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An abstract of the conference paper "The effectiviness of an innovative web-based application to monitor HRQOL problems in paediatric rheumatology clinical practice," by L. Haverman and colleagues is presented.

Gli Autori studiano con questionari il superamento delle tappe dello sviluppo infantile e adolescenziale in giovani pazienti adulti che hanno sofferto di dermatite atopica (DA) da bambini e dimostrano le conseguenze negative della malattia, soprattutto della DA grave. [ABSTRACT FROM AUTHOR]

Purpose: Germline DNA sequencing is increasingly used within pediatric oncology, yet parental experiences remain underexplored., Methods: Parents of children undergoing cancer predisposition gene panel sequencing (143 genes) were surveyed before and after disclosure of results. Questionnaires assessed knowledge, expectations, worries, satisfaction, and regret. Next to descriptives, linear mixed models and generalized mixed models were utilized to explore factors associated with knowledge and worries., Results: Out of 325 eligible families, 310 parents (176 mothers and 128 fathers of 188 families) completed all after-consent questionnaires, whereas 260 parents (150 mothers and 110 fathers of 181 families) completed all after disclosure questionnaires. Most parents hoped their participation would benefit others, although individual hopes were also common. Sequencing-related worries were common, particularly concerning whether their child would get cancer again, cancer risks for family members and psychosocial implications of testing. Parental satisfaction after disclosure was high and regret scores were low. Lower education was associated with lower knowledge levels, whereas foreign-born parents were at increased risk of experiencing worries., Conclusion: Germline sequencing of children with cancer is generally well received by their parents. However, careful genetic counseling is essential to ensure that parents are adequately informed and supported throughout the process., Competing Interests: Conflict of Interest All authors declare no conflicts of interest., (Copyright © 2024 The Authors. Published by Elsevier Inc. All rights reserved.)

Background: The purpose of this study was to determine factors associated with chronic fatigue (CF) in childhood cancer survivors (CCS)., Patients and Methods: Participants were included from the Dutch Childhood Cancer Survivor Study (DCCSS) LATER cohort, a nationwide cohort of CCS (≥5 years after diagnosis) and siblings as controls. Fatigue severity was assessed with the 'fatigue severity subscale' of the Checklist Individual Strength ('CIS-fatigue'). CF was defined as scoring ≥35 on the 'CIS-fatigue' and having fatigue symptoms for ≥6 months. Twenty-four parameters were assessed, categorized into assumed fatigue triggering, maintaining and moderating factors. Multivariable logistic regression analyses were carried out to investigate the association of these factors with CF., Results: A total of 1927 CCS participated in the study (40.7% of invited cohort), of whom 23.6% reported CF (compared with 15.6% in sibling controls, P < 0.001). The following factors were associated with CF: obesity [versus healthy weight, odds ratio (OR) 1.93; 95% confidence interval (CI) 1.30-2.87], moderate physical inactivity (versus physical active, OR 2.36; 95% CI 1.67-3.34), poor sleep (yes versus no, OR 2.03; 95% CI 1.54-2.68), (sub)clinical anxiety (yes versus no, OR 1.55; 95% CI 1.10-2.19), (sub)clinical depression (yes versus no, OR 2.07; 95% CI 1.20-3.59), pain (continuous, OR 1.49; 95% CI 1.33-1.66), self-esteem (continuous, OR 0.95; 95% CI 0.92-0.98), helplessness (continuous, OR 1.13; 95% CI 1.08-1.19), social functioning (continuous, OR 0.98; 95% CI 0.97-0.99) and female sex (versus male sex, OR 1.79; 95% CI 1.36-2.37)., Conclusion: CF is a prevalent symptom in CCS that is associated with several assumed maintaining factors, with lifestyle and psychosocial factors being the most prominent. These are modifiable factors and may therefore be beneficial to prevent or reduce CF in CCS., (Copyright © 2023 The Author(s). Published by Elsevier Ltd.. All rights reserved.)

Aim: To investigate the health-related quality of life (HRQOL) of Dutch adult childhood cancer survivors (CCS) and to identify risk factors of impaired HRQOL., Methods: Adult CCS (age >18, diagnosed <18, ≥5 years since diagnosis) from the Dutch LATER registry completed the Medical Outcome Study Short Form 36 (SF-36) to measure HRQOL and provided sociodemographic characteristics. Age-adjusted mean SF-36 scale scores of CCS were compared to the Dutch general population for men and women separately using t-tests, with effect size d. Multivariate logistic regression models were built to identify sociodemographic and cancer-related risk factors for impaired physical and mental HRQOL., Results: Both male and female CCS (N = 2301, mean age = 35.4 years, 49.6% female) reported significantly (p ≤ .005) worse HRQOL than the general population on almost all scales of the SF-36 (-.11 ≤ d ≤ -.56). Largest differences were found on vitality and general health perceptions. Significant risk factors (p ≤ .05) for impaired physical HRQOL were female sex, older age at diagnosis, not having a partner, low educational attainment, disease recurrence and exposure to radiotherapy, specifically to lower extremity radiation. Odds ratios (ORs) ranged from 1.6 to 3.7. Significant risk factors for impaired mental HRQOL were age 26-35 years, male sex, not having a partner and low educational attainment. ORs ranged from 1.3 to 2.0., Conclusion: Adult CCS had worse HRQOL than the general population. CCS most at risk were those with low educational attainment and without a partner. Adult CCS could benefit from routine surveillance of their HRQOL. Special attention for CCS' vitality and health perceptions and beliefs is warranted., Competing Interests: Conflict of interest statement The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this article., (Copyright © 2021 The Author(s). Published by Elsevier Ltd.. All rights reserved.)

Objective: The Psychosocial Assessment Tool (PAT) was developed to screen for psychosocial risk, aimed to be supportive in directing psychosocial care to families of a child with cancer. This study aimed to determine (i) the match between PAT risk score and provided psychosocial care with healthcare professionals blind to outcome of PAT assessment, and (ii) the match between PAT risk score and team risk estimation., Methods: Eighty-three families of children with cancer from four pediatric oncology centers in the Netherlands participated (59% response rate). The PAT and team risk estimation was assessed at diagnosis (M = 40.2 days, SD = 14.1 days), and the content of provided psychosocial care in the 5-month period thereafter resulting in basic or specialized care., Results: According to the PAT, 65% of families were defined as having low (universal), 30% medium (targeted), and 5% high (clinical) risk for developing psychosocial problems. Thirty percent of patients from universal group got basic psychosocial care, 63% got specialized care, and 7% did not get any care. Fourteen percent of the families at risk got basic care, 86% got specialized care. Team risk estimations and PAT risk scores matched with 58% of the families., Conclusions: This study showed that families at risk, based on standardized risk assessment with the PAT, received more specialized care than families without risk. However, still 14% of the families with high risks only received basic care, and 63% of the families with standard risk got specialized care. Standardized risk assessment can be used as part of comprehensive care delivery, complementing the team., (© 2017 Wiley Periodicals, Inc.)

Purpose: Intensive therapies in pediatric malignancies increased survival rates but also occurrence of treatment-related morbidities. Therefore, supportive care fulfills an increasingly important role. In planning development of guidelines with incorporation of shared decision making, we noticed that little is known about the needs and preferences of patients and their parents. Our goals were therefore to investigate (1) which supportive care topics patients and parents regard as most important and (2) the preferred role they wish to fulfill in decision making., Methods: This qualitative study consisted of three focus groups (two traditional, one online) with patients and parents of two Dutch pediatric oncology centers. Data were transcribed as simple verbatim and analyzed using thematic analysis., Results: Eleven adolescent patients and 18 parents shared detailed views on various aspects of supportive care. Themes of major importance were communication between patient and physician (commitment, accessibility, proactive attitude of physicians), well-timed provision of information, and the suitability and accessibility of psychosocial care. In contrast to prioritized supportive care topics by medical professionals, somatic issues (e.g., febrile neutropenia) were infrequently addressed. Patients and parents preferred to be actively involved in decision making in selected topics, such as choice of analgesics and anti-emetics, but not in, e.g., choice of antibiotics., Conclusions: Children with cancer and parents were provided a valuable insight into their views regarding supportive care and shared decision making. These results have important implications towards improving supportive care, both in selecting topics for guideline development and incorporating preferences of patients and parents herein.

Children with acute lymphoblastic leukemia (ALL) undergo intense anticancer treatment. We systematically reviewed 22 studies evaluating 2,073 ALL patients' health-related quality of life (HRQL) and its clinical/demographic correlates during treatment. Overall HRQL was significantly reduced on treatment. Despite HRQL improvements over time, longitudinal studies reported a proportion of children continued to experience reduced HRQL after treatment completion. We found inconsistent associations between clinical/demographic factors and HRQL outcomes. Tentative evidence emerged for worse HRQL being associated with intensive phases of chemotherapy, corticosteroid therapy, experiencing greater toxicity, older age, and female sex. Longitudinal studies are needed to identify children at-risk of reduced HRQL., (© 2017 Wiley Periodicals, Inc.)

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., (© 2017 John Wiley & Sons Ltd.)

Objective: To investigate psychosocial characteristics of children and parents as predictors and moderators of the effect of a group intervention for children with chronic illness and their parents., Methods: Data from a randomized controlled trial were used, including 194 children (8-18 years) who were assigned to a child-only intervention, a parent-child intervention, or a wait-list control group. Longitudinal multilevel regression analyses were used to test effects on change in parent and child reported internalizing and externalizing behavior problems., Results: For children with a more disengaged coping style or lower self-worth and for children who experienced a more secure parent-child relationship, the parent-child intervention was more effective than the child-only intervention in reducing behavior problems., Conclusions: Children who are more "at risk" appear to gain more from participating in an intervention, especially if their parents are involved as well. However, the benefit of parents' involvement may depend on the quality of the parent-child relationship., (© The Author 2014. Published by Oxford University Press on behalf of the Society of Pediatric Psychology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.)

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: β = .33, d: β = .25) and parenting stress (a: β = .30, d: β = .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.

Several studies in adults have shown patient reported outcomes (PROs) to be effective in enhancing patient-physician communication and discussion of Health Related Quality of Life outcomes. Although less studied, positive results have been demonstrated in children. A PRO-intervention needs to be feasible in clinical practice to be successful. In the current study, 74 parents of children who successfully completed their cancer treatment and 21 pediatric oncologists (POs) evaluated a PRO-intervention and gave recommendations for future use in their practice. Most parents and POs suggested PROs to be an important part of standard care, starting during treatment, with an assessment frequency of every 3 months., (© 2014 Wiley Periodicals, Inc.)

Background: The aim of this study was to investigate whether an unfavourable psychosocial developmental trajectory while growing up with childhood cancer is related to a smaller likelihood of labour participation in adult life., Methods: A total of 53 childhood cancer survivors (CCS) with and 313 CCS without disability benefits, and 508 peers from the general Dutch population (reference group) completed the Course of Life Questionnaire (CoLQ) about the achievement of psychosocial developmental milestones. Differences between the three groups were tested by conducting analysis of variance with contrasts (scale scores CoLQ) and logistic regression analysis (individual milestones). Effect sizes and odds ratios were calculated., Results: Compared with the reference group, both CCS with and CCS without benefits reported lower scale scores with respect to social and psychosexual development. CCS with disability benefits had lower social (d = - 0.6; p < 0.001) and psychosexual (d = -0.4; p < 0.01) scale scores than the CCS without disability benefits. CCS with disability benefits scored less favourably (p < 0.01) than peers from the general population on 14 out of 22 psychosocial milestones whereas the number was only six for those without disability benefits., Conclusions: CCS with an unfavourable developmental trajectory while growing up were more likely to apply for disability benefits in adulthood than CCS with a more favourable development. Early recognition and support are warranted. Further research is needed on risk factors of application for disability benefits. In addition, research should show whether stimulating the achievement of developmental milestones while growing up will create conditions for a better labour market position., (Copyright © 2011 John Wiley & Sons, Ltd.)

Background and Aims: Inflammatory Bowel Disease (IBD) patients with anxiety and/or depressive symptoms may not receive the care they need. Provision of care requires insight into the factors affecting these psychiatric symptoms. The study was designed to examine the extent to which: (1) IBD patients with anxiety and/or depressive symptoms receive mental treatment and (2) clinical and socio-demographic variables are associated with these symptoms., Methods: 231 adult IBD patients (79% response rate), attending a tertiary care center, completed standardized measures on anxiety and depressive symptoms (HADS), quality of life (SF-12) and mental health care use (TIC-P). Diagnosis and disease activity were determined by the gastroenterologist., Results: 43% had high levels of anxiety and/or depressive symptoms, indicative of a psychiatric disorder (HADS ≥ 8), of whom 18% received psychological treatment and 21% used psychotropic medication. In multivariate analysis, high disease activity was associated with anxiety (OR=2.72 | p<0.03) and depression (OR=3.36 | p<0.01), while Crohn's disease was associated with anxiety (OR=2.60 | p<0.03)., Conclusions: Despite high levels of anxiety and depressive symptoms and poor quality of life, psychiatric complaints in IBD patients were undertreated. Screening for and treatment of psychiatric symptoms should become an integral part of IBD medical care., (Copyright © 2011 European Crohn's and Colitis Organisation. Published by Elsevier B.V. All rights reserved.)

Background: Previous studies have assessed health-related quality of life (HRQOL) during several treatment stages in children with cancer, but there is limited knowledge about HRQOL shortly after completing therapy. This study determined HRQOL of children with cancer shortly after the end of successful treatment compared with normative values., Procedure: Several age-specific HRQOL questionnaires were administered: the ITQOL (generic, proxy-report, 0-4 years), CHQ PF 50 (generic, proxy-report, 5-7 years), Kidscreen (generic, self-report, 8-18 years) and Disabkids (chronic generic, self-report, 8-18 years)., Results: Children with cancer (N = 191, mean age 9.25, SD 5.06, 47.1% female) participated. Physical well-being was affected for all ages. Compared to normative values 0- to 7-year-olds were rated significantly lower on the majority of the scales. In addition, 12- to 18-year-olds had significantly better HRQOL than the norm on social scales. Compared to chronically ill norms, 8- to 18-year-olds demonstrated no differences, except for 12- to 18-year-olds who experienced significantly more physical limitations. Additionally, we found that HRQOL of parents of 0- to 7-year-olds was poorer than the norm., Conclusion: HRQOL in children with cancer and their parents can be impaired compared with the norm. Therefore, HRQOL should be monitored in clinical practice to make paediatric oncologists aware of these problems. For young children, we recommend checking whether certain HRQOL problems can be explained by parental worries. For older children and adolescents, paediatric oncologists need to consider social desirability and the child's adaptive style., (Copyright © 2010 Wiley-Liss, Inc.)

With the aim of assessing parental stress after SCT, 73 parents of children and adolescents who underwent SCT 5 or 10 years ago responded to questionnaires on general distress (General Health Questionnaire (GHQ)), disease-related stress (Pediatric Inventory for Parents-short form (PIP-SF)) and perceptions of child vulnerability (Child Vulnerability Scale (CVS)). General distress scores were comparable with the reference groups, but 40% of the mothers at 5 years after SCT reported increased stress levels as compared with 26% in the community-based reference group. Disease-related stress was comparable with the reference group of parents of children who were just off cancer treatment, 5 years after SCT. At 10 years after SCT, scores were lower than the reference group. Perceived child vulnerability did diminish over time, but remained high in parents of SCT survivors, compared with parents of healthy children: 96% of the parents at 5 years after SCT and 76% of the parents at 10 years after SCT scored above the cutoff point. Perceived vulnerability was found to be a predictor for parental disease-related stress. To conclude, although most parents of SCT survivors are resilient, the majority of parents perceive their child to be much more vulnerable as compared with parents of healthy children. This perception is associated with disease-related stress and may induce overprotective parenting.

Goals of Work: Pediatric stem cell transplantation (SCT) is a stressful treatment for children with relapsed or high-risk malignancies, immune deficiencies and certain blood diseases. Parents of children undergoing SCT can experience ongoing stress related to the SCT period. The aim of this article was to present a literature review of articles on parental distress and adaptation before, during, and after SCT and to identify risk and protective factors., Materials and Methods: The review was conducted systematically by using PubMed, Web of Science, PsychInfo, and Picarta databases. Eighteen articles met our inclusion criteria: publishing date between January 1, 1990 and January 1, 2009; studies concerning parents of children undergoing SCT; studies examining the psychological adjustment and/or stress reactions of parents as primary outcomes and studies available in English., Main Results: Highest levels of parental stress are reported in the period preceding SCT and during the acute phase. Stress levels decrease steadily after discharge in most parents. However, in a subgroup of parents, stress levels still remain elevated post-SCT. Parents most at risk in the longer term display highest levels of stress during the acute phase of the SCT., Conclusions: Psychosocial assessment before SCT, during the acute phase and in the longer term, is necessary to identify parents in need for support and follow-up care.

Although the need for insight in factors influencing the quality of life of patients with an inborn error of metabolism is recognized, psychological adjustment of adults with metabolic diseases has not been properly studied. Adult patients with PKU were demonstrated not to differ from healthy controls in terms of their course of life (CoL) and health-related quality of life (HRQoL). However, adults with galactosaemia had a lower HRQoL with significant lower scores on the domains of cognitive and social function. This study investigated the CoL and the social demographical outcomes in these young adults with galactosaemia, and compared them with the general population and with PKU patients. A total of 15 (88%) adult patients with classical galactosaemia participated in this study. Classical galactosaemia patients had a delayed social and psychosexual development compared to their peers from the general population and to PKU patients. Also, they were significantly less frequently married or living together and significantly less frequently employed than the general population. Our study shows a stark contrast between patients with galactosaemia and patients with PKU, although both are diagnosed in the neonatal period and need life-long dietary restrictions. The observed difference is likely due to the long-term somatic complications frequently seen in galactosaemia and thus not due to the burden of a chronic disease necessitating life-long dietary restrictions. We conclude that it is essential that parents and clinicians encourage children with galactosaemia to participate in peer-related activities in order to stimulate social performance, which may result in a more normal CoL.

Phenylketonuria (PKU; OMIM 261600) is an autosomal recessive disorder of phenylalanine metabolism caused by a deficiency of the enzyme phenylalanine hydroxylase (PAH; EC 1.14.16.1). Cognitive problems, neuropsychological abnormalities and psychosocial problems have been reported frequently in children and adolescents with PKU, even in those who are treated early and continuously. However, the developmental consequences in adulthood of growing up with PKU are not well known. The aim of this study was to assess the course of life, sociodemographic outcomes and health-related quality of life in young adult patients with PKU identified on neonatal screening who were continuously on treatment. A total of 32 PKU patients 18 to 30 years old completed the Course of Life questionnaire, the RAND-36 Health Survey, and the cognitive scale of the TNO-AZL Adult Quality of Life (TAAQoL) questionnaire. The results of the Course of Life and Health-Related Quality of Life questionnaires were comparable to controls, except that a higher percentage received special education in primary school. Their educational attainment, however, was comparable to that of their peers. The results of this study demonstrate that although PKU is a chronic disease with the burden of strict dietary control, early and continuously treated patients with PKU can have a normal health-related quality of life and course of life.

In paediatric research, Health-Related Quality-of-Life (HRQoL) has received increasing recognition as an important health outcome. This study aimed to investigate the nature and prevalence of HRQoL problems in children with different chronic diseases. Data were available on 318 children aged 8-11 years with different diseases: congenital heart disease (n = 50); coeliac disease (n = 105); asthma (n = 32); cancer (n = 23); juvenile chronic arthritis (n = 45); children with capillary haemangioma (n = 25) and severe meningococcal disease (n = 38). They all answered a validated generic instrument [TNO-AZL Children's Quality of life questionnaire] (TACQoL), in the outpatient clinic or at home. Analyses of variance were performed to investigate differences in mean scores for children with chronic conditions in comparison to healthy children. Prevalence of children at risk for substantial HRQoL problems was based on the 25th percentile in the norm population. In comparison to healthy children, only a small number of differences were found in mean scores of children studied. In contrast, prevalence of HRQoL problems in children with chronic diseases was higher in several domains. It is concluded that using an indicator variable of the norm 25th percentile seems important in identifying at-risk children with chronic disease.

Some specific aspects of communication in pediatric oncology will be outlined in this chapter. These include openness about the disease, which has become increasingly important. Furthermore, the law of double protection, a self-protective strategy used by children, parents, and hospital staff, will be sketched out. It is very striking that protection is often achieved through protection of the other. Several examples of this strategy will be presented. Finally, attention will be paid to communication about death in the palliative phase.

This study assessed quality of life, self-esteem and worries in young adult survivors of childhood cancer compared to a group of young adults with no history of cancer. The impact of demographic, medical and treatment factors and self-esteem on survivors' quality of life and worries was studied. Participants were 400 long-term survivors (LTS) of childhood cancer (age range 16-49 years, 45% female) who had completed treatment an average of 16 years previously and 560 persons (age range 16-53 years, 55% female) with no history of cancer. All participants completed the MOS-24 (Medical Outcome Study Scale), a Worry questionnaire consisting of three scales (cancer-specific concerns, general health concerns, present and future concerns), and the Rosenberg Self-Esteem Scale. Small to moderate differences were found in mean MOS-24 scores between the LTS group and controls (range effect sizes -0.36-0.22). No significant difference was found in the mean self-esteem scores between LTS and controls. Female LTS had more cancer-specific concerns than male LTS. In several related areas of general health, self-image and dying, the LTS group reported less worries than controls, but LTS worried significantly more about their fertility, getting/changing a job and obtaining insurance's. Multiple linear regression analysis revealed that female gender, unemployment, severe late effects/health problems and a low self-esteem were predictors of worse quality of life in survivors. In addition, age at follow-up, unemployment, years since completion of therapy and a low self-esteem were associated with a higher degree of survivors' worries. Quality of life and the level of self-esteem in LTS of childhood cancer is not different from their peers. Although many LTS worried not more or even less about health issues than their peers, they often are concerned about some present and future concerns. The investigated factors could explain poor quality of life and worries only to a limited extent. Further research exploring determinants and indices of quality of life and worries in LTS is warranted.

Unlabelled: Several studies have been conducted on sibling psychosocial adaptation to cancer in a brother or sister, but little is known on how the long-term adaptation of siblings to the illness develops. The concept quality of life has primarily been applied in research on the effects of chronic illness on the affected patient, but has not yet been studied in siblings., Aims: To investigate the prevalence of self-reported psychosocial problems in siblings of pediatric cancer patients, 2 years after the onset of the illness., Measurements: Two Dutch quality of life questionnaires, the TACQOL and the DUCATQOL are used, as well as generic non-health-related measures of emotional and behaviour problems (CBCL-YSR) and anxiety (STAI-C)., Participants: Participants were 103 siblings aged 7-18 years old. Fifty seven Siblings participated in a prospective and 46 in a retrospective study group., Results: Siblings aged 7-11 report lower overall quality of life than children in the norm group. No differences in mean scores were found on any of the other domains that were investigated. When the prevalence of problems was investigated, however, relatively more siblings compared to normative data had scores in the impaired group based on the 20th percentile norm. A relatively high number of siblings aged 7-11 reported impaired emotional (42%), social (34%) and total quality of life (47%) (DUCATQOL) and physical problems (26%) (TACQOL). Relatively many adolescent siblings (26%) reported significant internalising problems on the CBCL-YSR., Conclusions: Although acute emotional distress reactions seem to have normalised in most siblings as has been suggested in the literature, emotional distress of having a brother or sister with cancer may continue beyond diagnosis for a subgroup of children. Young siblings seem to be affected in their quality of life, whereas a subgroup of adolescent siblings experience clinically relevant internalising problems. The results support the use of quality of life measures for siblings. Predictors of long-term adaptation in siblings need to be investigated.

Background: Previous research suggests that posttraumatic stress disorder (PTSD) is present in survivors of childhood cancer. The aim of the current study was to explore posttraumatic stress symptoms in a sample of young adult survivors of childhood cancer. In addition, the impact of demographic, medical and treatment factors on survivors' posttraumatic stress symptoms was studied., Procedure: Participants were 500 long-term survivors of childhood cancer. The median age at follow-up was 24 years (age range, 16- 49 years, 47% female). To assess symptoms of posttraumatic stress, all participants completed the Impact of Event Scale (IES), a self-report instrument consisting of two subscales, intrusion and avoidance., Results: Twelve percent of this sample of adult survivors of childhood cancer had scores in the severe range, indicating they are unable to cope with the impact of their disease and need professional help. Twenty percent of the female survivors had scores in the severe range as compared with 6% of the male survivors. Linear regression models revealed that being female, unemployed, a lower educational level, type of diagnosis and severe late effects/health problems were associated with posttraumatic stress symptoms., Conclusions: The results indicate that, although the proportion of survivors reporting symptoms is well within the proportions found in the general population, a substantial subset of survivors report symptoms of posttraumatic stress. This finding supports the outcomes reported previously that diagnosis and treatment for childhood cancer may have significant long-term effects, which are manifested in symptoms of posttraumatic stress. The investigated factors could explain posttraumatic stress symptoms only to a limited extent. Further research exploring symptoms of posttraumatic stress in childhood cancer survivors in more detail is clearly warranted. From a clinical perspective, health care providers must pay attention to these symptoms during evaluations in the follow-up clinic. Early identification and treatment of PTSD symptoms can enhance the quality of life for survivors of childhood cancer., (Copyright 2004 Wiley-Liss, Inc.)

The aim of this study was to prospectively investigate the prevalence of and risk factors for psychosocial problems in siblings of paediatric cancer patients. One and 6 months after diagnosis, sibling self-reported anxiety, social-emotional problems and quality of life (QoL) were assessed, as were the predictor variables: sibling prediagnosis functioning, age and gender and the ill child's diagnosis. At 1 month, siblings reported a lower QoL and adolescent girls reported more emotional problems compared with peers. At 6 months, adolescent QoL remained relatively impaired. Over time, adolescent brothers reported fewer emotional and total problems and young girls reported decreased anxiety. No significant amelioration in QoL was found over time. The older the siblings were, the lower their observed QoL at both measurements and in several domains. The occurrence of life events predicted sisters' QoL at 1 month. Changes in sibling functioning were predicted by none of the investigated risk factors. Thus, QoL is impaired shortly after diagnosis. Adolescent siblings risk persisting problems in daily functioning. Further prospective research on other risk factors such as coping and family functioning over time is needed.

This paper investigated educational achievement, employment status, living situation, marital status and offspring in 500 Dutch long-term young adults survivors of childhood cancer (age range, 16-49 years, 47% female). The results were compared with a reference group of 1092 persons with no history of cancer (age range, 15-33 years, 55% female). The impact of demographic and medical characteristics on psychosocial adjustment was studied. All participants completed a self-report questionnaire. The results showed that, although many survivors are functioning well and leading normal lives, a subgroup of survivors were less likely to complete high-school, to attain an advanced graduate degree, to follow normal elementary or secondary school and had to be enrolled more often on learning disabled programs. The percentage of employed survivors was lower than the percentage of employed controls in the comparison group, but more survivors were student or homemaker. Survivors had lower rates of marriage and parenthood, and worried more about their fertility and the risk of their children having cancer. Survivors, especially males, lived more often with their parents. Cranial irradiation dose

Clinical reports suggest that many survivors of childhood cancer experience fatigue as a long-term effect of their treatment. To investigate this issue further, we assessed the level of fatigue in young adult survivors of childhood cancer. We compared the results with a group of young adults with no history of cancer. The impact of demographic, medical and treatment factors and depressive symptoms on survivors' fatigue was studied. Participants were 416 long-term survivors of childhood cancer (age range 16-49 years, 48% of whom were female) who had completed treatment an average of 15 years previously and 1026 persons (age range 16-53 years, 55% female) with no history of cancer. All participants completed the Multidimensional Fatigue Inventory (MFI-20), a self-report instrument consisting of five scales (general fatigue, physical fatigue, mental fatigue, reduced activity, reduced motivation) and the Center for Epidemiologic Studies Depression Scale (CES-D). Small differences were found in the mean scores for the different dimensions of fatigue between the long-term survivors and controls (range effect sizes -0.34 to 0.34). Women experienced more fatigue than men. Logistic regression revealed that being female and unemployed were the only demographic characteristics explaining the various dimensions of fatigue. With regard to medical and treatment factors, diagnosis and severe late effects/health problems were associated with fatigue. Finally, depression was significantly associated with fatigue on all subscales. Our clinical practice suggests a difference in fatigue in young adult childhood cancer survivors and their peers. This could not be confirmed in this study using the MFI-20. The well known correlation between fatigue and depression was confirmed in our study. Further research is needed to clarify the undoubtedly complex somatic and psychological mechanisms responsible for the development, maintenance and treatment of fatigue in childhood cancer survivors.

In recent years the necessity of measuring quality of life in childhood cancer survivors has been stressed. This paper gives an overview of the results of studies into the quality of life (QL) of young adult survivors of childhood cancer and suggest areas for future research. The review located 30 empirical studies published up to 2001. The results are described in terms of the following QL dimensions: physical functioning (QL, general health), psychological functioning (overall emotional functioning, depression and anxiety, self-esteem), social functioning (education, employment, insurance, living situation, marital status and family), and sexual functioning. Factors related to survivors' QL are reported: demographics and illness- and treatment related variables. Although the literature yields some inconsistent findings, a number of clear trends can be identified: (a) most survivors reported being in good health, with the exception of some bone tumour survivors; (b) most survivors function well psychologically; (c) survivors of CNS tumours and survivors of acute lymphoblastic leukaemia (ALL) are at risk for educational deficits; (d) job discrimination, difficulties in obtaining work and problems in obtaining health and life insurance were reported; (e) survivors have lower rates of marriage and parenthood; (f) survivors worry about their reproductive capacity and/or about future health problems their children might experience as a result of their cancer history. There is a need for methodological studies that measure QL among survivors of childhood cancer more precisely by taking into account the effects of the severity of the cancer and the long-term impact of different treatments. Additional data are needed to help us understand the needs of survivors and to identify those subgroups of survivors who are at greatest risk for the adverse sequelae of the disease and its treatment.

Unlabelled: This study assessed the impact of inflammatory bowel disease (IBD) on the health-related quality of life (HRQoL) of children and adolescents, using both a generic and a disease-specific instrument. Three questionnaires were sent to all patients (8-18 y old) from the database of two large secondary/tertiary hospitals in the western part of The Netherlands. In total, 83 (66%) children responded, 18 were between 8 and 12 y old and the remaining 65 were older. HRQoL was measured using a generic instrument, the TNO-AZL Children's Quality of Life questionnaire (TACQOL), assessing seven domains, and the Impact-II (NL), a disease-specific instrument assessing six domains. Disease activity was measured by a five-item symptom card. Compared with scores from a large reference population (n= 1810), younger children with IBD had a comparable HRQoL (measured by the TACQOL) on six domains, and better cognitive functioning, although they did not have severely active disease. Adolescent patients with IBD had a significantly impaired HRQoL on four domains (body complaints, motor functioning, autonomy and negative emotions). The Impact-II discriminated well between patients with varying disease activity states on all domains., Conclusion: Adolescents with IBD have a severely affected HRQoL. Impairment on motor functioning and autonomy is a threat to gaining independence from caregivers, and a high occurrence of negative emotions places patients at risk for depressive and behavioural disorders. The Impact-II is recommended for clinical use because of its high discriminative validity.

An overview is given of the social and emotional adjustment in young survivors of childhood cancer. The results are described in terms of self-esteem, anxiety, depression and posttraumatic stress (emotional adjustment), and in terms of behavioral functioning, social competence and school performance (socio-behavioral adjustment). Furthermore, factors related to survivors' adjustment are reported: demographics, illness- and treatment-related factors, coping and social support, and family and parental functioning. Limitations of the studies and consequences for future research are discussed. On the whole, the adjustment of young cancer survivors as a group was reasonably good, but the findings with respect to the emotional and social adjustment were inconsistent. This might be attributed to limitations of the study designs and the fact that the studies were not all directly comparable. In order to gain more insight into the predictors of adjustment, longitudinal studies are recommended, which should include control groups or standardized instruments with norm data, and use cancer-specific measures in addition to generic measures.

Childhood cancer can have a substantial emotional impact on the siblings of the sick child. In order to help these siblings adjust to the illness, supportive groups were started in 1994 at our medical center. The program is based on a model of psychosocial support; the aim of the group is to enhance control strategies and, thus, to reduce anxiety. It consists of five sessions and is offered to siblings aged 7-18. The study objective is to evaluate the effect of group participation on sibling anxiety. The State Trait Anxiety Inventory for Children was administered to 24 siblings before and after group participation. Results showed that siblings experience less anxiety after participating in the group. Before group participation, a majority of the siblings were more anxious compared with normal peers. The sibling's age and sex, whether the ill child is in treatment, survival perspective, and time since diagnosis were not related to anxiety reduction. The continuation of the group is supported by its positive impact on siblings. Results must nevertheless be interpreted cautiously, considering the small number of siblings participating in the study, the lack of a control group and the restriction to one outcome measure., (Copyright 2001 John Wiley & Sons, Ltd.)

The main objective of the present study was to investigate whether children with cancer with different survival perspectives differ in their psychological adjustment, defensiveness and their use of cognitive control strategies. Furthermore, the study investigated which variables predict emotional adjustment of these children with cancer most adequately. A total of n=84 children (n=43 children in remission and n=41 children not in remission) participated in the study. They answered questionnaires about control strategies, defensiveness, anxiety and depression. No differences were found in any of the questionnaires between children in remission and relapse. Emotional adjustment of the children was predicted by defensiveness and by positive expectations about the course of the illness. The findings demonstrate the importance of having positive expectations for the emotional adjustment of children with cancer. The strength of the study is the inclusion of a substantial group of children with cancer who have a reduced survival perspective., (Copyright 2001 John Wiley & Sons, Ltd.)

Studies concerning sibling adjustment to childhood cancer and published since 1980 were reviewed. Sibling distress was described in terms of emotional, socio-behavioral, academic, physical and positive reactions. Secondly, characteristics and coping resources of siblings that are related to adjustment were summarized. Thirdly, the families' functioning, parental coping resources and family events that relate to adjustment were described. Finally, relevant aspects of the illness that may influence adjustment were described. Results of the 35 studies reviewed are summarized in a table containing general descriptors, methods and results. There are salient difficulties in comparison of the findings owing to variations in design, heterogeneity of the groups of siblings, diversity in sample size, and differences in conceptualizations of adjustment and coping. The lack of longitudinal studies, the precarious definitions and confounding of coping and adaptation, and the marginal role of siblings' coping efforts were identified as the most striking deficiencies in current research.

The use of alternative treatment along with conventional cancer therapy is very popular. However, little is known about the use of alternative treatment in pediatric oncology. A study to determine which medical and demographic characteristics distinguish users from nonusers was conducted in a pediatric oncology sample of children with different survival perspectives. The parents of 84 children with cancer (43 patients in first continuous remission and 41 patients who had suffered a relapse or second malignancy) participated in the study and were surveyed with respect to the use of alternative treatment. The survival perspective appeared to be the most important variable distinguishing users of alternative treatment from nonusers. Twenty-six families (31%) had used or were using alternative treatment, of which 19 were families of children with cancer who had suffered a relapse (46%), and 7 were families of children with cancer in remission (16%). The most common types of alternative treatment used were based on homeopathy and anthroposophy (treatments based on autonomous medical concepts). Because of the finding that lower survival perspective is related to the use of alternative treatment, the use of alternative treatment conceivably could be a coping strategy based on illusions. Therefore, it is recommended that alternative treatment be discussed open-mindedly by all health care providers involved in the care of pediatric oncology patients.

In the case of childhood cancer, the personal threats are severe for both the child, the parents and other family members. For the child, there is the threat to physical integrity, safety, security, and above all, to life. For the parents, there is the threat of losing the child. However, a number of studies have shown that psychopathological disturbances are rarely found in children with cancer or their parents. We may conclude from this that most children and parents use coping strategies that protect them from developing psychopathology. In organising support for families with a child with cancer, much can be learned from children's and parent's perceptions and reactions. When problems of adjustment arise, a thorough analysis of how children and parents perceive their situation, as well as an extensive analysis of their coping efforts, is necessary to direct effective supportive actions. A psychosocial support model is proposed which can be helpful in interpreting these emotions and coping strategies.

The main objective of the present study was to determine which variables predict the emotional adjustment of parents of children with cancer. Therefore, parents' emotional adjustment, in terms of depression, anxiety, feelings of loneliness, helplessness, uncertainty and positive feelings, were predicted with three models. (1) With a child model (including age of the child, time since diagnosis, being in remission or having a relapse, and depression of the child); (2) with a control strategies model (including four distinguished control strategies of parents); and (3) with a child and control strategies model (including a combination of the aforementioned variables). The four control strategies of parents of children with cancer included: the reliance on predictive control (having positive expectations); vicarious control (attributing power to the medical setting); illusory control (relying on luck and wishful thinking); and interpretative control (gaining knowledge). A total of 84 mothers and 79 fathers, of 84 children with cancer with different survival perspectives (in remission or with a relapse) participated in the study, and were assessed about the use of control strategies and adjustment. Lack of positive expectations about the course of the illness was most strongly related to negative emotions for mothers and for fathers. For mothers having a child with a relapse, predicted feelings of helplessness and uncertainty, and reported feelings of depression of the child, proved to be related to the feelings of uncertainty of the fathers. The findings demonstrate that the use of secondary control strategies contribute significantly to the emotional adjustment of parents of children with cancer.

Oncology care is considered a stressful occupation. Little is known about the coping strategies oncology staff members use to deal with the stress of their work. The purpose of this study was to determine whether a self-protective strategy found in parents of children with cancer is also present in pediatric oncology staff. To explore this assumption, staff members' ratings were compared with the parents' ratings on the need for support and with the children's ratings on experienced pain. We also explored the characteristics which staff members attribute to the children and the parents. A total of 76 staff members, 84 children with cancer, and their 163 parents participated in the study. Both gender and number of years working in oncology care were positively associated with increased self-protective reactions in staff members. Male staff members rated medical procedures and the pain children experience in general as less painful than did female staff members. Their judgements about experienced pain in general and the lumbar puncture procedure in particular tended to be lower than the children's ratings. Female staff members attributed more positive characteristics to children with cancer and their parents than did male staff members. Staff members with more years of experience in oncology tended to rate all three medical procedures as less painful than those with less years of experience, and they also attributed more positive characteristics to the children. Staff members should be observant to their perceptions of children and parents, especially if their experience increases.