Osteosarcoma Survivors Research Articles

Predictors of inactive lifestyle among adult survivors of childhood cancer

: Participation in physical activity is important for childhood cancer survivors, because inactivity may compound cancer/treatment-related late effects. However, some survivors may have difficulty participating in physical activity, and these individuals need to be identified so that risk-based guidelines for physical activity, tailored to specific needs, can be developed and implemented. The objectives of the current study were to document physical activity patterns in the Childhood Cancer Survivor Study (CCSS) cohort, to compare the physical activity patterns with siblings in the CCSS and with a population-based sample from the Behavioral Risk Factor Surveillance System, and to evaluate associations between diagnosis, treatment, and personal factors in terms of the risk for an inactive lifestyle. : Percentages of participation in recommended physical activity were compared among survivors, siblings, and population norms. Generalized linear models were used to evaluate the associations between cancer diagnosis and therapy, sociodemographics, and the risk for an inactive lifestyle. : Participants included 9301 adult survivors of childhood cancer and 2886 siblings. Survivors were less likely than siblings (46% vs 52%) to meet physical activity guidelines and were more likely than siblings to report an inactive lifestyle (23% vs 14%). Medulloblastoma (35%) and osteosarcoma (27%) survivors reported the highest levels of inactive lifestyle. Treatments with cranial radiation or amputation were associated with an inactive lifestyle as were being a woman, black race, older age, lower educational attainment, underweight or obese status, smoking, and depression. : Childhood cancer survivors were less active than a sibling comparison group or an age- and sex-matched population sample. Survivors who are at risk for an inactive lifestyle should be considered high priority for developing and testing of intervention approaches. Cancer 2009. (c) 2009 American Cancer Society.

“So, are you back to work yet?” Re-conceptualizing ‘work’ and ‘return to work’ in the context of primary bone cancer

People with primary bone cancer typically are young (usual age-at-onset 16–35 years old) and undergo arduous treatments. The current standard of care (tumour resection and limb reconstruction with or without chemotherapy) results in survival rates in excess of 60%, but also results in significant disability at a time when patients are choosing career paths, establishing their independence and embarking on new roles. To date, the nature of the relationship between experiences of osteosarcoma illness and experiences of vocation has remained unclear. This study sought to examine this relationship using qualitative narrative methodology. In-depth audiotaped interviews were conducted with 14 osteosarcoma survivors (8 men, 6 women) who were being treated at Mount Sinai Hospital, Toronto, Canada. Interview transcripts were analyzed for story typology and thematic content via constant comparison. Respondents reported engaging in three types of ‘work’: ‘illness work’, ‘identity work’ and ‘vocational work’. Osteosarcoma illness represented a crisis for respondents, one which necessitated considerable illness work. Illness work was portrayed as all-consuming, whereby respondents were forced to stop vocational work for considerable periods. The illness crisis also precipitated ‘identity work’. Respondents recounted a transformative process, of ‘becoming other’ to whom they had been prior to illness. As a result, respondents told of re-entering the vocational sphere with a different sense of themselves from when they left it. When patients return for surgical follow up, clinicians routinely ask, “So, are you back to work yet?” expecting simple ‘yes/no’ answers. This study suggests that the answer is instead highly complex, and that patients could be seen as having been ‘working’ all along. This study offers a re-conceptualization of ‘work’ and ‘return to work’ in the context of osteosarcoma, with implications for clinical and return-to-work practices.

Education and Employment in Long-Term Survivors of High-Grade Osteosarcoma: A Japanese Single-Center Experience

Objective: To investigate the status of education and employment of long-term survivors who became physically handicapped after treatment for high-grade osteosarcoma. Methods: Of the osteosarcoma patients treated at our hospital, 41 patients aged less than 18 years at the initial presentation who were free of disease for 10 years or longer after the end of treatment were studied. The status of their education and employment was investigated via a questionnaire. Results: Twenty-seven patients responded to the questionnaire (response rate, 65.9%). Of these patients, 73.1% (19/26) could return to the school they had attended before the disease, and 52% (13/25) graduated from college or university. The percentage of those who went to college or university was higher in the limb-sparing group. Seventy-two percent of the patients were engaged in clerical work, and the mean annual income was 4.01 million JPY (corresponding to about 24,000 EUR). No difference was noted in the status of employment between the amputation and limb-sparing groups. Conclusions: The percentage of patients who went to college or university was similar to the percentage in all Japanese. However, the status of the diseased limb appeared to affect school attendance. The mean annual income of the patients was comparable to that of the national average, and they experienced no major problems in their employment. Physical disabilities posed few problems in their daily living.

Impact of family structure on long-term survivors of osteosarcoma

Long-term outcomes of osteosarcoma have dramatically improved with the use of modern combination therapies. Such aggressive treatments, however, entail chronic complications. In the present study, we assessed the functional, psychological, and familial status of long-term survivors of osteosarcoma treated at our institution. Fifteen long-term survivors of osteosarcoma were evaluated for functional and psychological sequelae. Functional assessment was based on a method described by Enneking et al. Psychological assessment was based on General Health Questionnaire 28, Inventory Scale for Traumatic Neurosis, and Family System Test. Ten patients showed mild functional impairments; only five patients were handicapped more seriously. Depressive symptoms were diagnosed in four patients. A total of six patients revealed unbalanced family structures, including three of the four patients with depressive symptoms, all four patients with symptoms of posttraumatic stress disorder, and five of seven patients who showed poor emotional acceptance. Osteosarcoma survivors will generally recover good functional performance. Only a minority of them remain seriously impaired. One third of the patients present depressive symptoms and posttraumatic stress disorder. Poor coping is closely associated with unbalanced family structures. Therefore, the psychological and familial situation of patients with newly diagnosed osteosarcoma should be carefully assessed.

Follow-up study of long-term survivors of osteosarcoma in the prechemotherapy era

Follow-up study of long-term survivors of osteosarcoma in the prechemotherapy era

Follow-up study of long-term survivors of osteosarcoma in the prechemotherapy era

Osteosarcoma is the most common primary bone sarcoma. Several studies published in the 1960s established that approximately one fifth of patients survive when treated with surgery alone. There is no information, however, about the long-term consequences of osteosarcoma. It is especially relevant to know if these patients are at risk for a second malignancy. We reviewed all clinical records from long-term (defined as more than 10 years) osteosarcoma survivors treated at Mayo Clinic in the prechemotherapeutic era from 1900 to 1960. We re-reviewed histological sections for most cases. Patients or next of kin provided follow-up information during telephone interviews. Rates of second malignancy were compared with expected rates in the population at large. We identified 465 patients treated for osteosarcoma. Of these patients, 83 (17.8%) were long-term survivors, including 19 who were alive up to 65 years after treatment. Of the 7 patients with pulmonary metastases, 3 died. A second malignancy developed in 26 patients, 15 of whom died of the malignancy. Although long-term survivors of osteosarcoma have a higher incidence of a second malignant tumor than a normal population, this increase was not statistically significant. No demographic or histological variables predicted long-term survival.

An examination of outcome measures to assess functional mobility in childhood survivors of osteosarcoma.

The purpose of this study is to examine outcome measures that assess functional mobility in adolescent survivors of osteosarcoma. We will also identify relationships between functional mobility measurements and specific measure(s) of health-related quality of life (HRQL). Eighteen adolescent survivors of osteosarcoma at least 2 years from surgery at the Children's Hospital of Philadelphia (CHOP) participated in this study. Functional mobility, endurance, perceived exertion, and locomotor efficiency is examined with the following measurement tools: timed up and go (TUG), timed up and down stairs (TUDS), 9-min run-walk, rate of perceived exertion (RPE), and physiological cost index (PCI). The currently used evaluation of function from the American Musculoskeletal Tumor Society (MSTS) is also administered. The Short Form 36 (SF-36) measuring HRQL is used. MSTS scores did not correlate with quantitative functional outcome measures. MSTS scores did not correlate with the HRQL as measured by the SF-36. In contrast, subjects with a higher rate of vitality on the SF-36 walked with more efficiency, farther distance during the 9-min run-walk, and performed the TUDS and TUG with a faster speed. The MSTS does not correlate with actual measures of function or with HRQL. These data demonstrate the need for a new measure to determine functional mobility in survivors of osteosarcoma.

Restoration of ovarian function after chemotherapy for osteosarcoma

Aim: To evaluate ovarian function after modern intensive multi-agent chemotherapy for osteosarcoma given during childhood or adolescence. Methods: After discontinuation of treatment, 10 female osteosarcoma survivors were followed up for...

Second malignant neoplasms in long-term survivors of osteosarcoma: Memorial Sloan-Kettering Cancer Center Experience.

The authors investigated the incidence and relative risk of secondary malignant neoplasms in long-term survivors of osteosarcoma. A comprehensive list of 509 patients with primary osteosarcoma treated at our institution between February 1973 and March 2000 was identified. All study patients received chemotherapy and/or surgery on one of six different protocols (T4, 5, 7, 10, 12, and CCG-7921/POG-9351). Chemotherapy was scheduled for up to 40 weeks with some variations in the actual treatment period and consisted of various combinations of the following agents: high-dose methotrexate, doxorubicin, bleomycin, cyclophosphamide, dactinomycin, vincristine, cisplatin, and ifosfamide. Secondary malignant neoplasms (SMN) occurred in 14 of 509 patients. Only one had pulmonary metastasis at diagnosis and subsequent multiple recurrences that required thoracotomies and further modification of the chemotherapy regimen. The median age at diagnosis for osteosarcoma was 16.6 years (range, 3.1-74.4 years). The median follow-up was 5.2 years (range, 0.1-25.0 years). The time interval from diagnosis of the primary osteosarcoma to the development of SMN was 1.3-13.1 years (median, 5.5; 95% confidence interval [CI], 3.6-9.6). The most common SMN occurred in the central nervous system (n = 4): anaplastic glioma, meningioma, high-grade glioma, and maxillary astrocytoma. There were two cases of acute myeloid leukemia and one case each of myelodysplastic syndrome, non-Hodgkin lymphoma, high-grade pleomorphic sarcoma, leiomyosarcoma, fibrosarcoma, breast carcinoma, and mucoepidermoid carcinoma. The overall 5 and 10-year cumulative incidences of SMNs were 1.4% +/- 1.1% and 3.1% +/- 1.8%. The standardized incidence ratio was 4.6 (95% CI, 2.53-7.78, P = 0.00001) for the cohort and 3.64 (95% CI, 1.82-6.52, P = 0.0007) when patients with a history of retinoblastoma or Rothmund-Thomson syndrome were excluded. The overall incidence of secondary malignancies in long-term survivors of osteosarcoma was significantly higher than the expected incidence of cancer in the general population. However, the standardized incidence ratios were much lower than those reported for Hodgkin disease and retinoblastoma. Although additional follow-up is warranted, the successes of current treatment regimens consisting of intensive, high-dose chemotherapy in combination with topoisomerase II inhibitors outweigh the risks.

Second malignant neoplasms occuring in survivors of osteosarcoma.

Second malignant neoplasms have been noted infrequently in survivors of osteosarcoma treated before 1970. For the above reason, the authors surveyed their patients to determine the actuarial incidence and relative risk of second malignancies among patients treated with adjuvant chemotherapy for osteosarcoma. Between March 1962 and March 1996, 334 patients received chemotherapy for newly diagnosed primary or metastatic osteosarcoma. Of these patients, 47 presented with metastases, 14 had multifocal osteosarcoma, and 273 had localized disease. Nine patients developed second malignant neoplasms 0.45-17.8 years (median, 6.3 years) after receiving definitive surgery and adjuvant chemotherapy for primary osteosarcoma; 2 of these patients had pulmonary metastasectomies before receiving adjuvant chemotherapy. The second neoplasms comprised two cases of malignant fibrous histiocytoma and one case each of melanoma, glioblastoma multiforme, chondrosarcoma, and carcinoma of the breast: stomach, colon, rectum. The overall 10-year cumulative incidence of second malignancies was 2% +/- 1%; by comparison, this rate was 2% +/- 1% for patients with localized osteosarcoma but was 8% +/- 5% (P = 0.15) for those who presented with metastatic disease. Since the advent of successful adjuvant chemotherapy, more patients are surviving primary osteosarcoma; therefore, the number of osteosarcoma patients who develop second malignancies can be expected to increase. Recognition of osteosarcoma patients who are members of families with Li-Fraumeni syndrome may lead to earlier intervention for these individuals.

Long‐term pulmonary toxicity of multiagent chemotherapy including bleomycin and cyclophosphamide in osteosarcoma survivors

Purpose To assess long-term pulmonary effects of multiagent chemotherapy, we studied serial pulmonary function tests (PFTs) of 35 children with osteosarcoma up to 12 years after diagnosis. Patients and Methods We analyzed 84 sets of PFTs from 35 patients diagnosed with osteosarcoma between 1981 and 1991. They received bleomycin, cyclophosphamide, methotrexate, doxorubicin, cisplatin, and actinomycin D over 9–12 months and we performed PFTs from 3 days to 152 months after diagnosis. Time period I included 36 PFTs (43%) performed between 1 and 5 months from diagnosis, time period II included 20 PFTs (24%) performed between 8 and 12 months from diagnosis, and time period III included 28 PFTs (33%) performed between 12 and 119 months from diagnosis. Total lung capacity (TLC), forced vital capacity (FVC), forced expiratory volume in 1 second (FEV1), and carbon monoxide diffusing capacity (DLCO) were analyzed. Maximal respiratory pressures and arterial blood gases were measured to assess muscle weakness and gas exchange, respectively. Mean differences in PFTs were compared among the three time periods and between time period pairs. Results All mean PFT values showed significant differences among time periods. Significant decline in DLCO; (P = .012), TLC (P = .020), and FEV1 (P = .028) between time periods I and II were noted followed by a trend towards recovery between time periods II and III. Time periods I and III were not significantly different from one another. Mean PFTs performed after 2 years of diagnosis were not different from mean PFTs performed from diagnosis to 2 years. Conclusion This dosage regimen of multiagent chemotherapy for osteosarcoma patients caused a transient, but significant, decline in PFTs within 8–12 months after administration but appears to cause no significant long-term pulmonary function abnormalities. © 1996 Wiley-Liss, Inc.

Long-term pulmonary toxicity of multiagent chemotherapy including bleomycin and cyclophosphamide in osteosarcoma survivors.

To assess long-term pulmonary effects of multiagent chemotherapy, we studied serial pulmonary function tests (PFTs) of 35 children with osteosarcoma up to 12 years after diagnosis. We analyzed 84 sets of PFTs from 35 patients diagnosed with osteosarcoma between 1981 and 1991. They received bleomycin, cyclophosphamide, methotrexate, doxorubicin, cisplatin, and actinomycin D over 9-12 months and we performed PFTs from 3 days to 152 months after diagnosis. Time period I included 36 PFTs (43%) performed between 1 and 5 months from diagnosis, time period II included 20 PFTs (24%) performed between 8 and 12 months from diagnosis, and time period III included 28 PFTs (33%) performed between 12 and 119 months from diagnosis. Total lung capacity (TLC), forced vital capacity (FVC), forced expiratory volume in 1 second (FEV1), and carbon monoxide diffusing capacity (DLCO) were analyzed. Maximal respiratory pressures and arterial blood gases were measured to assess muscle weakness and gas exchange, respectively. Mean differences in PFTs were compared among the three time periods and between time period pairs. All mean PFT values showed significant differences among time periods. Significant decline in DLCO; (P=.012), TLC (P=.020), and FEV1 (P=.028) between time periods I and II were noted followed by a trend towards recovery between time periods II and III. Time periods I and III were not significantly different from one another. Mean PFTs performed after 2 years of diagnosis were not different from mean PFTs performed from diagnosis at 2 years. This dosage regimen of multi-agent chemotherapy for osteosarcoma patients caused a transient, but significant, decline in PFTs within 8-12 months after administration but appears to cause no significant long-term pulmonary function abnormalities.

Psychological defenses of young osteosarcoma survivors

AbstractTwo self‐report measures—one assessing psychiatric symptoms (SCL‐90) and the other ego defenses (DSQ)—were completed by 16 young survivors of osteosarcoma and 72 healthy controls. The survivors were found to be well adjusted, with no evidence of the self‐report being biased by defensiveness. There was one difference in the way the two study groups described themselves, however. Though far from conclusive, the finding seems to suggest that survivors have problems in trusting that they can obtain support and understanding from other people when they need it most. Possibly one of the most vulnerable areas where the negative impact of major illness may become evident is the capacity to feel secure in interpersonal relationships.

Late effects of therapy in survivors of childhood and adolescent osteosarcoma.

Adult survivors of childhood or adolescent osteosarcoma require ongoing medical follow-up in order to monitor for potentially life-threatening consequences of therapy, including second cancers and anthracycline cardiotoxicity. In the future, additional knowledge of tumor biology will likely change staging methods and allow intensive therapy to be given only to those most likely to benefit from it [25]; those with less risk of relapse may require less toxic therapy and still achieve acceptable levels of survival.

Late effects of therapy in adult survivors of osteosarcoma and Ewing's sarcoma

To study the late consequences of primary bone cancer, we interviewed 82 osteosarcoma and 29 Ewing's sarcoma survivors regarding their health, fertility and offspring, employment, annual income, and activities of daily living. All subjects had been diagnosed before age 20 (mean age, 14.6 years), had survived at least 5 years from diagnosis, and were at least 21 years of age. On average, they were 32.5 years of age at interview. As controls, 151 siblings were interviewed. During the follow-up period, eight survivors had died, and eight survivors had been diagnosed with a second malignancy (7.2%; P = .002). No other health condition distinguished survivors from controls. Although the survivors were more likely than controls to have some difficulty climbing stairs and to have had employment disability, employment status and annual income at follow-up were similar. Deficits in marriage and fertility were not significant. Adult survivors of primary bone tumors diagnosed during childhood or adolescence are at high risk for second malignancies and premature death, making continued medical follow-up of utmost importance. Despite the physical impairment following limb amputation for many, the majority of outcomes we measured did not differ from controls, suggesting few adverse psychosocial outcomes in this group of cancer survivors.