Incidence, treatment and survival of paediatric patients with bone sarcomas in Finland from 1991 to 2005

Serlo, Joni; Tarkkanen, Maija; Sampo, Mika; Vettenranta, Kim; Riikonen, Pekka; Helenius, Ilkka

doi:10.1111/apa.12986

Cited by 17 publications

(15 citation statements)

References 19 publications

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“…Thirty‐five studies met the inclusion criteria (Table ): very high HDI (n = 23), high HDI (n = 7), and medium/low HDI (n = 5) . Figure depicts the prevalence of metastatic OS at diagnosis stratified by HDI group.…”

Section: Resultsmentioning

confidence: 99%

“…Thirty-five studies met the inclusion criteria (Table I): very high HDI (n = 23), [1,6,8,10,[13][14][15][16][21][22][23][24][25][26][27][28][29][30][31][32][33][34][35] high HDI (n = 7), [3,11,12,[36][37][38][39] and medium/low HDI (n = 5). [2,[40][41][42][43] Figure 1 depicts the prevalence of metastatic OS at diagnosis stratified by HDI group.…”

Section: Resultsmentioning

confidence: 99%

“…Osteosarcoma (OS) is the most common primary malignant bone tumor in many countries, with a peak in adolescence and often a second smaller peak starting in the sixth decade of life . Despite combined therapies, treatment failure is experienced within 5 years of diagnosis by over 40% of patients, generally due to metastatic disease developed before or after diagnosis .…”

Section: Introductionmentioning

confidence: 99%

“…[7] Survival has not improved substantially over the past 30 years, [5] and metastatic OS is usually incurable and requires palliation. Older age, [6,8,9] axial tumor location, [6,[8][9][10][11][12][13][14][15] and tumor size [1,9,14] have been reported by a number of research groups to increase risk of metastatic disease and worsen survival outcomes. The incidence of OS is fairly constant worldwide, particularly among individuals ࣘ24 years, [4] but an international comparison of the prevalence of metastasis at diagnosis has not been compiled.…”

Section: Introductionmentioning

confidence: 99%

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Prevalence of Metastasis at Diagnosis of Osteosarcoma: An International Comparison

Marko

Diessner

Spector

2016

Pediatric Blood & Cancer

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Background Osteosarcoma is the most common primary malignant bone tumor in many countries, with metastatic disease responsible for most patient deaths. This study compares the prevalence of metastatic osteosarcoma at diagnosis across countries to inform the critical question of whether diagnostic delay or tumor biology drives metastases development prior to diagnosis. Procedure A literature search of the PubMed database was conducted to compare the prevalence of metastatic disease at the time of OS diagnosis between countries. A pooled prevalence with 95% confidence intervals was calculated for each study meeting inclusion criteria. Studies were grouped for analysis based on human development index (HDI) scores. Results Our analysis found an 18% (95% CI: 15%, 20%) average global pooled proportion of metastasis at osteosarcoma diagnosis. The average prevalence of metastasis at diagnosis increased as HDI groupings decreased, with very high HDI, high HDI, and medium/ low HDI groups found to be 15% (95% CI: 13%, 17%), 20% (95% CI: 14%, 28%), and 31% (95% CI: 15%, 52%), respectively. Conclusions Our evidence suggests there is a biological baseline for metastatic OS at diagnosis, which is observed in countries with very high HDI. In countries with medium/ low HDI, where there are more barriers to accessing healthcare, the higher prevalence of metastasis may result from treatment delay or an artificial prevalence inflation due to patients with less severe symptoms not presenting to clinic. Additional research in countries with medium/ low HDI may reveal that earlier detection and treatment could improve patient outcomes in those countries.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Prevalence of Metastasis at Diagnosis of Osteosarcoma: An International Comparison

Marko

Diessner

Spector

2016

Pediatric Blood & Cancer

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“…Bone sarcomas are rarer still with Ewing sarcoma occurring in 1.2 per million children and osteosarcoma in 3.6 per million children 4. They are once-in-a-career diagnoses for the majority of general paediatricians and it is perhaps not surprising that they are easy to miss.…”

Section: Discussionmentioning

confidence: 99%

Navigating chronic pain: how taking a few wrong turns can miss sinister pathology

Turnbull¹,

Dodds²,

Wainwright³

et al. 2017

Arch Dis Child Educ Pract Ed

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Bone sarcomas are rare in childhood, and their presentation can often mimic more benign complaints or chronic musculoskeletal pain. Ewing sarcomas in particular are often diagnosed after a significant delay from the onset of symptoms. At a population level, a long diagnostic delay is not necessarily associated with worse survival , as tumours that display slow growth also tend to be less aggressive. For any specific individual however, a delayed diagnosis can result in a larger tumour that is more difficult to treat. We explore a case of Ewing sarcoma and discuss how the presenting features, approach to imaging and the role of clinician cognitive bias may have led to diagnostic delay.Ewing sarcoma is treated with chemotherapy and surgery and/or radiotherapy based on the initial site of disease, size of tumour and response to initial treatment. With current UK treatments, overall survival is approximately 70% for localised tumours and up to 20% in those with metastatic disease. Bone sarcomas usually present with deep-seated mechanical bone pain akin to toothache. The pain can be intermittent over the course of days or weeks, but pain occurring at night should be considered a red flag. Swelling may also present. On plain X-ray, bone sarcomas can demonstrate areas of bone destruction, new bone formation, periosteal inflammation and soft tissue swelling, but in some cases the changes are very subtle. Persistent unexplained symptoms require MRI to exclude tumours and detect potential benign causes that are amenable to treatment.

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A comparison of risk factors for metastasis at diagnosis in humans and dogs with osteosarcoma

et al. 2019

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Background Canine osteosarcoma (OS) is a relevant spontaneous model for human OS. Identifying similarities in clinical characteristics associated with metastasis at diagnosis in both species may substantiate research aimed at using canine OS as a model for identifying mechanisms driving distant spread in the human disease. Methods This retrospective study included dog OS cases from three academic veterinary hospitals and human OS cases from the Surveillance, Epidemiology, and End Results program. Associations between clinical factors and metastasis at diagnosis were estimated using logistic regression models. Results In humans, those with trunk tumors had higher odds of metastasis at diagnosis compared to those with lower limb tumors (OR = 2.38, 95% CI: 1.51, 3.69). A similar observation was seen in dogs with trunk tumors compared to dogs with forelimb tumors (OR = 3.28, 95% CI 1.36, 7.50). Other associations were observed in humans but not in dogs. Humans aged 20‐29 years had lower odds of metastasis at diagnosis compared to those aged 10‐14 years (OR = 0.67, 95% CI: 0.47, 0.96); every 1‐cm increase in tumor size was associated with a 6% increase in the odds of metastasis at diagnosis (95% CI: 1.04, 1.08); compared to those with a white, non‐Hispanic race, higher odds were observed among those with a black, non‐Hispanic race (OR: 1.51, 95% CI: 1.04, 2.16), and those with a Hispanic origin (OR 1.35, 95% CI: 1.00, 1.81). Conclusion A common mechanism may be driving trunk tumors to progress to detectable metastasis prior to diagnosis in both species.

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Incidence, treatment and survival of paediatric patients with bone sarcomas in Finland from 1991 to 2005

Abstract: The 10-year overall survival of children and adolescents diagnosed with a bone sarcoma in Finland during 1991 to 2005 was very good, at 82% if the disease was localised at diagnosis and 47% if it was metastatic at diagnosis.

Cited by 17 publications

References 19 publications

Prevalence of Metastasis at Diagnosis of Osteosarcoma: An International Comparison

Prevalence of Metastasis at Diagnosis of Osteosarcoma: An International Comparison

Navigating chronic pain: how taking a few wrong turns can miss sinister pathology

A comparison of risk factors for metastasis at diagnosis in humans and dogs with osteosarcoma

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