Bone tumours in European children and adolescents, 1978–1997. Report from the Automated Childhood Cancer Information System project

Stiller, Charles; Bielack, Stefan; Jundt, Gernot; Steliarova‐Foucher, Eva

doi:10.1016/j.ejca.2006.05.015

Cited by 230 publications

(151 citation statements)

References 28 publications

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“…Bone tumours in this age group have been previously reported to occur in the United Kingdom at an incidence rate of 5 per million (Stiller, 2007). In addition, the increasing preponderance with age, the equal occurrence of male and female cases in the ages 0 -14 years and the predominance of osteosarcoma and Ewing sarcoma found in this study are also typical patterns reported in earlier studies (Parkin et al, 1998;Stiller et al, 2006a).…”

Section: Discussionsupporting

confidence: 75%

“…The overall incidence rate was 4.84 per million, similar to that reported worldwide in countries with predominantly white populations (Parkin et al, 1998;Stiller et al, 2006a). Bone tumours in this age group have been previously reported to occur in the United Kingdom at an incidence rate of 5 per million (Stiller, 2007).…”

Section: Discussionsupporting

confidence: 65%

“…Among European countries (19 countries), 5-year survival for all childhood bone tumours improved from 37% in 1978 -1982 to 63% in 1993 -1997. For osteosarcoma and Ewing sarcoma, 5-year survival also improved from 37 to 61% and from 34 to 66%, respectively over this period (Stiller et al, 2006a). We describe the incidence and survival from bone tumours at ages 0 -14 years in northern England and the West Midlands during the period 1981-2002, updating previous analyses (Cotterill et al, 2000Stiller et al, 2006c).…”

supporting

confidence: 57%

“…Worldwide, trends in incidence rates for childhood bone tumours have remained constant (McNally et al, 2001;Stiller et al, 2006a;Magnanti et al, 2008), although a few studies have reported increases (Ajiki et al, 1994;Gurney et al, 1996). The substantial improvement in survival over the past 40 years (Arndt et al, 2007) has been attributed to chemotherapy (Stiller et al, 2006a). Among European countries (19 countries), 5-year survival for all childhood bone tumours improved from 37% in 1978 -1982 to 63% in 1993 -1997.…”

mentioning

confidence: 99%

“…The majority of these tumours are osteosarcoma or Ewing sarcoma (Stiller et al, 2006a), in the United Kingdom 54% being osteosarcoma, 39% Ewing sarcoma, 2% chondrosarcoma and 5% other types (Stiller, 2007). Worldwide, trends in incidence rates for childhood bone tumours have remained constant (McNally et al, 2001;Stiller et al, 2006a;Magnanti et al, 2008), although a few studies have reported increases (Ajiki et al, 1994;Gurney et al, 1996). The substantial improvement in survival over the past 40 years (Arndt et al, 2007) has been attributed to chemotherapy (Stiller et al, 2006a).…”

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confidence: 99%

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Incidence and survival of childhood bone cancer in northern England and the West Midlands, 1981–2002

et al. 2009

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There is a paucity of population-based studies examining incidence and survival trends in childhood bone tumours. We used high quality data from four population-based registries in England. Incidence patterns and trends were described using Poisson regression. Survival trends were analysed using Cox regression. There were 374 cases of childhood (ages 0 -14 years) bone tumours (206 osteosarcomas, 144 Ewing sarcomas, 16 chondrosarcomas, 8 other bone tumours) registered in the period 1981 -2002. Overall incidence (per million person years) rates were 2.63 (95% confidence interval (CI) 2.27 -2.99) for osteosarcoma, 1.90 (1.58 -2.21) for Ewing sarcoma and 0.21 (0.11 -0.31) for chondrosarcoma. Incidence of Ewing sarcoma declined at an average rate of 3.1% (95% CI 0.6 -5.6) per annum (P ¼ 0.04), which may be due to tumour reclassification, but there was no change in osteosarcoma incidence. Survival showed marked improvement over the 20 years (1981 -2000) for Ewing sarcoma (hazard ratio (HR) per annum ¼ 0.95 95% CI 0.91 -0.99; P ¼ 0.02). However, no improvement was seen for osteosarcoma patients (HR per annum ¼ 1.02 95% CI 0.98 -1.05; P ¼ 0.35) over this time period. Reasons for failure to improve survival including potential delays in diagnosis, accrual to trials, adherence to therapy and lack of improvement in treatment strategies all need to be considered.

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Section: Discussionsupporting

confidence: 75%

Section: Discussionsupporting

confidence: 65%

supporting

confidence: 57%

mentioning

confidence: 99%

mentioning

confidence: 99%

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Incidence and survival of childhood bone cancer in northern England and the West Midlands, 1981–2002

et al. 2009

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DDRGK1 Enhances Osteosarcoma Chemoresistance via Inhibiting KEAP1‐Mediated NRF2 Ubiquitination

et al. 2023

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Chemoresistance is the main obstacle in osteosarcoma (OS) treatment; however, the underlying mechanism remains unclear. In this study, it is discovered that DDRGK domain‐containing protein 1 (DDRGK1) plays a fundamental role in chemoresistance induced in OS. Bioinformatic and tissue analyses indicate that higher expression of DDRGK1 correlates with advanced tumor stage and poor clinical prognosis of OS. Quantitative proteomic analyses suggest that DDRGK1 plays a critical role in mitochondrial oxidative phosphorylation. DDRGK1 knockout trigger the accumulation of reactive oxygen species (ROS) and attenuate the stability of nuclear factor erythroid‐2‐related factor 2 (NRF2), a major antioxidant response element. Furthermore, DDRGK1 inhibits ubiquitin‐proteasome‐mediated degradation of NRF2 via competitive binding to the Kelch‐like ECH‐associated protein 1 (KEAP1) protein, which recruits NRF2 to CULLIN(CUL3). DDRGK1 knockout attenuates NRF2 stability, contributing to ROS accumulation, which promotes apoptosis and enhanced chemosensitivity to doxorubicin (DOX) and etoposide in cancer cells. Indeed, DDRGK1 knockout significantly enhances osteosarcoma chemosensitivity to DOX in vivo. The combination of DDRGK1 knockdown and DOX treatment provides a promising new avenue for the effective treatment of OS.

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Mesenchymal chondrosarcoma of soft tissues and bone in children, adolescents, and young adults

Dantonello

Int-Veen

Leuschner

et al. 2008

Cancer

138

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BACKGROUND. Mesenchymal chondrosarcoma (MCS) is a rare tumor with a strong tendency toward late recurrences leading to reported 10‐year survival rates below 50%. The recommended treatment is resection with wide margins; the effectiveness of chemo‐ and radiotherapy remain poorly defined. As reports about MCS in young patients are scarce, treatment and outcomes of children/adolescents/young adults in the CWS and COSS studies were investigated. METHODS. Since 1977, 15 of >7000 CWS and COSS patients ≤25 years had a confirmed diagnosis of MCS. RESULTS. The median age was 16.6 (range, 1–25) and median follow‐up 9.6 years (range, 1–22). Four MCS were osseous and 11 extraosseous. All but 1 patient had nonmetastatic disease. Tumor sites were head/neck (n = 6), paravertebral (n = 3), pelvis (n = 3), limbs (n = 2), and kidney (n = 1). All tumors were resected, but only 8 completely. Thirteen individuals received chemotherapy, 6 were irradiated. Actuarial 10‐year event‐free and overall survival rates were 53% and 67%, respectively. Four recurrences occurred, all within 4 years from diagnosis (3 local, 1 combined; 2 of these in irradiated patients). One of these patients survived after surgery and radiation for local recurrence. Seven of 8 patients whose tumors were completely resected during primary treatment, but only 4 of 7 patients with incomplete surgery survived disease‐free. CONCLUSIONS. This series of 15 MCS differs from others: the median age was young, most tumors were extraosseous, most patients received chemotherapy, and the outcome was better than published. Despite long‐term follow‐up, characteristic late metastatic recurrences were not observed. Treating MCS according to standard multimodal soft tissue/bone sarcoma regimens is proposed. Cancer 2008. © 2008 American Cancer Society.

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Bone tumours in European children and adolescents, 1978–1997. Report from the Automated Childhood Cancer Information System project

Cited by 230 publications

References 28 publications

Incidence and survival of childhood bone cancer in northern England and the West Midlands, 1981–2002

Incidence and survival of childhood bone cancer in northern England and the West Midlands, 1981–2002

DDRGK1 Enhances Osteosarcoma Chemoresistance via Inhibiting KEAP1‐Mediated NRF2 Ubiquitination

Mesenchymal chondrosarcoma of soft tissues and bone in children, adolescents, and young adults

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