Has the Affordable Care Act Been Associated with Increased Insurance Coverage and Early-stage Diagnoses of Bone and Soft-tissue Sarcomas in Adults?

BACKGROUND: Treatment of bone and soft-tissue sarcomas can be costly, and therefore, it is not surprising that insurance status of patients is a prognostic factor in determining overall survival. Furthermore, uninsured individuals with suspected bone and/or soft-tissue masses routinely encounter difficulty in obtaining access to basic healthcare (such as office visits, radiology scans), and therefore are more likely to be diagnosed with later stages at presentation. The Patient Protection and Affordable Care Act (ACA) mandate of 2010 aimed to increase access to care for uninsured individuals by launching initiatives, such as expanding Medicaid eligibility, subsidizing private insurance, and developing statewide mandates requiring individuals to have a prescribed minimum level of health insurance. Although prior reports have demonstrated that the ACA increased both coverage and the proportion of early-stage diagnoses among patients with common cancers (including breast, colon, prostate, and lung), it is unknown whether similar improvements have occurred for patients with bone and soft-tissue sarcomas. Understanding changes in insurance coverages and stage at diagnosis of patients with bone and soft-tissue sarcomas would be paramount in establishing policies that will ensure orthopaedic cancer care is made equitable and accessible to all. QUESTIONS/PURPOSES: (1) Has the introduction of the ACA been associated with changes in insurance coverage for adult patients with newly diagnosed bone and soft-tissue sarcomas? (2) Did the introduction of health reforms under the ACA lead to an increased proportion of sarcoma diagnoses occurring at earlier disease stages? METHODS: The 2007 to 2015 Surveillance, Epidemiology and End Results database was queried using International Classification of Diseases for Oncology codes for primary malignant bone tumors of the upper and lower extremity (C40.0 to C40.3), unspecified or other overlapping bone, articular cartilage, and joint and/or ribs, sternum, or clavicle (C40.8 to C40.9, C41.3, and C41.8 to C41.9), vertebral column (C41.2), pelvis (C41.4, C41.8, and C41.9), and soft-tissue sarcomas of the upper or lower extremity and/or pelvis (C49.1, C49.2, and C49.5). A total of 15,287 patients with newly diagnosed cancers were included, of which 3647 (24%) were malignant bone tumors and 11,640 (76%) were soft-tissue sarcomas. The study sample was divided into three cohorts according to specified time periods: pre-ACA from 2007 to 2010 (6537 patients), pre-Medicaid expansion from 2011 to 2013 (5076 patients), and post-Medicaid expansion from 2014 to 2015 (3674 patients). The Pearson chi square tests were used to assess for changes in the proportion of Medicaid and uninsured patients across the specified time periods: pre-ACA, pre-expansion and post-expansion. A differences-in-differences analysis was also performed to assess changes in insurance coverage for Medicaid and uninsured patients among states that chose to expand Medicaid coverage in 2014 under the ACA's provision versus those who opted out of Medicaid expansion. Since the database switched to using the American Joint Commission on Cancer (AJCC) 7th edition staging system in 2010, linear regression using data only from 2010 to 2015 was performed that assessed changes in cancer stage at diagnosis from 2010 to 2015 alone. After stratifying by cancer type (bone or soft-tissue sarcoma), Pearson chi square tests were used to assess for changes in the proportion of patients who were diagnosed with early, late, and unknown stage at presentation before Medicaid expansion (2011-2013) and after Medicaid expansion (2014-2015) among states that chose to expand versus those who did not. RESULTS: After stratifying by time cohorts: pre-ACA (2007 to 2010), pre-expansion (2011 to 2013) and post-expansion (2014 to 2015), we observed that the most dramatic changes occurred after Medicaid eligibility was expanded (2014 onwards), with Medicaid proportions increasing from 12% (pre-expansion, 2011 to 2013) to 14% (post-expansion, 2014 to 2015) (p < 0.001) and uninsured proportions decreasing from 5% (pre-expansion, 2011 to 2013) to 3% (post-expansion, 2014 to 2015) (p < 0.001). A differences-in-differences analysis that assessed the effect of Medicaid expansion showed that expanded states had an increase in the proportion of Medicaid patients compared with non-expanded states, (3.6% [95% confidence interval 0.4 to 6.8]; p = 0.03) from 2014 onwards. For the entire study sample, the proportion of early-stage diagnoses (I/II) increased from 56% (939 of 1667) in 2010 to 62% (1137 of 1840) in 2015 (p = 0.003). Similarly, the proportion of unknown stage diagnoses decreased from 11% (188 of 1667) in 2010 to 7% (128 of 1840) in 2015 (p = 0.002). There was no change in proportion of late-stage diagnoses (III/IV) from 32% (540 of 1667) in 2010 to 31% (575 of 1840) in 2015 (p = 0.13). CONCLUSION: Access to cancer care for patients with primary bone or soft-tissue sarcomas improved after the ACA was introduced, as evidenced by a decrease in the proportion of uninsured patients and corresponding increase in Medicaid coverage. Improvements in coverage were most significant among states that adopted the Medicaid expansion of 2014. Furthermore, we observed an increasing proportion of early-stage diagnoses after the ACA was implemented. The findings support the preservation of the ACA to ensure cancer care is equitable and accessible to all vulnerable patient populations. LEVEL OF EVIDENCE: Level III, therapeutic study.

Career and Financial Situation of Patients Diagnosed with Soft Tissue Sarcomas.

BACKGROUND: After receiving a cancer diagnosis patients face many challenges. The association between career situation and financial problems caused by cancer has a substantial impact on quality of life (QoL) among cancer patients. Indeed, the QoL such as physical and mental health of cancer patients is lower when the risk of psychological disorders or distress increases, and chances for cancer cure are reduced. Progress in therapeutic intervention allows many cancer patients a social reintegration into their careers. About one third of cancer patients are younger than 65 years, and with the constant increase in work life periods, a cancer diagnosis also presents a financial burden for those affected. OBJECTIVES: The main objective of this study was to analyse the social QoL in the context of factors related to career and financial situation among patients diagnosed with soft tissue sarcomas (STS). METHODS: A descriptive non-experimental research design was used to conduct a cross-sectional survey over a period of 6 months, between May and November 2016, in collaboration with the Sarcoma Unit at the Mannheim University Medical Centre, Mannheim, Germany, the patient organisation "Das Lebenshaus e.V.," and the German Pension Insurance Hessen (Deutsche Ren-tenversicherung: DRV Hessen)/Pension Fund. We analysed data of 30 patients diagnosed with STS using self-outcome questionnaires in combination with retirement insurance data from the date of first diagnosis up to 3 years afterwards. Out of 280 questionnaires, we received 86 completed forms, of which 56 were excluded. The remaining questionnaires of 30 patients were analysed according to self-determined outcomes and included a calculation of the financial changes caused by the disease. Only patients covered by pension insurance were included in the study. RESULTS: Thirty patients (24 women) whose median age at first diagnosis was 48.7 years (range 31-61 years) were included in the analysis. The average unemployment rate was 8.8 months, and for 67% (20 patients) the employment situation changed after the period of unemployment. Eight patients requested a retirement pension (reduced income insurance), 4 patients reduced their weekly working hours, and 3 patients lost their jobs due to complications of the disease. The data analysis revealed that, among these patients, one benefited from an income increase of about 24%, another one received a regular old-age pension, and 4 patients reported reduced income for other reasons. In total, mean income has been reduced by 26%. Considering only the 8 patients who applied for a pension, partial or total unemployment benefits resulted in an average loss of income of up to 62%. CONCLUSIONS: Reduced ability to work may cause severe financial problems for those affected by the diagnosis of STS. We found an average income reduction of 26%, for those requesting pension payments of 62%. This eventually relates to a higher risk of reduced wealth and may lower the patients' social standing.

Is There an Association Between Insurance Status and Survival and Treatment of Primary Bone and Extremity Soft-tissue Sarcomas? A SEER Database Study.

BACKGROUND: Several recently published population-based studies have highlighted the association between insurance status and survival in patients with various cancers such as breast, head and neck, testicular, and lymphoma [22, 24, 38, 41]. Generally, these studies demonstrate that uninsured patients or those with Medicaid insurance had poorer survival than did those who had non-Medicaid insurance. However, this discrepancy has not been studied in patients with primary bone and extremity soft-tissue sarcomas, a unique oncological population that typically presents late in the disease course and often requires referral and complex treatment at tertiary care centers-issues that health insurance coverage disparities could aggravate. QUESTIONS/PURPOSES: (1) What is the relationship between insurance status and cause-specific mortality? (2) What is the relationship between insurance status and the prevalence of distant metastases? (3) What is the relationship between insurance status and the proportion of limb salvage surgery versus amputation? METHODS: The Surveillance, Epidemiology, and End Results database (SEER) was used to identify a total of 12,008 patients: 4257 patients with primary bone sarcomas and 7751 patients with extremity soft-tissue sarcomas, who were diagnosed and treated between 2007 and 2014. Patients were categorized into one of three insurance groups: insured with non-Medicaid insurance, insured with Medicaid, and uninsured. Patients without information available regarding insurance status were excluded (2.7% [113 patients] with primary bone sarcomas and 3.1% [243 patients] with extremity soft-tissue sarcomas.) The association between insurance status and survival was assessed using a Cox proportional hazards regression analysis adjusted for patient age, sex, race, ethnicity, extent of disease (lymph node and metastatic involvement), tumor grade, tumor size, histology, and primary tumor site. RESULTS: Patients with primary bone sarcomas with Medicaid insurance had reduced disease-specific survival than did patients with non-Medicaid insurance (hazard ratio 1.3 [95% confidence interval 1.1 to 1.6]; p = 0.003). Patients with extremity soft-tissue sarcomas with Medicaid insurance also had reduced disease-specific survival compared with those with non-Medicaid insurance (HR 1.2 [95% CI 1.0 to 1.5]; p = 0.019). Patients with primary bone sarcomas (relative risk 1.8 [95% CI 1.3 to 2.4]; p < 0.001) and extremity soft-tissue sarcomas (RR 2.4 [95% CI 1.9 to 3.1]; p < 0.001) who had Medicaid insurance were more likely to have distant metastases at the time of diagnosis than those with non-Medicaid insurance. Patients with primary bone sarcomas (RR 1.8 [95% CI 1.4 to 2.1]; p < 0.001), and extremity soft-tissue sarcomas (RR 2.4 [95% CI 1.9 to 3.0]; p < 0.001) that had Medicaid insurance were more likely to undergo amputation than patients with non-Medicaid insurance. Patients with primary bone and extremity soft-tissue sarcomas who were uninsured were not more likely to have distant metastases at the time of diagnosis and did not have a higher proportion of amputation surgery as compared with patients with non-Medicaid insurance. However, uninsured patients with extremity soft-tissue sarcomas still displayed reduction in disease-specific survival (HR 1.6 [95% CI 1.2 to 2.1]; p = 0.001). CONCLUSIONS: Disparities manifested by differences in insurance status were correlated with an increased risk of metastasis at the time of diagnosis, reduced likelihood of treatment with limb salvage procedures, and reduced disease-specific survival in patients with primary bone or extremity soft-tissue sarcomas. Although several potentially confounding variables were controlled for, unmeasured confounding played a role in these results. Future studies should seek to identify what factors drive the finding that substandard insurance status is associated with poorer survival after a cancer diagnosis. Candidate variables might include medical comorbidities, treatment delays, time to first presentation to medical care and time to diagnosis, type of treatment received, distance travelled to treatments and transportation barriers, out-of-pocket payment burden, as well as educational and literacy status. These variables are almost certainly associated with socioeconomic deprivation in a vulnerable patient population, and once identified, treatment can become targeted to address these systemic inequities. LEVEL OF EVIDENCE: Level III, therapeutic study.

Economic evaluation of the GENESIS-SEFH report of olaratumab with doxorubicin in soft tissue sarcoma in advanced stages.

OBJECTIVE: The economic evaluation of the drug olaratumab is carried out in the treatment of soft tissue sarcoma. METHOD: The data were analyzed following the recommendations contained in  the MADRE program of the GENESIS-SEFH report model. RESULTS: Progression free survival and overall survival results published in the  pivotal clinical trial; Tap, WD. et al. (2016) were improvement of 2.5 months in  median progression free survival (primary endpoint) HR = 0.672; IC95%  (0.442-1.021) and gain of 11.8 months in median OS (secondary endpoint) HR  = 0.463; IC95% (0.301-0.710). A cost-effectiveness analyses was performed  considering 2 scenarios; scenario 1: with use of whole vials and scenario 2: use  of whole vials and associating non-pharmacological costs (day hospital visits,  mucositis, neutropenia and dexrazoxane use). In both cases we considered the  cost of drugs and the efficacy data of the pivotal clinical trial. In Scenario 1, we  would have an Incremental-Cost-Effectiveness-Ratio of €28,443.81/month of  progression-free survival and €72,560.74 per year of life gained and in scenario  2 we would have an incremental-cost-effectivenessratio of €30,879.79/  progression-free survival and €78,774.99/ year of life gained. The budgetary  impact of this drug would range from €61,759.592 to €92,639.388 estimated to  be 800 to 1,200 patients likely to receive treatment in Spain. CONCLUSIONS: Olaratumab is a drug that provides a significant benefit in overall  survival but not in progression free survival. To be used in soft tissue sarcoma  and to be cost-effective, the acquisition cost of the 500 mg vial should be  between €101.91 and €506.54 and that of the 190 mg vial between €39.31 and  €195.37.

Objetivo: Desarrollar la evaluación económica del fármaco olaratumab en el  tratamiento del sarcoma de partes blandas.Método: Los datos se analizaron siguiendo las recomendaciones contenidas en  el programa MADRE del modelo de informe GENESIS-SEFH.Resultados: Los resultados de supervivencia libre de progresión y supervivencia global publicados en el ensayo clínico pivotal: Tapm WD. et al.  (2016) fueron: la ganancia en supervivencia libre de progresión (variable  principal) en términos absolutos fue de 2,5 meses, HR = 0,672; IC95% (0,442- 1,021). La ganancia absoluta en supervivencia global (variable secundaria) fue  de 11,8 meses, HR = 0,463; IC95% (0,301-0,710). Se realizó un análisis coste- efectividad considerando dos escenarios; escenario uno: sin aprovechamiento de viales; y escenario dos: sin aprovechamiento de viales y asociando costes no  farmacológicos. En ambos casos se consideraron los costes de adquisición de los medicamentos y los datos de eficacia del ensayo clínico pivotal. En el primero  determinamos una ratio coste-efectividad-incremental de 28.443,81 euros/mes  libre de progresión ganado y 72.560,74 euros/año de vida ganado. En el  segundo obtenemos una ratio coste-efectividad incremental de 30.879,79 euros libre de progresión ganado y 78.774,99 euros/año de vida ganado. El  impacto económico estatal, por tanto, se situaría entre 61.759.592 millones de  euros y 92.639.388 de euros, considerando una población diana de 800-1.200  pacientes a nivel nacional.Conclusiones: Olaratumab es un fármaco que aporta un beneficio significativo  en la supervivencia global, no así en la supervivencia libre de progresión. Para  poder utilizarse en el sarcoma de partes blancas y que resultase costeefectivo, el coste de adquisición del vial de 500 mg debería situarse entre 101,91 y 506,54  euros y el del vial de 190 mg entre 39,31 y 195,37 euros.

The cost-saving effect of centralized histological reviews with soft tissue and visceral sarcomas, GIST, and desmoid tumors: The experiences of the pathologists of the French Sarcoma Group.

OBJECTIVE: This study examined the types of discordance occurring in the diagnosis of soft tissue and visceral sarcomas, gastrointestinal stromal tumors (GIST), and desmoid tumors, as well as the economic impact of diagnostic discrepancies. METHODS: We carried out a retrospective, multicenter analysis using prospectively implemented databases performed on a cohort of patients within the French RRePS network in 2010. Diagnoses were deemed to be discordant based on the 2013 World Health Organization (WHO) classification. Predictive factors of discordant diagnoses were explored. A decision tree was used to assess the expected costs of two strategies of disease management: one based on revised diagnoses after centralized histological review (option 1), the other on diagnoses without centralized review (option 2). Both were defined based on the patient and the disease characteristics, according to national or international guidelines. The time horizon was 12 months and the perspective of the French National Health Insurance (NHI) was retained. Costs were expressed in Euros for 2013. Sensitivity analyses were performed using low and high scenarios that included ± 20% estimates for cost. RESULTS: A total of 2,425 patients were included. Three hundred forty-one patients (14%) had received discordant diagnoses. These discordances were determined to mainly be benign tumors diagnosed as sarcomas (n = 124), or non-sarcoma malignant tumors diagnosed as sarcomas (n = 77). The probability of discordance was higher for a final diagnosis of desmoid tumors when compared to liposarcomas (odds ratio = 5.1; 95%CI [2.6-10.4]). The expected costs per patient for the base-case analysis (low- and high-case scenarios) amounted to €8,791 (€7,033 and €10,549, respectively) for option 1 and €8,904 (€7,057 and €10,750, respectively) for option 2. CONCLUSIONS: Our findings highlight misdiagnoses of sarcomas, which were found to most often be confused with benign tumors. Centralized histological reviews are likely to provide cost-savings for the French NHI.

Clinicians' adherence versus non adherence to practice guidelines in the management of patients with sarcoma: a cost-effectiveness assessment in two European regions.

BACKGROUND: Although the management of sarcoma is improving, non adherence to clinical practice guidelines (CPGs) remains high, mainly because of the low incidence of the disease and the variety of histological subtypes. Since little is known about the health economics of sarcoma, we undertook a cost-effectiveness analysis (within the CONnective TIssue CAncer NETwork, CONTICANET) comparing costs and outcomes when clinicians adhered to CPGs and when they did not. METHODS: Patients studied had a histological diagnosis of sarcoma, were older than 15 years, and had been treated in the Rhône-Alpes region of France (in 2005/2006) or in the Veneto region of Italy (in 2007). Data collected retrospectively for the three years after diagnosis were used to determine relapse free survival and health costs (adopting the hospital's perspective and a microcosting approach). All costs were expressed in euros (€) at their 2009 value. A 4% annual discount rate was applied to both costs and effects. The incremental cost-effectiveness ratio (ICER) was expressed as cost per relapse-free year gained when management was compliant with CPGs compared with when it was not. To capture uncertainty surrounding ICER, a probabilistic sensitivity analysis was performed based on a non-parametric bootstrap method. RESULTS: A total of 219 patients were included in the study. Compliance with CPGs was observed for 118 patients (54%). Average total costs reached 23,571 euros when treatment was in accordance with CPGs and 27,313 euros when it was not. In relation to relapse-free survival, compliance with CPGs strictly dominates non compliance, i.e. it is both less costly and more effective. Taking uncertainty into account, the probability that compliance with CPGs still strictly dominates was 75%. CONCLUSIONS: Our findings should encourage physicians to increase their compliance with CPGs and healthcare administrators to invest in the implementation of CPGs in the management of sarcoma.

[Cost analysis of the diagnosis and treatment of soft tissue sarcomas in reference centres]. / Análisis de los costes de diagnóstico y tratamiento de los sarcomas de partes blandas en centros de referencia.

OBJECTIVE: To determine the diagnostic and treatment costs in patients referred to a reference centre with a suspected soft tissue sarcoma. MATERIAL AND METHODS: The study consisted of a historic cohort of 48 consecutive patients who were diagnosed with soft tissue sarcomas and treated in our centres between the years 2009 and 2011, both prior to and after performing the biopsy. The cost per procedure was taken from the official list of prices published in the year 2009. A comparative study of the data was performed using the Mann-Whitney U and Wilcoxon tests. RESULTS: The mean cost per patient was 14,427.58€. In those referred before the biopsy, the overall mean cost was 11,818.67€, and in those referred afterwards, it was 6,456.74€ (p=.0073). There were no significant differences in the diagnostic costs between the groups. However, the mean cosy of the treatment per patient was higher in the second group (p=.0121). DISCUSSION: The referral to centres with experienced multidisciplinary teams in this disease is a common fact highlighted in many articles, where the financial savings have also been demonstrated. CONCLUSIONS: This study shows that, as well as improving the care aspect of the patients when they are referred to a specialist centre prior to the biopsy, there is also a lower health care cost.

Negative-pressure wound therapy for musculoskeletal tumor surgery.

OBJECTIVE: To evaluate the effect of negative-pressure wound therapy in musculoskeletal tumor surgery patients. MATERIALS AND METHODS: The authors analyzed the medical records of 32 patients treated at the authors' institution for bone and soft-tissue sarcomas and secondary wound-healing complications, from 2005 to 2008; there were 11 men and 21 women, with a mean age of 56 years (range, 35-72 years). RESULTS: A statistically significant difference (P = .036) was found regarding the length of hospitalization in the conventional wound treatment group, group A (mean, 25.2 days; range, 15-52 days), compared with the negative wound pressure therapy group, group B (mean, 16.5 days; range, 12-33 days). CONCLUSIONS: The use of negative-pressure wound therapy for the management of complicated wound healing in sarcoma patients following tumor surgery is safe and effective and is associated with lower overall complications rates, infection rate, and the need for further surgery and a lower total cost of wound-healing treatment.

Trabectedin for the treatment of advanced metastatic soft tissue sarcoma.

This paper presents a summary of the evidence review group (ERG) report into the clinical effectiveness and cost-effectiveness of trabectedin for the treatment of advanced metastatic soft tissue sarcoma, in accordance with the licensed indication, based on the evidence submission from the manufacturer to NICE as part of the single technology appraisal (STA) process. The outcomes stated in the manufacturer's definition of the decision problem were overall survival (OS), progression-free survival (PFS), response rates, adverse effects of treatment, health-related quality of life, and cost per quality-adjusted life-year (QALY) gained. The clinical evidence was derived from one randomised controlled trial (RCT), in which the licensed dose of trabectedin was compared with a different dose of trabectedin, and three phase II studies. In the RCT, the median OS was 13.9 months for the licensed dose of trabectedin, which was not significantly different from that for the comparator dose of trabectedin, which was 11.8 months. From the phase II uncontrolled trials, median OS was reported as 9.2 or 12.8 months. The RCT reported significantly superior PFS for the licensed dose of trabectedin (median 3.3 months) over the comparator trabectedin dose (median 2.3 months). One phase II uncontrolled trial reported median PFS as 1.9 months in the licensed dose of trabectedin. The RCT reported PFS rates at 6 months were 35.5% for the licensed dose of trabectedin, and 27.5% for the comparator dose of trabectedin. From the phase II uncontrolled trials, PFS rates at 6 months were 24.4% or 29%. For the RCT, deaths attributed to trabectedin occurred in 3.1% of the licensed dose, and 2.3% of the comparator group. The most common severe adverse events were neutropenia, although with a low rate of febrile neutropenia, thrombocytopenia, and aspartate aminotransferase and alanine aminotransferase elevation, although these were reported to be non-cumulative and reversible. Following dialogue iterations with the ERG team, the manufacturer revised the model twice. However, despite revisions, errors/inconsistencies were found in the latest version of the model and were corrected by the ERG (only for the base case). In the latest manufacturer's submission, the cost per QALY gained of trabectedin compared with best supportive care (BSC) was estimated to be 56,985 pounds for the base case using effectiveness from the STS (Soft Tissue Sarcomas)-201 trial for trabectedin and a pool analysis of the European Organisation for Research and Treatment of Cancer data set for BSC. This analysis was constrained to patients with L-sarcomas only. When the joint uncertainty between parameters was considered, the cost-effectiveness acceptability curve showed that trabectedin has a very low probability of being cost-effective at a threshold of 30,000 pounds per QALY gained compared with BSC for any scenario. The guidance has yet to be issued by NICE.

MRI surveillance after resection for primary musculoskeletal sarcoma.

This study reports the experience of one treatment centre with routine surveillance MRI following excision of musculoskeletal sarcoma. The case notes, MRI and histology reports for 57 patients were reviewed. The primary outcome was local tumour recurrence detected on either surveillance MRI in asymptomatic patients, or interval MRI in patients with clinical concern. A total of 47 patients had a diagnosis of soft-tissue sarcoma and ten of a primary bone tumour. A total of 13 patients (22%) had local recurrence. Nine were identified on a surveillance scan, and four by interval scans. The cost of surveillance is estimated to be pound4414 per recurrence detected if low-grade tumours with clear resection margins are excluded. Surveillance scanning has a role in the early detection of local recurrence of bone and soft-tissue sarcoma.

Surgical biopsy with intra-operative frozen section. An accurate and cost-effective method for diagnosis of musculoskeletal sarcomas.

The most appropriate protocol for the biopsy of musculoskeletal tumours is controversial, with some authors advocating CT-guided core biopsy. At our hospital the initial biopsies of most musculoskeletal tumours has been by operative core biopsy with evaluation by frozen section which determines whether diagnostic tissue has been obtained and, if possible, gives the definitive diagnosis. In order to determine the accuracy and cost-effectiveness of this protocol we have undertaken a retrospective audit of biopsies of musculoskeletal tumours performed over a period of two years. A total of 104 patients had biopsies according to this regime. All gave the diagnosis apart from one minor error which did not alter the management of the patient. There was no requirement for re-biopsy. This protocol was more labour-intensive and 38% more costly than CT-guided core biopsy (AU$1804 vs AU$1308). However, the accuracy and avoidance of the anxiety associated with repeat biopsy outweighed these disadvantages.

Cost analysis of functional restoration surgery for extremity soft-tissue sarcoma.

BACKGROUND: Limb-sparing surgery, consisting of wide-margin tumor resection and preoperative or postoperative radiotherapy/chemotherapy, has become the surgical treatment of choice for extremity sarcomas. However, adequate tumor resection can sometimes compromise crucial limb function, necessitating functional restoration surgery. The purpose of this study was to determine the cost impact and functional outcomes of such procedures. METHODS: Patients receiving either functional restoration surgery or soft-tissue-only reconstruction following extremity soft-tissue sarcoma excision were identified. Patients were then compared along several dimensions: overall length of stay and its subdivisions, surgical time, and total charges and its subdivisions. Patients' functional outcomes were assessed with the Toronto Extremity Salvage Score. RESULTS: Sixty-seven patients who underwent 69 limb-sparing procedures were identified. Fifteen of these procedures (eight upper extremity, seven lower extremity) required functional restoration surgery; 54 of these procedures (13 upper extremity, 41 lower extremity) required only soft-tissue coverage. In the upper extremity, there was a statistically significant increase in overall length of stay (2.8 days) and its subdivisions, surgical time (3.7 hours), and total charges (12,484 dollars) and its subdivisions associated with performing functional restoration surgery. In lower extremity cases, statistically significant increases were determined in only the total charges (9190 dollars) and medical supply charges (13,204 dollars) following functional restoration. Patients who underwent functional restoration surgery had better postoperative function (mean Toronto Extremity Salvage Score, 82 versus 80), but this difference was not statistically significant. CONCLUSION: Although functional restoration surgery is more costly than soft-tissue reconstruction alone, the authors believe that the associated better functional outcome justifies its performance.

Costs of follow-up after potentially curative treatment for extremity soft-tissue sarcoma.

Soft-tissue sarcoma is an uncommon cancer with the potential for high rates of recurrence after initial therapy. Multiple surveillance strategies have been developed to follow patients after primary treatment. The purpose of this study was to quantify the costs associated with various published post-treatment surveillance strategies. A literature review covering the years 1982-2003 was performed to find all modern published surveillance methods for extremity soft-tissue sarcoma. Only articles describing an explicit 5-year follow-up strategy were included. Total costs of 5-year follow-up were calculated for each strategy using Medicare-allowed charges as a proxy. Thirty-four articles depicting 54 strategies were identified. Total Medicare-allowed charges in year 2003 dollars ranged from 485 dollars for follow-up of low-grade sarcoma to 21,235 dollars for follow-up of high-grade sarcoma, a 42.8-fold cost differential. The average charge for these 54 strategies was 6,401 dollars. Physical examination and chest x-ray were the most commonly used screening modalities. Several guidelines have been proposed for extremity soft-tissue sarcoma patient follow-up, most prominently those of the National Comprehensive Cancer Network. The literature has yet to reflect the consensus these guidelines suggest. This study shows wide disparity in the costs of 54 specific methods of following soft-tissue sarcoma patients. Clinical trials are needed to identify an optimal surveillance strategy, one balancing gains in survival, quality of life, costs, and societal willingness to expend resources. Such trials have not been conducted due to the rarity of extremity soft-tissue sarcomas and the costs associated with conducting long-term trials. Alternatively, prospective evaluation of imaging modalities used in follow-up should be assessed as part of other trials. Computer simulation analysis also holds great promise as an assessment tool for surveillance strategies because patient participation is not required.

Prereferral evaluation of patients with suspected bone and soft tissue tumors.

One hundred consecutive patients referred to an orthopaedic oncology practice for evaluation of suspected bone or soft tissue tumors were studied prospectively. There were 76 patients with bone lesions and 24 patients with soft tissue lesions. At the time of initial consultation, information regarding the referring diagnosis, number of prior physician office visits, and prereferral imaging studies obtained was collected. There were 50 patients with benign tumors, 17 patients with primary malignant tumors, 11 patients with metastatic tumors, and 22 patients with nonneoplastic conditions. The average number of physician visits before referral for the entire group was 4.8 visits and was highest for patients with malignant bone tumors (6.2 visits). Imaging studies obtained before referral included plain radiographs, magnetic resonance imaging, bone scans, computed tomography scans, and ultrasound. None of the plain radiographs were thought to be unnecessary; however, 26 of 76 (34.2%) magnetic resonance imaging scans, 17 of 40 (42.5%) bone scans, and 13 of 36 (36.1%) computed tomography scans were excessive and did not contribute to the evaluation of the lesion. Although only 58% of the study group included patients with benign bone tumors and nonneoplastic bone lesions, they accounted for the majority (79%) of unnecessary imaging studies. Primary care physicians and general orthopaedic physicians were equally likely to order unnecessary imaging studies (48% and 52%, respectively).

Local recurrence of soft tissue sarcoma. A Scandinavian Sarcoma Group Project.

The aim of this project was to investigate the diagnosis, treatment and consequences of local recurrence of soft tissue sarcoma (STS). It is based on patients reported to the Karolinska Hospital Sarcoma Register and the Scandinavian Sarcoma Group Register. Demographic and treatment data, based on 1613 adult patients reported to the Scandinavian Sarcoma Group Register by sarcoma centers in Norway, Sweden and Finland are presented. They all had STS of the extremities or trunk wall, and were diagnosed between 1986 and 1995. One third of the tumors were subcutaneous and two thirds deep-seated. The median size was 7 (1-35) cm and 75% were high grade. Metastases at presentation were diagnosed in 8% of the patients. Two thirds of the patients were referred to a sarcoma center before surgery. The preoperative morphologic diagnosis was made by fine-needle aspiration cytology in 72%. Among patients with final treatment for primary tumor at a sarcoma center (n = 1331), the surgical margins were wide or better in 76% of subcutaneous lesions, and in 58% of deep-seated lesions. Adjuvant radiotherapy has not generally been considered indicated after wide or compartmental excisions in Scandinavia. Overall, 23% of patients managed by surgery had adjuvant radiotherapy. Among patients with an intralesional or marginal excision, 44% had postoperative radiotherapy. Patients treated outside of sarcoma centers were seldom referred for radiotherapy. The crude local recurrence rate was 225/1331 (17%) among the patients with final treatment for primary tumor at a sarcoma center. The local recurrence rate after local surgery for high-malignant deep-seated STS was 103/391 (26%). The rate was 25/64 (39%) after an intralesional/marginal margin without postoperative radiotherapy versus 28/119 (24%) when radiotherapy was given. Fine-needle aspiration cytology (FNAC) was used to diagnose suspected local recurrences. 95 FNAC were performed in 86 patients from Karolinska Hospital. There were 47 local recurrences, of which 44 were diagnosed correctly by FNAC; one biopsy was inconclusive, and two lesions were incorrectly assessed as benign. 39 patients proved to have benign lesions in the scar examined cytologically on 50 occasions. None of the specimens was regarded as malignant, but in 4 cases FNAC was inconclusive. The inconclusive or false cytological diagnoses had no serious clinical consequences. Among 205 patients with local recurrence identified in the SSG Register 1987-1995, 169 patients were surgically treated. An intralesional or marginal margin was achieved in 110 of these patients, 59 of whom were also given radiotherapy. 54 of the 169 patients had a second local recurrence. The second local recurrence rate was 0.50 if the first local recurrence was treated using surgery with a marginal margin alone, compared to 0.28 if treated using either surgery with a marginal margin and radiotherapy, or a wide margin (p = 0.0008). In extremity STS, the amputation rate for local recurrences was 0.22, compared to 0.09 for primary tumors. The overall 5-year MFS was 0.72 (95% CI 0.68-0.76). High histopathological malignancy grade (Relative Risk 3.0; 95% CI 1.5-6.3) and an inadequate surgical margin (2.9; 95% CI 1.8-4.6) were independent risk factors for local recurrence. High histopathological malignancy grade and large tumor size (> 7 cm) were the most important risk factors for metastasis. Local recurrence was associated with an increased risk of metastasis (4.4; 95% CI 2.9-6.8), but an inadequate surgical margin was not a risk factor for metastasis (1.1; 95% CI 0.8-1.7). In conclusion, it is unlikely that local recurrence of STS is a major source of metastases. It nevertheless represents a costly, complicated and emotionally difficult problem. More radical surgical margins would improve the local recurrence rate, but this can hardly be achieved for center-operated patients without increasing the amputation rate. Instead, local control will improve by giving radiotherapy to all patients after marginal surgery, and to selected patients with wide margins. Radiotherapy is indicated especially after a previous open biopsy or when a local recurrence might lead to an amputation. Furthermore, radiotherapy seems indicated after local recurrence, regardless of margin or grade. The most effective way of reducing costs and detriment associated with local recurrence is to increase referral to sarcoma centers before biopsy or surgery as primary surgical margins would then improve.

Consequences of local recurrence of soft tissue sarcoma: 205 patients from the Scandinavian Sarcoma Group Register.

From the Scandinavian Sarcoma Group Register, information on 1,224 surgically-treated patients with soft tissue sarcoma (STS) of the extremity or trunk wall, diagnosed between 1987 and 1995, was collected. 205 patients, one third of whom were referred to a center with a local recurrence, had a total of 284 local recurrences. This analysis describes the treatment for these local recurrences, complications and risk of further recurrences. 169 patients were surgically treated for their first local recurrence. An intralesional or marginal margin was achieved in 110 of these patients, 59 of whom were also given radiotherapy. 54 of the 169 patients had a second local recurrence. The second local recurrence rate was 0.50 if the first local recurrence had been treated with only surgery with a marginal margin, compared to 0.28 if treated with surgery with a marginal margin and radiotherapy or with a wide margin (p = 0.0008). In extremity STS, the crude amputation rate for local recurrences was 0.22 (31 of 142)-i.e., higher than for primary tumors 0.09 (96 of 1065) (p < 0.0001). A high local recurrence rate after treatment outside of sarcoma centers has earlier been shown. We conclude that the consequences of local recurrence in terms of morbidity and costs justifies referral of STS patients for multidisciplinary evaluation and multimodality treatment.

Effective follow-up strategies in soft tissue sarcoma.

The value of surveillance for detection of recurrences in patients with soft tissue sarcoma (STS) after definitive surgical resection of the primary tumor is based on the premise that early recognition and treatment of local or distant recurrence can prolong survival. Surveillance strategies should meet the criteria of easy implementation, accuracy, and cost-effectiveness. Although guidelines have been proposed for follow-up of patients with STS, there are few data in the medical literature on the effectiveness of these recommendations. We reviewed the effectiveness of a surveillance program for primary extremity STS in an effort to provide an evidence-based rationale for follow-up of STS. We concluded that clinical assessment of patient symptoms, chest X-ray imaging, and physical examination are effective strategies for follow-up of extremity STS. Chest X-ray imaging also appears to be cost-effective, at least for high-grade extremity STS. Imaging of the primary extremity site by computed tomography (CT) scan or magnetic resonance imaging (MRI) on an annual basis and routine laboratory blood tests were ineffective strategies for recurrence detection. However, certain patient characteristics such as body habitus, previous radiation therapy, and location of the primary tumor site may require the use of CT scans and MRI for adequate clinical assessment. The role of specific surveillance strategies for recurrence detection for sarcomas of the trunk, head and neck, retroperitoneum, and viscera has yet to be defined.