Stigma in Pediatric Cancer: An Exploratory Study of Osteosarcoma and Retinoblastoma in Guatemala, Jordan, and Zimbabwe.

PURPOSE: Stigma is an understudied barrier to health care acceptance in pediatric oncology. We sought to explore the stigma experience, including its impact on cancer treatment decision making, and identify strategies to mitigate stigma for patients with osteosarcoma and retinoblastoma in Guatemala, Jordan, and Zimbabwe. METHODS: Participants included caregivers, adolescent patients (age 12-19 years), and health care clinicians. A semistructured interview guide based on The Health Stigma and Discrimination Framework (HSDF) was adapted for use at each site. Interviews were conducted in English, Spanish, Arabic, or Shona, audio-recorded, translated, and transcribed. Thematic analysis focused on stigma practices, experiences, outcomes, drivers, mitigators, and interventions. RESULTS: We conducted 56 interviews (28 caregivers, 19 health care clinicians, nine patients; 20 in Guatemala, 21 in Jordan, 15 in Zimbabwe). Major themes were organized into categories used to adapt the HSDF to global pediatric cancer care. Themes were described similarly across all sites, ages, and diagnoses, with specific cultural nuances noted. Pediatric cancer stigma was depicted as an isolating and emotional experience beginning at diagnosis and including internalized and associative stigma. Stigma affected decision making and contributed to negative outcomes including delayed diagnosis, treatment abandonment, regret, and psychosocial fragility. Overcoming stigma led to positive outcomes including resilience, treatment adherence, pride, and advocacy. Identified stigma drivers and mitigators were linked to potential interventions. CONCLUSION: Participants describe a shared stigma experience that transcends geography, cultural context, age, and diagnosis. Stigma manifestations have the potential to impact medical decision making and affect long-term psychological outcomes. Stigma assessment tools and interventions aimed at stigma mitigation including educational initiatives and support groups specific to pediatric cancer should be the focus of future research.

A Specific, Measurable, Achievable, Relevant, Timed (SMART) Nursing Project Reduces Anxiety, Depression, and Post-Traumatic Stress Disorder in Parents of Childhood or Adolescent Osteosarcoma Patients.

Specific, measurable, achievable, relevant, timed (SMART) principle improves the nursing utility by setting individual goals for participants and helping them to achieve these goals. Our study intended to investigate the impact of a SMART nursing project on reducing mental stress and post-traumatic stress disorder (PTSD) in parents of childhood or adolescent osteosarcoma patients. In this randomized, controlled study, 66 childhood or adolescent osteosarcoma patients and 126 corresponding parents were enrolled and divided into SMART or normal care (NC) groups at a 1:1 ratio. All parents received a 3-month corresponding intervention and a 6-month interview. Our study revealed that the self-rating anxiety scale score at the 3rd month (M3) (P < 0.05) and the 6th month (M6) (P < 0.01), and anxiety rate at M3 (P < 0.05) and M6 (P < 0.05) were lower in parents in SMART group vs. NC group. The self-rating depression scale score at M3 and M6, and depression rate at M3 and M6 were lower in parents in SMART group vs. NC group (all P < 0.05). Impact of events scale-revised score at the 1st month (M1) (P < 0.05), M3 (P < 0.05), and M6 (P < 0.01) were lower in parents in SMART group vs. NC group. By subgroup analyses, the SMART nursing project showed better impacts on decreasing anxiety, depression, and PTSD in parents with an undergraduate education or above than in those with a high school education or less. Conclusively, SMART nursing project reduces anxiety, depression, and PTSD in parents of childhood or adolescent osteosarcoma patients, which is more effective in those with higher education.

OLIE, ITCC-082: a Phase II trial of lenvatinib plus ifosfamide and etoposide in relapsed/refractory osteosarcoma.

While survival rates for patients with relapsed/refractory osteosarcoma are low, kinase inhibitors have shown efficacy in its treatment. The multikinase inhibitor lenvatinib, plus ifosfamide and etoposide, showed antitumor activity in a Phase II study in patients with relapsed/refractory osteosarcoma. This Phase II randomized controlled trial (OLIE) will assess whether the combination of lenvatinib + ifosfamide + etoposide is superior to ifosfamide + etoposide alone in children, adolescents and young adults with relapsed/refractory osteosarcoma. The primary end point is progression-free survival; secondary and exploratory end points include, but are not limited to, overall survival, objective response rate, safety and tolerability, pharmacokinetic characterization of lenvatinib in the combination treatment, quality of life and quantification of baseline unresectable lesions that are converted to resectable.

Lay abstract Traditional treatment for osteosarcoma (bone cancer) includes drugs that cause cell damage, such as ifosfamide and etoposide. The study in this article looked at adding lenvatinib, a drug that works to block development of blood vessels, to traditional chemotherapy to see whether it worked better. 'OLIE' is a Phase II clinical study comparing lenvatinib + chemotherapy (ifosfamide + etoposide) to the chemotherapy alone (ifosfamide + etoposide). An earlier study indicated that this drug combination may be successful. The study includes children, adolescents and young adults with osteosarcoma that has come back after, or did not respond to, previous treatment. OLIE will look at the antitumor activity and the side effects of this drug combination. The OLIE study is currently enrolling patients worldwide. Clinical trial registration number: NCT04154189 (ClinicalTrials.gov).

Future Directions in the Treatment of Osteosarcoma.

Osteosarcoma is the most common primary bone sarcoma and is often diagnosed in the 2nd-3rd decades of life. Response to the aggressive and highly toxic neoadjuvant methotrexate-doxorubicin-cisplatin (MAP) chemotherapy schedule is strongly predictive of outcome. Outcomes for patients with osteosarcoma have not significantly changed for over thirty years. There is a need for more effective treatment for patients with high risk features but also reduced treatment-related toxicity for all patients. Predictive biomarkers are needed to help inform clinicians to de-escalate or add therapy, including immune therapies, and to contribute to future clinical trial designs. Here, we review a variety of approaches to improve outcomes and quality of life for patients with osteosarcoma with a focus on incorporating toxicity reduction, immune therapy and molecular analysis to provide the most effective and least toxic osteosarcoma therapy.

Curar al alumno, no solo su enfermedad. Descripción de un caso / Healing a student, not just his or her disease. A Case description

INTRODUCCIÓN: En los últimos años las aulas hospitalarias se han configurado como un elemento de gran importancia en la vida de los niños y adolescentes que se encuentran en tratamiento de diferentes enfermedades. Los pacientes de enfermedades oncológicas no son una excepción. OBJETIVO: Aunque las aulas hospitalarias en la actualidad son capaces de sincronizar su trabajo con los colegios de referencia de los pacientes, el propósito de este artículo es presentar y describir un estudio de caso que subraya el papel del aula hospitalaria en sí misma, como una herramienta educativa que es útil en la tarea de educar a sus alumnos de una forma global e inclusiva, sin soslayar los niveles de exigencia adecuados. MÉTODO: Presenta el caso de un adolescente de 16 años diagnosticado de un osteosarcoma localizado que requirió quimioterapia preoperatoria y postoperatoria, cirugía de alta complejidad y finalmente un tratamiento inmunomodulador por un tiempo prolongado. No tenía ningún tipo de actividad académica al haber terminado la etapa de aprendizaje obligatorio y no estaba matriculado en ningún colegio, y a través del Aula Hospitalaria de la Unidad de Hematología y Oncología Pediátrica del Hospital HM Montepríncipe llegó a ser un estudiante interesado y motivado. A pesar de que su enfoque es necesariamente multidisciplinar, este estudio presenta una perspectiva educativa y pone de manifiesto la función, posibilidades, ventajas y desventajas de las aulas hospitalarias y su coordinación con el resto del personal. Lo hace poniendo al paciente en medio de su enfoque, a través de una visión humanística que enfatiza el verdadero significado de la educación. CONCLUSIONES: El propósito de un aula hospitalaria no debe ser solo académico, pues tiene un papel fundamental en el objetivo de que el niño y adolescente con cáncer se convierta en un adulto sano física, psíquica, social y espiritualmente

INTRODUCTION: The importance of hospital schools has been rising during the last decades. They have gained a lot of influence in the lives of children and teenagers under treatment for different diseases. Those in pediatric hematology/oncology units are not an exception. OBJECTIVE: Though the hospital schools are nowadays capable of synchronize their work with the patients' regular schools, the purpose of this project is to present and describe a case report which underlines the role of the hospital schools as themselves, as a teaching tool which is useful in the goal of educating its students in a global and comprehensive way, which includes an adequate level of demand. METHOD: It presents the case of a 16 years old boy diagnosed with a localized osteosarcoma that required preoperative and post-operative chemotherapy, high complexity surgery and, finally, immunomodulatory therapy for a long time. He had not any kind of learning activity, as he had finished the obligatory educational stage and was not enrolled in any school, and through the Hospital School in the Pediatric Hematology/Oncology Unit in the HM Montepríncipe Hospital became an interested and motivated student. Though its approach is necessarily cross-disciplinary, the present report has an educational perspective and it stresses the function, possibilities, advantages and disadvantages of hospital schools within pediatric oncology units and their coordination with the rest of the staff. It does it by putting the patient in the middle of its scope, through a humanist vision that emphasizes the real meaning of education. RESULTS: The purpose of hospital schools should not be just academic, since they have a fundamental role in achieving that children and teenagers with cancer become healthy adults in a physic, psychic, social and spiritual way

The Patient's Narrative Agenda as an assessment tool: the story of Robert, suffering from osteosarcoma.

BACKGROUND AND AIM OF THE WORK: To activate the participation of the person in his/her care path, the literature highlight the impact of the professional's ability to show a genuine interest in the problems brought by the patient and to recognize him/her as 'competent'. In these sense the narrative patient's agenda could be a useful relational tool, because is focused on the perception of patient experiences of his/her illness. Thus this study aims to analyze the usefulness of patient's narrative agenda during the assessment phase. METHOD: A semi-structured interview has been adopted to explore the agenda of Robert, 21 years old, suffering from osteosarcoma. A first level analysis identified the four functional areas of the agenda: ideas and beliefs; expectations and desires and context in which he lives and interacts. A second level analysis assessed the main Robert's problems. RESULTS: The narrative agenda has highlighted many central problems of Robert (e.g. therapeutic adherence, quality of life, mood, body image, existential problems related to experiences, hopes and expectations). Of course these results could be integrated with other tools: qualitative, to understanding difficulties and to formulate hypotheses, and quantitative, to measure the level of severity of problems reported. DISCUSSION AND CONCLUSION: The narrative agenda has not only proved to be a valid instrument of assessment, allowing an adequate insight on the patient's problems, as we exemplified, but it can be also used for monitoring the dynamic situation of the person's history, lending itself to the re-exploration of its functional areas over time.

Validation process of Toronto Exremity Salvage Score in Italian: A quality of life measure for patients with extremity bone and soft tissue tumors.

BACKGROUND AND OBJECTIVES: Limb salvage surgery remains the standard treatment in bone and soft tissue tumors. Toronto Extremity Salvage Score (TESS) is the most used quality of life measure. Our objective was to perform cross-cultural adaptation and validation in Italian, testing test-retest reliability, construct validity, and responsiveness. METHODS: We interviewed patients already treated for content validity. A total of 124 patients completed TESS and other questionnaires presurgery, at 3 months, 3 months + 2 weeks, and 6 months follow-up. We calculated intraclass correlation coefficients (ICCs) for reliability, associations with Pearson's r, and change over time with paired T tests. RESULTS: A new item regarding touch-screen devices was added to the upper extremity (UE) questionnaire. ICC resulted of 0.99 for lower extremity (LE) and 0.98 for UE patients, Pearson's r between TESS and Musculoskeletal Tumor Society was .66 and .64, EuroQol-5D-5L r was .62 and .61, and r between TESS and short form-36 physical function subscale was .76 and .71 for LE and UE groups, respectively. Paired T test results were statistically significant to detect change over time (0.03, 0.04, and 0.04 for LE groups and 0.03, 0.01, and 0.04 for UE groups). CONCLUSION: The Italian version of TESS can be used for the bone and soft tissue sarcoma population in clinical trials in Italy and with Italian speaking patients abroad to ensure patients' perspectives for efficacy and efficiency of treatments.

Effects of mindfulness-based stress reduction combined with music therapy on pain, anxiety, and sleep quality in patients with osteosarcoma

Objectives: To evaluate the effects of mindfulness-based stress reduction (MBSR) combined with music therapy (MT) on clinical symptoms in patients with osteosarcoma. Methods: Patients diagnosed with osteosarcoma were assessed for eligibility. A total of 101 patients were ultimately randomized into the intervention and control groups. Both groups received routine care. Eight sessions of MBSR and MT psychotherapy were conducted in the intervention group, while the control group received no psychological intervention. Patients were assessed regarding pain, anxiety, and sleep quality at two distinct stages: before and after the intervention. Results: There were no significant differences in sociodemographic and clinical parameters between the intervention and control groups at baseline. The intervention program significantly alleviated psychological and physiological complications in patients with osteosarcoma. Specifically, the study revealed that 8 weeks of the combined MBSR/MT intervention effectively reduced pain and anxiety scores and improved the quality of sleep in patients. Conclusion: MBSR combined with MT significantly alleviated clinical symptoms, and could be considered a new, effective psychotherapeutic intervention for patients with osteosarcoma.

Identifying bone sarcoma survivors facing psychosocial challenges. A study of trajectories following treatment.

OBJECTIVE: Bone sarcoma survivors face a number of physical and psychosocial challenges in relation to the late effects they experience following treatment. The present study aimed to identify and explore the different trajectories that bone sarcoma survivors might navigate during follow-up. METHODS: In-depth and semi-structured interviews were conducted, and an inductive thematic analysis was performed. RESULTS: When they were interviewed three to ten years after the primary diagnosis, the eighteen bone cancer survivors were found to be in three different rehabilitation phases that followed fairly distinct trajectories, namely, back to normal, a new normal and still struggling. Only three participants felt that they had returned to a life that was quite similar to the one they had lived prior to having cancer. Fifteen participants considered their lives and their bodies to be significantly altered. CONCLUSION: Sarcoma survivors who undergo life-changing treatment and return to very different lives than they had before should be identified by healthcare professionals and guided through this demanding phase to better cope with their new living conditions. Information on and tailored guidance related to psychosocial challenges may be of particular importance. Active focus on reorientation, as well as possibilities for growth, seems to be important.

Psychosocial Distress in Follow-up Care - Results of a Tablet-based Routine Screening in 202 Patients With Sarcoma.

BACKGROUND: Patients with sarcoma are particularly vulnerable to psychosocial distress. The aim of this study was to collect preliminary data on the prevalence of psychosocial distress in such patients during follow-up care and identify risk factors associated with higher psycho-oncological stress levels. PATIENTS AND METHODS: The study retrospectively enrolled 202 patients with bone or soft-tissue sarcomas who underwent routine psychosocial distress screening during their follow-up care. All patients were screened using an electronic cancer-specific questionnaire. RESULTS: Females and patients who underwent radiotherapy were more distressed. Psychosocial distress levels were markedly higher in the early postoperative phase, but approximately one-third of patients showed high psychosocial distress levels even more than 2 years postoperatively. CONCLUSION: The results underscore the importance of routine psychosocial distress screenings in patients with sarcoma, which should be performed throughout the follow-up period.

Effects of mindfulness-based stress reduction combined with music therapy on pain, anxiety, and sleep quality in patients with osteosarcoma.

OBJECTIVES: To evaluate the effects of mindfulness-based stress reduction (MBSR) combined with music therapy (MT) on clinical symptoms in patients with osteosarcoma. METHODS: Patients diagnosed with osteosarcoma were assessed for eligibility. A total of 101 patients were ultimately randomized into the intervention and control groups. Both groups received routine care. Eight sessions of MBSR and MT psychotherapy were conducted in the intervention group, while the control group received no psychological intervention. Patients were assessed regarding pain, anxiety, and sleep quality at two distinct stages: before and after the intervention. RESULTS: There were no significant differences in sociodemographic and clinical parameters between the intervention and control groups at baseline. The intervention program significantly alleviated psychological and physiological complications in patients with osteosarcoma. Specifically, the study revealed that 8 weeks of the combined MBSR/MT intervention effectively reduced pain and anxiety scores and improved the quality of sleep in patients. CONCLUSION: MBSR combined with MT significantly alleviated clinical symptoms, and could be considered a new, effective psychotherapeutic intervention for patients with osteosarcoma.

Health-Related Quality of Life and Survival Outcomes of Pediatric Patients With Nonmetastatic Osteosarcoma Treated in Countries With Different Resources.

PURPOSE: Health-related quality of life (HRQOL) improves throughout treatment of patients with nonmetastatic osteosarcoma. We compared HRQOL for patients in the United States and Chile treated on an international trial (OS99) with polychemotherapy and surgery, and we assessed the relationships among HRQOL measures, event-free survival (EFS), and overall survival (OS). MATERIALS AND METHODS: Patients with newly diagnosed, localized osteosarcoma and their parents completed three HRQOL instruments (PedsQL v.4, PedsQL Cancer v.3, and Symptom Distress Scale [SDS]). Data were collected at four time points throughout therapy. Repeated measures models were used to investigate the effect of treatment site on instrument scores. The log-rank test examined the impact of treatment site on survival outcomes, and Cox proportional hazards regression models evaluated baseline HRQOL measures as predictors of EFS and OS. RESULTS: Of 71 eligible patients, 66 (93%) participated in the HRQOL studies in the United States (n = 44) and Chile (n = 22). The median age was 13.4 years (range, 5 to 23 years). Clinical characteristics were similar between treatment sites. US patients reported better scores for physical ( P = .030), emotional ( P = .027), and school functioning ( P < .001). Chilean patients reported poorer scores for worry ( P < .001) and nausea ( P = .007). Patient and parent nausea scores were similar between patients treated in the United States and Chile by the end of therapy. Differences in symptom distress were not observed between the countries. Neither HRQOL measures nor treatment site were associated with EFS or OS. CONCLUSION: Although significant differences in HRQOL were observed between countries, outcomes were similar, and HRQOL measures were not associated with prognosis.

Clinical results and quality of life after reconstruction following sacrectomy for primary bone malignancy.

BACKGROUND: Sacrectomy is a rare and demanding surgical procedure that results in major soft tissue defects and spinopelvic discontinuity. No consensus is available on the optimal reconstruction algorithm. Therefore, the present study evaluated the results of sacrectomy reconstruction and its impact on patients' quality of life (QOL). METHODS: A retrospective chart review was conducted for 21 patients who underwent sacrectomy for a primary bone tumour. Patients were divided into groups based on the timing of reconstruction as follows: no reconstruction, immediate reconstruction or delayed reconstruction. QOL was measured using the EQ-5D instrument before and after surgery in patients treated in the intensive care unit. RESULTS: The mean patient age was 57 (range 22-81) years. The most common reconstruction was gluteal muscle flap (n = 9) and gluteal fasciocutaneous flap (n = 4). Four patients required free-tissue transfer, three latissimus dorsi flaps and one vascular fibula bone transfer. No free flap losses were noted. The need for unplanned re-operations did not differ between groups (p = 0.397), and no significant differences were found for pre- and post-operative QOL or any of its dimensions. DISCUSSION: Free flap surgery is reliable for reconstructing the largest sacrectomy defects. Even in the most complex cases, surgery can be safely staged, and final reconstruction can be carried out within 1 week of resection surgery without increasing peri­operative complications. Sacrectomy does not have an immoderate effect on the measured QOL.

The Effect of Clown Intervention on Self-Report and Biomarker Measures of Stress and Fatigue in Pediatric Osteosarcoma Inpatients: A Pilot Study.

BACKGROUND: Pediatric cancer patients experience different psychological processes during hospitalization that may regulate the immune response and affect recovery and response to cancer treatment. In this study, we aimed to examine the feasibility of longitudinal testing of psychophysiological parameters of stress and fatigue in pediatric osteosarcoma patients hospitalized for chemotherapy submitted to clown intervention; and to investigate whether changes in the levels of biomarkers are associated with psychological stress and fatigue levels in these patients after the clown intervention. METHODS: A pretest-posttest quasi-experimental pilot study was conducted at the pediatric oncology inpatient unit in a comprehensive cancer care center in Brazil including children and adolescents with osteosarcoma hospitalized for chemotherapy. Eight saliva samples were collected, comprising 4 at baseline (pre-intervention) and 4 after the clown intervention (+1, +4, +9, and +13 hours post-awakening). Salivary cortisol, α-amylase (sAA), cytokines, and matrix metalloproteinase-9 (MMP-9) levels were determined using high-sensitivity enzyme-linked immunosorbent assay (ELISA) kits. Stress and fatigue were measured by Child Stress Scale-ESI and PedsQL Multidimensional Fatigue Scale respectively. Bivariate association analysis between stress and fatigue scores and biomarker levels were investigated using nonparametric statistics. Effect sizes were calculated for each outcome variable. RESULTS: Six pediatric osteosarcoma patients were enrolled with no missing data. No significant effects sizes were observed for psychophysiological outcomes. Effect sizes ranged from 0.54 (cortisol) to 0 (interleukin-1ß [IL-1ß]). Decreasing overall trends were observed for cortisol levels for all 6 pediatric osteosarcoma patients over time. In addition, a similar pattern of tumor necrosis factor-α (TNF-α) levels over time was found for all 6 patients. Patients with metastatic osteosarcoma showed a linear trend for a decrease in MMP-9 levels between 1 and 9 hours after the clown intervention and restoration to basal levels after 13 hours. CONCLUSIONS: The results of this pilot study suggest that it is feasible longitudinally measure psychophysiological outcomes in the pediatric osteosarcoma inpatients for chemotherapy. Clown intervention merits further study as a way to reduce stress as well as fatigue, since that the stress and cytokines measurements are feasible based on our work.

Function and quality of life among primary osteosarcoma survivors in Iran: amputation versus limb salvage.

PURPOSE: This study aimed to evaluate the psychometric properties of the European Organization for Research and Treatment of Cancer Quality of Life Questionnaire (EORTC QLQ-C30) in Iranian osteosarcoma patients and apply this to compare the functional level, quality of life, symptoms and financial burden of the patients who underwent amputation and limb-salvage operations. METHODS: This study was conducted at the main referral orthopedic centers in the southwest of Iran from 2006 to 2016. After complete review of medical records, 48 patients were invited to attend the outpatient clinic and participate in the study via initial telephone interview. All data were entered in the Statistical Package for the Social Sciences version 15.0, and p values <0.05 were considered statistically significant. RESULTS: In total, 48 patients with extremities osteosarcoma completed the study. Of these, 31 had been treated with limb-salvage operation and 17 had undergone amputation. In functioning subscale, all the mean score of items, except social function, were higher in the limb salvage group than the amputee group. The mean scores (SD) of global health and quality of life were 64.5(13.2) and 61.2± 12.4 in the limb salvage and amputee groups, respectively. In the financial impact subscale, the mean score (SD) in the limb salvage group was 68.8± (29.7) compared to 74.5(25.0) in the amputee group. CONCLUSION: Results support the responsiveness of the EORTC QLQ-C30 for Iranian osteosarcoma patients. Applying this questionnaire revealed similar functional outcome, quality of life, symptoms and financial burden between amputation and limb-salvage groups.

Quality of life after bone sarcoma surgery around the knee: A long-term follow-up study.

It remains unclear if quality of life (QoL) improvements could be expected in young patients after malignant bone tumour surgery after 2 years. To assess the course of QoL over time during a long-term follow-up, malignant bone tumour survivors of a previous short-term study were included. Assessments were done at least 5 years after surgery. QoL was measured with Short-form (SF)-36, TNO-AZL Questionnaire for Adult's Quality of Life (TAAQOL) and Bone tumour (Bt)-DUX. QoL throughout the follow-up was analysed by linear mixed model analysis. From the original cohort of 44 patients; 20 patients were included for this study, 10 males; mean age at surgery 15.1 years and mean follow-up 7.2 years. Twenty-one patients of the initial cohort (47%) deceased. Fifteen patients (75%) underwent limb-salvage and five (25%) ablative surgery. QoL improved significantly during follow-up at Physical Component Summary Scale scale of the SF-36 and TAAQOL and all subscales of the Bt-DUX (p < .01). No significant differences were found between current evaluations and previous evaluations at 2 years after surgery (p = .41-.98). Significant advantages after limb-salvage were seen at the PCS scale of the SF-36 (MD 13.7, p = .05) and the cosmetic scale of the Bt-DUX (MD 17.7, p = .04).

IT-based Psychosocial Distress Screening in Patients with Sarcoma and Parental Caregivers via Disease-specific Online Social Media Communities.

BACKGROUND: Psychosocial distress can be frequently observed in patients with sarcoma, depicting a relevant clinical problem. However, prospective data collection on psychosocial distress in patients with rare tumors is often time-consuming. In this context, social media such as Facebook can serve as a potential platform to expand research. The aim of this study was to assess the feasibility of psychosocial distress screening in patients with osteosarcoma and Ewing's sarcoma via social media. MATERIALS AND METHODS: For this study an online questionnaire including general information and self-assessment distress measurement tools for patients and parents was created. The link to the questionnaire was then posted on the main page of the two largest disease-specific Facebook communities on osteosarcoma and Ewing's sarcoma. RESULTS: Within 2 months, 28 patients and 58 parents of patients were enrolled. All patients with osteosarcoma and Ewing's sarcoma, as well as the majority of parental caregivers of such patients, showed relevant psychosocial distress levels. CONCLUSION: Crowdsourcing via disease-specific patient communities on Facebook is feasible and provides great potential for acquisition of medical data of rare diseases.

Dissociation between the relief of skeletal pain behaviors and skin hypersensitivity in a model of bone cancer pain.

Recent studies have suggested that in humans and animals with significant skeletal pain, changes in the mechanical hypersensitivity of the skin can be detected. However, whether measuring changes in skin hypersensitivity can be a reliable surrogate for measuring skeletal pain itself remains unclear. To explore this question, we generated skeletal pain by injecting and confining GFP-transfected NCTC 2472 osteosarcoma cells unilaterally to the femur of C3H male mice. Beginning at day 7 post-tumor injection, animals were administered vehicle, an antibody to the P2X3 receptor (anti-P2X3) or anti-NGF antibody. Pain and analgesic efficacy were then measured on days 21, 28, and 35 post-tumor injection using a battery of skeletal pain-related behaviors and von Frey assessment of mechanical hypersensitivity on the plantar surface of the hind paw. Animals with bone cancer pain treated with anti-P2X3 showed a reduction in skin hypersensitivity but no attenuation of skeletal pain behaviors, whereas animals with bone cancer pain treated with anti-NGF showed a reduction in both skin hypersensitivity and skeletal pain behaviors. These results suggest that although bone cancer can induce significant skeletal pain-related behaviors and hypersensitivity of the skin, relief of hypersensitivity of the skin is not always accompanied by attenuation of skeletal pain. Understanding the relationship between skeletal and skin pain may provide insight into how pain is processed and integrated and help define the preclinical measures of skeletal pain that are predictive end points for clinical trials.