Getting control during follow-up visits: the views and experiences of parents on tumor surveillance after their children have completed therapy for rhabdomyosarcoma or Ewing sarcoma.

PURPOSE: Patients treated for rhabdomyosarcoma (RMS) or Ewing sarcoma (ES) are subject to extensive follow-up after completion of therapy. The aim of this follow-up is to monitor treatment side effects and to detect relapse in an early phase to improve prognosis after relapse. Little is known about parental emotional experiences during this period. We assessed the views and experiences of parents of children treated for RMS or ES on the follow-up examinations after completion of therapy. METHODS: We conducted two focus group meetings and four semi-structured telephone interviews with parents of children treated for RMS or ES in Dutch pediatric oncology centers. Parents of children 0-5 years after end-of-therapy were invited via letters (response rate 31%) and via social media channels of "Dutch Childhood Association for Children and Parents" (VOKK). An inductive thematic approach was used to analyze the data. RESULTS: In total, 12 parents (fathers, n = 3; mothers, n = 9) of 12 patients treated for RMS (n = 6) or ES (n = 6) participated. Median age at diagnosis for their children was 7.9 years and median time after end-of-treatment was 37 months. Four major themes were identified: content of follow-up, distress and anxiety, search for reassurance and hope, and interaction with others. Parents of children treated for RMS or ES report experiencing significant distress after completion of treatment. They report that their distress was decreased by adequate communication about content, timing, and reasoning behind follow-up. CONCLUSION: Physicians should pay attention to the needs of individual parents to reduce distress in the period after completion of therapy.

Male gender, school attendance and sports participation are positively associated with health-related quality of life in children and adolescents with congenital bleeding disorders.

BACKGROUND: This study assesses health-related quality of life (HRQOL), and variables associated with HRQOL, in children and adolescents with haemophilia and congenital bleeding disorders (CBD) in the Netherlands. METHODS: Patients <18 years with CBD under treatment at the Hemophilia Comprehensive Care Center of the Academic Medical Center were included. Participants completed generic HRQOL questionnaires (TAPQOL 0-5 years; PedsQL 6-18 years). Differences and effect sizes in HRQOL compared to healthy peers, and between hemophilia severity groups, were tested using Mann Whitney U-tests. Multivariate regression analyses were performed to assess variables associated with HRQOL. RESULTS: Data of 145 patients (81%) were analyzed (N = 32 with severe haemophilia). Children (0-12 years) show no significant impairments in HRQOL compared to healthy peers. Adolescent boys (13-18 years) with CBD report a slightly higher HRQOL on the total and emotional functioning scales than healthy peers (small-moderate effect sizes). In contrast, adolescent girls experience lower HRQOL on total, social functioning and psychosocial health scales compared to healthy peers (moderate effect sizes). No differences between severity groups were found in HRQOL, but more problem behaviour was found in young boys (0-5 years) with severe haemophilia. Male gender, participation in sports and school attendance are positively associated with HRQOL. Parental country of birth, type of treatment and number of bleeds are not associated with HRQOL. CONCLUSION: Continuing monitoring HRQOL in daily clinical practice for children with CBD is important, since possible influencing psychosocial factors can change over time, with special focus on adolescent girls, sports participation and school absence.

Health-related quality of life, developmental milestones, and self-esteem in young adults with bleeding disorders.

BACKGROUND: The treatment of bleeding disorders improved in the last decades. However, the effect of growing up with bleeding disorders on developmental, emotional, and social aspects is understudied. Therefore, this study assesses HRQOL, developmental milestones, and self-esteem in Dutch young adults (YA) with bleeding disorders compared to peers. METHODS: Ninety-five YA (18-30 years) with bleeding disorders (78 men; mean 24.7 years, SD 3.5) and 17 women (mean 25.1 years, SD 3.8) participated and completed the Pediatric Quality of Life Inventory Young Adult version, the Course of Life Questionnaire, and the Rosenberg Self-Esteem Scale. Differences between patients with bleeding disorders and their peers, and between hemophilia severity groups, were tested using Mann-Whitney U tests. RESULTS: YA men with bleeding disorders report a slightly lower HRQOL on the total scale, physical functioning, and school/work functioning in comparison to healthy peers (small effect sizes). YA men with severe hemophilia report more problems on the physical functioning scale than non-severe hemophilia. YA men with bleeding disorders achieved more psychosexual developmental milestones than peers, but show a delay in 'paid jobs, during middle and/or high school.' A somewhat lower self-esteem was found in YA men with bleeding disorders in comparison to peers (small effect size). For YA women with bleeding disorders, no differences were found on any of the outcomes in comparison to peers. CONCLUSION: This study demonstrates some impairments in HRQOL and self-esteem in YA men with bleeding disorders. By monitoring HRQOL, problems can be identified early, especially with regard to their physical and professional/school functioning.

Health-related quality of life questionnaires in individuals with haemophilia: a systematic review of their measurement properties.

BACKGROUND: The evaluation of health related quality of life (HRQOL) is essential for a full assessment of the influence of an illness on patients' lives. The aim of this paper is to critically appraise and compare the measurement properties of HRQOL questionnaires studied in haemophilia. METHODS: Bibliographic databases (Embase, Medline, Cinahl and PsycInfo) were searched for articles evaluating measurement properties of HRQOL questionnaires in haemophilia. Articles were excluded that did not report HRQOL measurement properties, or when <50% of the study population had haemophilia. The methodological quality of the selected studies was evaluated using the COSMIN checklist. The measurement properties of the HRQL questionnaires were rated as 'positive', 'indeterminate' or 'negative', accompanied by levels of evidence. RESULTS: The search resulted in 1597 unique hits, of which 22 studies were included. These articles evaluated three questionnaires for children (CHO-KLAT, Haemo-QoL and one unnamed measure) and five for adults (Hemofilia-QoL, Haemophilia Well-Being Index, HAEMO-QoL-A, Haem-A-QoL, and SF-36). The CHO-KLAT was the paediatric measure that showed the strongest measurement properties in high-quality studies. The Haemophilia Well-Being Index and HAEMO-QoL-A performed best among the adult measures. None of the studies reported measurement error and responsiveness. CONCLUSION: Our findings suggest that there is no need for new disease-specific HRQOL questionnaires for haemophilia, but rather that additional research is necessary to document the measurement properties of the currently available questionnaires, specifically focusing on the structural validity, measurement error and responsiveness of these questionnaires.

Psychosocial care for children with haemophilia and their parents in the Netherlands.

INTRODUCTION: Children growing up with haemophilia are at greater risk for psychosocial problems than their healthy peers. Providing psychosocial care to children with haemophilia and their families is indispensable, since psychosocial factors can have a significant impact on health and health-related quality of life (HRQOL). AIMS: Our aim was to give a description of psychosocial care provided by the multidisciplinary team of the Hemophilia Comprehensive Care Centre (HCCC) at the Emma Children's Hospital in Amsterdam, the Netherlands. With this overview, other caregivers and hospitals can benefit in organizing their psychosocial care for children with haemophilia. METHODS: The focus of the psychosocial care provided by the multidisciplinary team is on preventing psychosocial problems and medical-related stress, and supporting and equipping the child with haemophilia and its parents with as many skills as possible to lead an independent life with a high HRQOL. RESULTS: Core elements of the psychosocial care are therefore monitoring and screening of HRQOL (e.g. in daily clinical practice via www.hetklikt.nu), psychoeducation (haemophilia camp, haemophilia school, disease-specific activities, meetings for girls, parent meetings), practical help (Emma at Work, an employment agency for adolescents and young adults; Educational Facility and school visits), psychosocial interventions (the On Track group intervention and the Haemophilia Coping and Perception Test) and individual care (psychological counselling and referrals). CONCLUSION: By providing this overview of psychosocial support offered and by sharing this knowledge, psychosocial care can become more structured and consistent between HCCCs around the world. Potentially, processes and outcomes of care can be improved.

Anxiety and depression in mothers and fathers of a chronically ill child.

We aimed to determine the levels of anxiety and depression in mothers and fathers of a chronically ill child (0-18 years) and to study which parental and child variables are associated with anxiety and depression. In a cross-sectional design, anxiety and depression were assessed with the Hospital Anxiety and Depression Scale. Scores were compared to a Dutch reference group by analysis of variance and logistic regression analysis. Linear regression analyses were performed to examine which variables were associated with anxiety and depression. Mothers of a chronically ill child (n = 566) scored significantly higher than the reference group (p < .001) on anxiety (Mean 5.9 vs 4.8) and depression (Mean 4.5 vs 3.1). Fathers (n = 123) had higher depression scores (Mean 4.5 vs 3.6; p < .05), but fathers' anxiety scores were comparable to the reference group. The percentages of mothers in the clinical range of anxiety (31.8 vs 20.7 %, OR 2.03, 95 % CI 1.46-2.83) and depression (23.0 vs 12.0 %, OR 2.76, 95 % CI 1.84-4.13) were higher (p < .001) than in the reference group. No differences were found for fathers in the clinical range for anxiety and depression. Practical problems in daily life (a: ß = .33, d: ß = .25) and parenting stress (a: ß = .30, d: ß = .32) showed the strongest association with anxiety and depression for parents as a group. Illness-related characteristics of the child were not related. Parents of a chronically ill child, especially mothers, reported high levels of anxiety and depression. Awareness about parental anxiety and depression in pediatrics is important as well as targeted interventions.