1.11‐6.76, P = 0.003) and 1.13 (1.00‐1.26, P = 0.016), respectively. Most delays in TCC (85%) were due to extramedical reasons (e.g., lack of available hospital beds). Conclusion The LD did not seem to influence the EFS and OS in pediatric patients with osteosarcoma. However, a delay in TCC from any cause is independently associated with poor outcome in pediatric patients with osteosarcoma. Based on these results, further efforts may be needed to avoid treatment delays in patients with osteosarcoma in middle‐income countries.

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Section: Discussionsupporting

confidence: 87%

Prognostic impact of diagnostic and treatment delays in children with osteosarcoma

Vásquez

Silva

Chávez

et al. 2020

Pediatric Blood & Cancer

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“…Thirty-four studies involving a total of 17 258 patients investigated the total interval in BS (table 1)8–41; five of these studies prospectively collected follow-up data. A broad range in the length of the total interval was found, which varied from 9 to 120.4 weeks.…”

Section: Resultsmentioning

confidence: 99%

“…Tumour histology was found to be of influence on the total interval. Goedhart et al performed a retrospective study among 102 patients with high-grade BS and reported a significantly longer patient interval and secondary care interval for chondrosarcoma versus Ewing sarcoma and osteosarcoma,21 which resulted in a significantly longer total interval, with a mean of 98.3 weeks for chondrosarcoma, versus 22.9 and 23.3 weeks for Ewing sarcoma and osteosarcoma, respectively.…”

Section: Resultsmentioning

confidence: 99%

The sarcoma diagnostic interval: a systematic review on length, contributing factors and patient outcomes

et al. 2020

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Sarcomas are rare and heterogeneous mesenchymal tumours of soft tissue or bone, making them prone to late diagnosis. In other malignancies, early diagnosis has an impact on stage of disease, complexity of therapeutic procedures, survival and health-related quality of life (HRQoL). Little is known about what length of diagnostic interval should be considered as delay in patients with bone (BS) or soft tissue sarcomas (STS). To quantify total interval (defined as time from first symptom to histological diagnosis) and its components, identify contributing factors to its length and determine the impact on patients’ outcome in terms of mortality and HRQoL. A systematic review was conducted according to Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Seventy-six articles out of 2310 met the predefined inclusion criteria. Total intervals, varied broadly; 9–120.4 weeks for BS and 4.3–614.9 weeks for STS. Older age and no initial radiological examinations were contributing factors for a long interval in BS, while in STS results were conflicting. The impact of length of total interval on clinical outcomes in terms of survival and morbidity remains ambiguous; no clear relation could be identified for both BS and STS. No study examined the impact on HRQoL. The length of total interval is variable in BS as well as STS. Its effect on outcomes is contradictory. There is no definition of a clinically relevant cut-off point that discriminates between a short or long total interval. Prospero: CRD42017062492.

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“…Osteosarcoma (OS) is the most common primary malignant bone tumour among adolescents and young adults (1) and has a high metastatic rate and poor prognosis. With advances in the treatment modality for OS, such as chemotherapy or surgery combined with chemotherapy, the survival of patients with OS has markedly improved in recent years (2), and the 5-year survival rate in patients with non-metastatic OS has reached 60-70% (3). However, the survival of patients with distant metastasis remains poor with the 5-year survival rate rarely reaches 20% (4).…”

Section: Introductionmentioning

confidence: 99%

Ubiquitin‑specific protease 7 promotes osteosarcoma cell metastasis by inducing epithelial‑mesenchymal transition

Zeng

Zhao

et al. 2018

Oncol Rep

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Osteosarcoma (OS) is the most common primary malignant bone tumour among adolescents and young adults; however, its molecular pathogenesis has not been completely elucidated. Ubiquitin-specific protease 7 (USP7), a member of the deubiquitinating enzyme family, plays a role in the malignancy process of various cancer types by targeting the key oncoprotein; however, its biological function and mechanism in OS have not been elucidated. The present study demonstrated that USP7 expression in OS tumour tissues was markedly higher than that in the paired surrounding tissues, and high USP7 expression was positively correlated with the TNM stage and metastasis in patients with OS. Next, biological function assays demonstrated that USP7 knockdown markedly inhibited OS cell migration and invasion, whereas USP7 overexpression enhanced it. Notably, USP7 can directly bind with β-catenin to activate the Wnt/β-catenin signalling pathway and induce epithelial-mesenchymal transition (EMT) of OS cells. Overall, USP7 overexpression could promote OS cell metastasis by activating the Wnt/β-catenin signalling pathway by inducing EMT, suggesting that USP7 is a potential therapeutic target for OS.

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Delay in Diagnosis and Its Effect on Clinical Outcome in High‐grade Sarcoma of Bone: A Referral Oncological Centre Study

Cited by 35 publications

References 32 publications

Prognostic impact of diagnostic and treatment delays in children with osteosarcoma

Prognostic impact of diagnostic and treatment delays in children with osteosarcoma

The sarcoma diagnostic interval: a systematic review on length, contributing factors and patient outcomes

Ubiquitin‑specific protease 7 promotes osteosarcoma cell metastasis by inducing epithelial‑mesenchymal transition

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