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Consequences of success in pediatrics: young adults with disability benefits as
a result of chronic conditions since childhood
Verhoof, E.J.A.
Publication date
2015
Document Version
Final published version
Link to publication
Citation for published version (APA):
Verhoof, E. J. A. (2015). Consequences of success in pediatrics: young adults with disability
benefits as a result of chronic conditions since childhood.
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General introduction
and outline of the thesis
This thesis is about the psychosocial functioning, the emotional well-being and the factors affecting the vocational success and well-being of young adults with disability benefits as a result of a chronic somatic condition since childhood. The following paragraphs explain the process of growing up with a chronic so-matic condition and the additional challenges it entails. The added value of work participation in general, the work situation of people with chronic conditions and an explanation of the disability benefits are described below. This intro-duction continues with the research questions, the research model and gives an elaboration of the research topics.
CONSEQUENCES OF SUCCESS IN PEDIATRICS
In the past, most childhood diseases were fatal for the majority of children. As a result of improved medical knowledge, technology and practices, including the development of pediatric surgical interventions, pediatric cardiac surgery and pediatric anaesthesiology, pediatricians were able to change the natural course of numerous diseases. This resulted on the one hand in higher survival rates for many diseases, including cancer, metabolic diseases, congenital heart disease and cystic fibrosis 1, 2, but on the other, in an increased number of
dren growing up with chronic health problems and morbidities since these chil-dren have been treated but have not been cured. In addition, the prevalence of some diseases like obesity, asthma and attention deficit hyperactivity disorder has increased as well 1.
Consequently, the number of children growing up with a chronic illness has increased enormously in recent decades. Although numbers vary depending on methods and definitions, by any estimate the scope of the problem is huge. The number of chronically ill children approximates 7 to 20 percent of all children
1, 3 and will likely further increase because of medical advancements 1. In the
Nether lands there are about 500,000 children (14%) with a chronic disorder
3. The prevalence of specific disease and conditions range widely. In the
Neth-erlands, the most common diagnosed illnesses in childhood are asthma (10%) and eczema (6%). Other moderately prevalent illnesses (<1%) are diabetes mel-litus type 1, epilepsy, juvenile idiopathic arthritis (JIA), sickle cell anemia, cys-tic fibrosis, hemophilia, and inflammatory bowel diseases (CBS statiscys-tics Neth-erlands, 2013).
With the increased rates of successful treatment and its positive effect on life expectancy, new questions emerge. Now survival is no longer the major chal-lenge, additional challenges are added. The focus has shifted to the influence of growing up with a chronic condition and to how pediatric illness affects adult life. What happened to those with a childhood onset illness or disability while growing up? And how do they function in adult life? Are they independent and
successful? Are they happy? What help do they need? They live longer, they ma-ture, but what does it mean to grow up with a chronic illness for later societal participation as an adult in our society?
GROWING UP
Growing up and learning to be an adult is for everyone a complicated process wherein people during childhood, adolescence and young adulthood experi-ence multiple transitions 4, 5. These transitions are processes during which youth
develop attitudes, behaviors, and skills to successfully move to adulthood 6. The
transitions to adulthood comprise different developmental tasks, e.g. the con-solidation of their identity, the transition from family life to independent living, and from education to employment. Some transitions are determined by age or development, while others are dictated by ‘systems’, for example, the edu-cational system or healthcare system 5. This journey from childhood to
adult-hood is characterized by starts and stops, anxiety and anger, and hope and de-spair, coupled with an ever-increasing mastery of an essential set of life skills necessary for independence in adulthood 7. Youth development is much more
complex than biological growth and maturation. There are patterns of physi-cal, cogni tive, social and emotional changes that youth go through, but these changes often occur simultaneously or in rapid succession 5. In the 21st
centu-ry, youth development and transition into adulthood have become less straight-forward 5, 8.
Arnett 9 has described a new phase of development called ‘emerging
adult-hood’, a period that exists only in highly industrialized or post-industrial cul-tures that postpone the entry into adult roles and responsibilities until well past the late teens. Such countries require a high level of education and training for entry into the information-based professions that are the most prestigious and lucrative, so many of their young people remain in school into their mid-20s. Marriage and parenthood are typically postponed until well after school-ing is completed, permittschool-ing a period of exploration of various relationships be-fore marriage and for exploration of various jobs bebe-fore taking on the respon-sibility of supporting a family. These young people between the ages of 18 and 30 are on the threshold of adulthood 6. Emerging adulthood is also notable for
its hete rogeneity. It is the time when people’s lives are least likely to be struc-tured by social institutions 10. Children’s and adolescents’ lives are structured by
family and school, and adults lives (in young adulthood and thereafter) by family and work. However, emerging adults typically move away from their families, and their school and work patterns tend to be formed, but are relatively unstruc-tured and unstable 9. The characteristics of this developmental stage include the
li-ving situations and world views; instability; being self-focused and feeling in-be-tween-neither adolescent nor adult; and possibilities - when hopes flourish and young people have unparalleled opportunities to transform their lives. These are not features that exist only in emerging adulthood, but they are more pro-nounced in emerging adulthood than at other ages 9.
GROWING UP INTO ADULTHOOD WITH A CHRONIC DISEASE OR DISABILITY (1)
Children and adolescents with chronic conditions are expected to pass through similar developmental stages as their nondisabled peers, to leave home, deve-lop psychosocially, and define a role for themselves in the community through employment or other activities 12. A chronic condition can have a profound
in-fluence on aspects of development during the transition into adulthood. Chil-dren and adolescents with chronic conditions face many additional challeng-es during this procchalleng-ess. Most children experience several consequencchalleng-es of their health care needs; they depend on medical care or medical aids to some ex-tent, varying from medication use to medical interventions or medical aids (e.g., daily inhalation of corticosteroids, intravenous alimentation, and wheelchair). Some of them also have to deal with (late) treatment effects. Further, transition into adulthood may be more difficult as there will be less exposure to everyday events and therefore to opportunities to learn and explore. Social opportunity is lessened by ill health including frequent hospitalizations, and exclusion from the opportunities afforded by mainstream schooling or unsupervised play with in the age-appropriate peer group 13. The adolescent process of exploring li mits,
reality testing, and self-image development may be severely delayed 7. Enhanced
vulnerability owing to physical or psychological stress may also complicate the transition to adulthood 14.
Concern has risen about the implications of growing up with a chronic condi-tion. The condition and late treatment effects may affect the physical functio-ning and emotional functiofunctio-ning 15, 16. Children with chronic diseases are
some-what more likely than healthy children to show maladjustment 17-19. They tend
to suffer more than average from behaviour problems, especially internalizing problems such as depression, anxiety, and social withdrawal 20. A meta-analysis
of Pinquart and Teubert 21 found children and adolescents with chronic physical
illness to have, on average, lower levels of academic, physical, and social func-tioning than their healthy peers. Impairments were largest for physical
function-(1) International consensus about the conceptual definition of chronic health conditions in childhood is needed. Essential components of a definition are that the disease can be diagnosed according to professional standards, a basic requirement to ensure validity, as well as an expected duration of at least 3 months or the impossibility of cure 11. Disability is the consequence of an impairment that may be physical, cognitive, mental, sensory, emotional, developmental, or some combination of these. A disability may be present from birth, or occur during a person’s lifetime.
ing and smallest for social functioning. In line with this, evidence suggests that school-aged children with chronic conditions, regardless of their diagnosis, are at risk for lower participation in everyday activities than their peers; they par-ticipate less frequently in almost all activities compared with children without a physical disability 22, 23. Furthermore, it is known that youth who have grown up
with a disease are at risk for a delay in age-specific developmental tasks com-pared with peers 24, resulting in a delayed course of life. In summary, a part of
the children with chronic conditions may have difficulties while growing up. Except for survivors of childhood cancer 25 and those with end-stage renal
disease 26, little research exists about whether the challenges and gaps between
youth with chronic conditions and those without, persist into adulthood. Will they finish education, get jobs, and live independently? Can they become au-tonomous in a society where self-sufficiency and independence receive wide-spread recognition and social endorsement 27? And most important, how do they
fulfill their lives in a meaningful and satisfying way? Given that the majority of re-search on emerging adulthood focuses on typically developing, college-attend-ing healthy youth, little is known about the impact of growcollege-attend-ing up with a chron-ic condition on the milestones of emerging adulthood including work partchron-icipa- participa-tion and the adapparticipa-tion to adult life.
WORK
An important way to participate in social life is work. Work generates materi-al and immaterimateri-al benefits. The materimateri-al purpose of work is earning an income to be able to support oneself. Besides money, employment offers a lot of other additional immaterial benefits like the possibility for further self-development, social relationships, development of skills, daily rhythms, and often, self-esteem and meaning in life 28, 29.
Having a paying job at some time during high school has become a near-uni-versal adolescent experience. At first, paid work is episodic and generally quite limited. Many youth start to work informally even earlier, at about the age of twelve, most often in their own neighbourhoods, babysitting, cutting grass, or doing various odd jobs. By the age of 16, adolescent workers are more likely to have formal jobs, working in the retail and service sectors of the economy, for example in fast food restaurants, or retail stores. Employment becomes more regular and more time-consuming during the latter years of high school, with many teens working 20 or more hours per week 30. During and after their study
this process continues; most young adults start a working career which conti-nues in adulthood.
Somatic conditions and work participation
Having a chronic disease or disability often negatively affects the capacity to participate in gainful employment. A poor health condition, however, does not necessarily imply exclusion from the labour market 31. Yet, in various studies, the
labour market position of people with a chronic disease or disability is found to be problematic, with regard to getting a job as well as job continuation 25, 28, 31-33. This, in turn, leads to a variety of economic, social and quality of life
prob-lems 34, 35. The consequences of work disability for patients with chronic
dis-eases include financial difficulties, increased social isolation, decreased confi-dence and self-esteem and stress 36. Besides, several studies among people with
chronic somatic diseases show lower Health Related Quality of Life (HRQoL) 37-40,
worse mental health and more anxiety and depression 41-43 in non-workers than
in those who are participating in the workforce.
Adolescents and young adults with somatic conditions and work participation
Most of the research on the work experiences of people with disabilities has fo-cused on adults while much less attention has been paid to younger people just entering the workforce 29. However, employment and its skill-building
opportu-nities are essential elements for a successful transition to adulthood for young people 44. Adolescents want to work to earn their own spending money to be
able to buy the accoutrements of adolescent life and take part in the leisure ac-tivities among their peers. Working enriches young people’s life experiences, thereby enhancing self-confidence, self-discipline, self-esteem, and indepen-dence. Furthermore, working experiences help young people think about their possible career trajectories and develop work attitudes. The shift out of school into education and occupation is a developmental stage in which young people gain increasing sense of control over their lives, experience a greater responsi-bility, and form an identity which they can use to shape their sense of self that will likely endure throughout adulthood 9. Avoiding unemployment in early
adult-hood is particularly important, because the decisions and actions that occur during this time, can affect income levels and occupational achievement across the lifespan.
In general, youth with disabilities are much less likely to be employed than their peers during high school and often encounter barriers when striving for independence 45-47. This trend of unemployment during adolescence appears
to be continuing in young adulthood. Young adults with disabilities experience considerably lower employment rates compared to their non-disabled peers 29, 48, 49 and their unemployment periods often last longer and they face higher risks
YOUNG ADULTS WITH SOMATIC CONDITIONS AND DISABILITY BENEFITS IN THE NETHERLANDS
In the second half of the 1970s the Netherlands government made a crucial de-cision to support that young people with chronic conditions or disabilities can participate in society with a personal income, independent from parents and others 50. This was effected through the General Disability Act (AAW), a national
insurance against disability, which in principle all residents of Netherlands were insured for. There have been a number of developments and changes in this in-surance for young people over time. The following Act was effective during this study:
The Disablement Assistance (Young Persons) Act; ‘old’ Wajong (2)
The General Disablement Act was abolished in 1997, and on 1 January 1998 a new Act came into force for young people and students who have become disabled at an early age and therefore do not qualify for wage-related benefits: the Dis-ablement Assistance (Young Persons) Act, known as ‘Wajong’ in Dutch. This Act provides (supplementary) income support as well as support to find employment and if necessary support at the work place. The benefit is not obli gatory or au-tomatically including all young adults with disabilities: the initiative for applica-tion lies with the individual and his/her social environment. A Wajong benefit is payable not earlier than the 18th birthday, for as long as the inability to work lasts and ends when the recipient reaches the age of 65. The Employee Insur-ance Agency (UWV) implements the Act and is also responsible for determining the level of benefit paid. The level of benefit received under the terms of the Act depends on age and the amount someone can earn from a job; Wajong in-come support is a supplementary payment on top of what a young person with a chronic disease or handicap is able to earn from work. The UWV insu rance physicians assess the work ability of the claimant considering the medical his-tory and prognosis and describe the limitations for work. Based on the limita-tions assessed by the insurance physician, employment specialists investigate what jobs the claimant could perform on the labour market despite these lim-itations and what work opportunities are available. Depending upon the ques-tion whether or not the young disabled can theoretically earn a minimum wage with a job, a disability pension will be refused or awarded 46. A part of those who
receive a Wajong benefit is potentially (partially) capable to work. The other part is declared fully unfit for work.
Numbers of Wajong applicants
The numbers claiming benefits under the Wajong scheme rose from 40,000 in 1976 to 127,400 in 2000 and 238,700 in 2013 (table 1) 50, 51. This meant that one
out of every 20 people aged 18 (4.8%) was claiming and receiving this disability benefit 52. This increase contrasted with the numbers of claims made under the
WAO/WIA Act – which caters for people who develop a disability at some stage during adulthood – which has seen modest falls in recent years. A number of studies have examined the possible causes of this take-up of the scheme. The reasons behind the increase in claimants of this disability benefit are complex and likely to be multi-factorial. For example, the eligibility rules for entry into benefits systems are likely to have changed, diagnostic criteria may also have changed, and systems may have become more efficient at recognising health problems. At a personal level, there may have been a real increase in health problems or there may have been a decline in employment prospects which has pushed people to register as disabled. Social reasons, especially those relating to the level of family support, may also play a role. Also, statistical or admini-strative aspects, the maturation of the scheme, and greater awareness of the scheme amongst potential claimants can have influence. In addition, the cur-rent economic climate could affect attitudes towards the employment of young people with health problems. It is not known what proportion of children and young people who grow up with a chronic somatic disease or disability eventu-ally ends up in the Wajong.
TABLE 1: Number of recipients of Wajong benefits (2003 – 2013) Influx Wajong benefit Outflux Wajong benefit Current benefits end of year 2003 8.200 4.400 138.500 2004 9.400 4.900 142.600 2005 10.400 5.600 147.200 2006 13.600 4.900 155.900 2007 15.300 4.400 166.800 2008 16.100 4.300 178.600 2009 17.600 4.300 192.000 2010 17.800 4.600 205.100 2011 16.300 5.200 216.200 2012 15.300 5.100 226.500 2013 17.700 5.500 238.700
Gender, age and diagnoses of applicants at Wajong influx
Table 2 shows the gender, age and diagnoses of new Wajong applicants. There are slightly more men than women applying for a Wajong benefit. Most applicants enter the Wajong at an early age. The Wajong influx contains for the most part of youngsters having developmental disorders. Another large group of young peo-ple have been diagnosed with psychiatric disorders (25%, for instance schizo-phrenia or personality disorder). These statistics only partly reflect the reali-ty because 42% have multiple conditions diagnosed 52. About 14 percent is
hav-ing a disability benefit because of somatic (physical) malfunctions or disorders.
Work participation of Wajong applicants
Although some of the young adults with Wajong benefits were unable to work in any way because of their limitations, others could and were willing to work. However, those young adults with a Wajong benefit who were able to work re-main under-represented in the labour market. Data from the Social and Eco-nomic Council of the Netherlands (SER) indicated that around 60% of people on Wajong benefit were able to work (at least 12 hours a week). In reality, around 25% did so (table 3) 51, 51, 53. The greatest part of the young adults with Wajong
benefits has difficulty participating in employment. The problem of the low
em-TABLE 2: Gender, age and diagnoses of Wajong applicants at influx in 2008, 2010 and 2013 2008 2010 2013 % % % Gender Men 58 57 58 Women 42 43 42 % % % Age 18 and 19 year 58 62 62 20 t/m 24 year 20 22 24 25 t/m 34 year 13 11 11 35 year and older 9 5 2 Diagnosis group % % % Development disorders 60 66 67 Mental retardation (intellectual
impairments, learning problems)
38 37 35 Autistic spectrum impairments 11 15 18 Attention deficit (ADHD, ADD) 4 8 8 Other development discorders 6 5 5 Psychiatric diseases 26 21 19 Somatic diseases 15 13 14
ployment rate among people on Wajong benefit is in finding employment, as well as in holding on to it. A high proportion of those who find employment lose their jobs within a short time 53. Around 20% of the people who are not working
when they get the Wajong disability benefit find a job within a year. Almost half of those who find a job, lose their job again within one year 53.
THE EMWAJONG STUDY
Among the young adults with Wajong benefits as a result of a chronic illness or disability, some have a congenital disease or disability, some are survivors of a severe disease in childhood, some have a long-standing yet less severe condi-tion and some have been ill or disabled since adolescence only. Health status and functional limitations, however, do not tell us everything about the impact of growing up with a chronic condition on well-being and participation in so-ciety. The major question is that of their well-being, independence and future. Since the Wajong population can be considered as the most vulnerable young adults with chronic conditions - those who have to apply for disability bene-fits as a result of their conditions - it is essential to gain insight into the chal-lenges they may be encountering in dealing with their condition while becom-ing an adult. All in all, the (often hidden) psychosocial consequences of the pe-diatric success story on growing up and social integration deserves attention. If we can gain insight into the adjustment and process of growing up of the Wa-jong population, their emotional well-being and factors affecting their vocation-al careers, we may be able to develop strategies to support this vulnerable pop-ulation towards adulthood independence and economic participation. It is cru-cial that these young adults in our society are able to find their place in the adult world.
TABLE 3: Number of working people with a Wajong benefit Number of people with a Wajong benefit Number of working people with a Wajong benefit Percentage working people with a Wajong benefit 2005 147.161 38.733 26,3% 2006 155.860 39.814 25,5% 2007 166.805 45.613 27,3% 2008 178.590 45.900 25,7% 2009 191.956 47.598 24,8% 2010 205.134 49.700 24,2% 2011 216.240 53.844 24,9% 2012 226.481 53.749 23,7% 2013 238.708 53.890 22,6%
Aim and research questions
The purpose of this study was to evaluate the psychosocial functioning, the emo-tional well-being and the factors affecting the vocaemo-tional success and well-be-ing in young adults with a Wajong benefit for a childhood-onset chronic somat-ic condition while growing up. The study was aimed at young adults with a dis-ability benefit as a result of a somatic illness or condition. The primary research questions of this study are as follows:
1. How does the psychosocial developmental trajectory (course of life) of young adults with disability benefits as a result of a somatic condition com-pare to that of a reference group?
2. How does the Health Related Quality of Life (HRQoL) and anxiety and de-pression of young adults with disability benefits as a result of a somatic con-dition compare to that of reference groups?
3. What are the Illness Cognitions (IC) of young adults with disability benefits as a result of a somatic condition? Is there an association of IC with HRQoL, anxiety and depression in this study population?
4. Is there a difference in the psychosocial development trajectory of young adults with and without a disability benefit?
5. What are the disease characteristics, work characteristics, and work moti-vation of young adults with disability benefits as a result of chronic condi-tions and the association of those factors and IC with the likelihood of be-ing employed?
Research model
The guiding framework (figure 1) for this thesis is the International Classification of Function, Disability and Health (ICF) (World Health Organisation, 2001). This model integrates the major models of disability - the medical model and the so-cial model - as a bio-psycho-soso-cial synthesis and measures health and disabili-ty at both individual and population level and covers the entire life span. The ICF classification does not classify people according to a diagnosis but describes different ways of functioning. The model identifies three levels of human func-tion: body functions and structures (perspective of the body), activi ties (per-spective of the individual), and participation (per(per-spective of society) 54. The
functioning (both the capacity and the performance) of the individual can be in-fluenced by the disease or disorder, but also by personal factors (e.g. age, gen-der, self-esteem and motivation) and by environmental factors (e.g. the social environment) 54. ‘Disability’ is the ICF umbrella term for impairments (a problem
in bodily functioning), activity limitations (like walking or personal care) and par-ticipation restrictions (a restriction an individual may experience in involvement
in life situations, like labour, going to museums, going on holidays and meeting friends) 55, 56, influenced by personal and environmental factors.
The ICF model conceptualises functioning and disability as a dynamic interac-tion; disability can lead to loss in abilities, which in turn can lead to problems in participation in society. Young adults with disability benefits because of chronic conditions may experience problems in every day life. They may have participa-tion restricparticipa-tions while growing up because of difficulties in performing desired activities, which can influence their functioning and health condition. Outlined below are the topics in this study that could influence the interrelation between the functioning and disability of young adults with Wajong benefits.
Medical parameters and socio-demographic factors
The influence of socio-demographic factors on adjustment is often studied in pediatric literature. For instance, boys tend to have more externalizing prob-lems than girls 57. Furthermore, higher socio-economic status, higher
educa-tional level of the parents, higher income, and belonging to a non-minority were protective factors associated with child and parent adjustmen 58. A number of
illness severity parameters are found to be associated with psychosocial prob-lems, such as: the intensity of the treatment and hospitalization 59, visible
symp-toms of illness 60, 61, limitations in school attendance 60, and a poor or
uncer-tain disease prognosis 62. However, despite these empirical associations with
ill-ness characteristics, most studies that include both illill-ness- and psychosocial risk-factors in a multivariate regression model show that illness factors account
FIGURE 1: The international Classification of Functioning, Disability and Health (ICF)
HEALT CONDITION (disorder or disease) Body functions and structures Environmental factors Personal factors Participation Activities
for little of the variability in psychosocial adjustment compared to psychosocial factors 63-65. Less is known about medical and socio-demographic parameters
and adjustment in emerging adulthood. A meta-analysis of Pinquart 66
indicat-ed that stronger differences in the achievement of developmental milestones - finishing advanced education, finding employment, leaving the parental home, marrying, and becoming parents - were found for respondents with neurologi-cal illnesses and sensory impairment than in individuals with other chronic dis-eases. Lower success rates were also observed if the illness/disability is highly visible to others and in the case of longer illness duration 66.
Course of Life
The developmental consequences of growing up with a childhood chronic con-dition may have consequences in adulthood. The fulfilling of age-specific de-velopmental tasks in youth is of great importance to the adjustment in adult life
67, 68. The developmental tasks and the resulting developmental milestones that
are necessary in the development of a child are referred to as ‘the course of life’ (CoL). The normal developmental tasks of childhood and adolescence involve the attainment of social and academic competence, the development of peer relationships and increasing independence from parents. Children and adoles-cents with chronic diseases are expected to pass similar developmental stages as their nondisabled peers. For patients with impairments, reaching these de-velopmental stages can be challenging. They face many additional challenges to negotiate and obtain normative developmental milestones and maintain adap-tive functioning. The burden of the disease or disability, treatment, hospitaliza-tion and long-term medical sequelae, and decrease in social and school-based participation could be a threat to the accomplishment of developmental tasks, resulting in a delayed course of life.
From a developmental psychological point of view, risk behaviour is also rel-evant. To a certain extent, displaying risk behaviour – in terms of trying out – is part of the development from being a teenager to becoming an adult. Adoles-cents grown up with a chronic condition may display less risk behaviour than do their healthy peers, because they are keenly aware of the vulnerability of their health 69, 70. Moreover, increased parental involvement as a result of the
paediat-ric condition experience 71 may limit children’s opportunities to have
unsuper-vised time with peers, which may decrease their opportunities to engage in risk activities with peers. On the other hand, we could possibly expect to observe more risk behaviour among adolescents with a childhood-onset, in compensa-tion for the limitacompensa-tions that were imposed upon them by disease in their youth. The course of life has been investigated in several disease specific groups 24, 72, 73.
for a delayed CoL compared with the peer group 24. Particularly the CoL of
pa-tients with end-stage renal disease and survivors of childhood cancer was found to be hampered 24, 73. So far, little is known about the CoL of young adults with
somatic chronic diseases or disabilities with disability benefits.
Health Related Quality of Life
WHO defines Quality of Life (QoL) as an individual’s perception of their position in life in the context of the culture and value systems in which they live and in relation to their goals, expectations, standards and concerns. It is a broad rang-ing concept affected in a complex way by the person’s physical health, psycho-logical state, personal beliefs, social relationships and their relationship to sa-lient features of their environment (www.who.int). Health-related quality of life (HRQoL) refers to how health impacts on an individual’s ability to function and his or her perceived well-being in physical, psychological and social domains of life. Physical functioning refers to activities of daily living, as well as to physical symptoms resulting from disease or treatment. This physical functioning part of HRQoL includes basic activities, such as self-care (eg, bathing, dressing), as well as work-related activities (whether paid or not) such as housework and ca-reer. Social functioning refers to social relationships and interactions, and to societal integration. The psychological well-being part of HRQoL is somewhat more subjective than the functioning part, because it relies almost exclusively on the internal, subjective perceptions of the respondent. Psychological func-tioning ranges from severe psychological distress to a positive sense of well-be-ing, and it may encompass cognitive functioning. Included is whether the person feels happy, sad, depressed, or anxious (emotional well-being), whether they are in severe pain or have no pain at all, and whether they are energetic or lethar-gic 74. Beyond this core set of HRQOL domains, additional issues may be
rele-vant. In the case of young people with chronic illness, developmental changes should be taken into consideration. Definitions of HRQoL therefore should in-clude goals and aspirations as well as the constraints imposed through ill-health and treatment 75.
The past 20 years has seen an explosion of studies of HRQOL in with chron-ic conditions, reflecting a widening of focus from biomedchron-ical outcomes to in-clude psychological and social dimensions of health. This expansion in the field of HRQoL studies is mainly focussed on children and adults. Overall, children with a chronic disease are at a greater risk of HRQoL problems than their healthy peers, but not in all cases, and not on all domains 60, 76, 77. A lot of studies
de-scribe the HROoL of adolescents with a chronic childhood condition 25, 78-80, but
less studies report the HRQoL of young adults with a chronic childhood condi-tion. Most studies on HRQoL on young adults with a chronic condition have been
illness specific and show affected HRQoL scores 81, 82. For example, survivors
of childhood cancer reported a lower HRQoL than their peers 81 and specific
chronic health problems had a negative impact on survivors’ HRQoL 83. The
lite-rature on adults with chronic illness since childhood found a lower HRQOL and more emotional problems compared to the general population 12-15.
Socio-de-mographic characteristics such as gender, age and education are generally of little significance for the general health perception of adults with a chronic ill-ness or disability. The severity of the condition seems to have more impact on their HRQOL than the nature of the condition itself 16.
Having a paid job can have positive effects on the wellbeing and QoL of peo-ple of working age. Several studies among peopeo-ple with chronic somatic diseas-es show lower Health Related Quality of Life in non-workers than in those who are participating in the workforce 37-40. Nevertheless, there are also studies
in-dicating only small differences in the HRQoL between working and non-work-ing people with a chronic somatic disease 56, 84. Particularly, the severity of the
physical disability carries weight in the evaluation of someone’s HRQoL. Par-ticipation in work and well-being appears to be less associated when adjust-ed for the severity of physical disability 56. However, few studies have focused
on the emotional well-being of young adults with childhood onset chronic con-ditions who encounter barriers when pursuing employment, as compared to young adults without chronic conditions. Also, the HRQoL and emotional func-tioning of young adult beneficiaries with a childhood-onset somatic condition as a group has never been studied.
Anxiety and Depression
In the general adult population, having a chronic illness is a significant risk factor for the development of mental health disorders such as anxiety and depression
85. Internalizing problems, such as anxiety and depression are also described as
potential concerns for children with chronic illness 86. Several meta-analyses on
children with chronic illness have found elevated levels of anxiety and depres-sion in children with chronic physical illness 87, 88. Two meta-analyses of Pinquart
and Shen 89, 90 compared levels of anxiety and depression of children and
ado-lescents suffering from chronic illnesses with their healthy peers across a large number of diseases. Strongest elevations were found for chronic fatigue syn-drome, migraine⁄tension headache, sensory impairment and epilepsy in rela-tion to anxiety, and in chronic fatigue syndrome, fibromyalgia, cleft lip and pa-late, migraine/tension head ache, and epilepsy in the case of depression. Most research on mental health and chronic conditions concern children or adult populations. Less is known about anxiety and depression on those in the peri-od of emerging adulthoperi-od.
Illness cognitions
There is increasing evidence that psychological factors play an important role in the adaption to living with a chronic disease 36. Studies on adaptation to
chron-ic illness have shown that there often is a considerable discrepancy between the level of illness-related dysfunction as reported by patients and the underly-ing pathology of their disease. The magnitude of the physical, mental, and social problems that patients with chronic diseases present can vary greatly from pa-tient to papa-tient, even in papa-tients with the same medical condition or the same severity of disease. The weak relationship between biomedical parameters and wellbeing has given rise to the hypothesis that other, psychosocial, factors con-tribute to health outcomes in patients with chronic diseases.
In recent years, the role of illness perceptions and coping responses of pa-tients have especially been highlighted. Health psychologists have shown that, to make sense and respond to the problems caused by a chronic illness, patients create their own explanations or ‘beliefs’ on their illness 91. When patients are
diagnosed with an illness they generally develop an organised pattern of beliefs about their condition. These illness perceptions or cognitive representations directly influence the individual’s emotional response to the inherently aversive character of a chronic condition, to maintain a sense of balance and to achieve a satisfying quality of life. This in turn determines how patients respond to the chronic condition; in their coping behaviour such as adherence to treatment 36, 92. It has been commonly assumed that the way adult patients perceive and think
about their illness accounts for much of the individual differences in their phys-ical and psychologphys-ical health status 92. Specifically, patients who report high
lev-els of helplessness and low levlev-els of acceptance with regard to their illness, em-phasize the negative aspects of their condition. They generalize their cognitions to all facets of daily life and consequently experience worse physical and psy-chological functioning 92-95. In this view, illness cognitions (IC) can be considered
as prognostic factors predicting physical functioning, psychological distress and therefore possibly adaption to society.
In addition, there is increasing evidence that illness cognitions could play a role in work participation. A review study of Hoving et al. 36 exploring the
rela-tionship between illness perceptions and work participation in patients with somatic diseases and complaints found promising evidence. Even though the number of studies in the review was limited, all included studies report signifi-cant findings between one or more illness perception dimensions and measures of work participation. In particular, all studies found that non-working people perceive more negative consequences of their disease. This suggests that ill-ness perceptions play a role in the work participation of patients with somatic diseases or complaints. Most of the research on IC has focused on adults while
much less attention has been paid to younger people who grew up with a chron-ic condition. Also, the IC of young adult benefchron-iciaries as a group has never been studied.
Factors that influence participation in work
There is little detailed information about the employment patterns of young people with chronic conditions compared to their non-disabled peers. Al-though there are studies on (return-to-)work factors among adults with chron-ic conditions 96, the factors found for impaired adults may not be the same as
those for young adults who are at the beginning of their career. Knowledge re-garding factors that influence the work participation of these young adults in practice is hardly available. Previous studies mentioned the following factors: socio-demographics (age, gender, education, geographic location and the num-ber of people living in the household), psycho-social functioning, severity of dis-ability, type and duration of disdis-ability, level of depression and dispositional op-timism 29, 42, 45, 97, 98. A systematic review concerning factors that promote or
hin-der work participation in young adults with disability benefits found motor im-pairment, low physical ability, co-morbidity, epilepsy, IQ lower than 80, young-er age at diag nosis and highyoung-er radiation grade in cancyoung-er survivors to be relat-ed to negative employment outcome 42. However, the abovementioned studies
focussed on specific disease categories or examined both physical and mental conditions. So far, little is known about the early determinants of work integra-tion of young adults with disability benefits because of a chronic somatic con-dition as a group.
Motivation
Young people’s motivation points out to be a critical ingredient in achievement in education and in occupational outcomes later in life. Those with a firmer work motivation are more likely to participate successfully on the labour mar-ket. Among the young adults with chronic conditions, motivation likely leads to greater initiative to search for employment. In addition, motivated people re-ceive more guidance or support to find a job, and coaches and employers react positively to motivated individuals 99, 100.
Also, lack of motivation has been found to be an internal barrier to employ-ment for these young adults 101. It is hypothesised that motivation and early
em-ployment history are interrelated, such that early frequent job losses had an im-pact on the individual’s self-confidence and increased anxiety, which led to a likelihood of a pattern of job loss 101.
The work motivation of young adults with Wajong benefits as a result of so-matic complaints has never been studied. What about their work motivation?
Are their aspirations associated with their illness cognitions and better employ-ment outcomes in early adulthood?
STUDY DESIGN
The EMWAjong study is a large cross-sectional study. All young adults between 22 and 31 years of age who claimed a Wajong benefit (3) in the year 2003 or 2004 for a chronic somatic condition were invited to fill in an online question-naire via a letter. The target group is a heterogeneous group with different types of health conditions and different abilities and limitations. Those with no sus-tainable work opportunities (classified as fully incapable for work) were exclud-ed because the EMWAjong study aimexclud-ed to identify factors that could help to im-prove vocational success. Those with serious cognitive impairment or psychiat-ric conditions were also excluded because the EMWAjong study was directed at young adults with childhood-onset somatic illness or disability.
In total, 2,046 persons were invited to take part in the study. To maintain the privacy of the beneficiaries, the invitation letter was sent by UWV, the Dutch benefits agency. The letter contained a personal log-in code, a password and a link to the online questionnaire. After two weeks, participants received a re-minder letter. Participants who completed the entire questionnaire received a gift voucher. The sample was recruited at the end of 2009. The study was per-formed according to the regulations of the medical ethical committee; due to the once-only internet-based nature of the survey, no formal approval by the medical ethics committee was required.
The standardized and validated questionnaires that were used in the EMWA-jong-study, related to the study questions, are presented in table 4. Due to pri-vacy reasons, no information about the chronic conditions of the participants was provided by the benefits agency. This information was therefore derived through beneficiaries’ self reports. The questions concerning disease-charac-teristics were based on existing questionnaires 58 and recommendations of
ex-perts in the field. Work participation was defined in conformity with Statistics Netherlands (CBS), as performance of paid work for at least 12 hours per week for an uninterrupted period of 6 months.
(3) During this study, people who were not able to earn as much as a healthy person with the same qualifications and experience were classified as being occupationally disabled under the terms of Wajong. Those eligible for Wajong benefit were at least 25 per cent occupationally disabled on their 17th birthday or were younger than 30 and became 25 per cent occupationally disabled during the time as a student, as a result of which they were unable to work on a full-time basis after their studies.
OUTLINE OF THE THESIS
This thesis focuses on evaluating the psychosocial functioning, the emotio nal well-being and the factors affecting the vocational success and well-being in young adults with a Wajong benefit for a childhood-onset chronic somatic con-dition while growing up. The general introduction of this thesis is covered in the current chapter (chapter 1). In chapter 2 we assessed the developmental trajec-tory (course of life) of young adults with a disability benefit as a result of chro-nic somatic conditions and compared this with that of peers from the gene ral Dutch population. Chapter 3 compares the HRQoL, anxiety and depression of
TABLE 4: The standardized and validated questionnaires used in the EMWAjong-study Research question Purpose Measures
1 (Chapter 2 and 4) How does the courses of life young adults with disability benefits as a result of a somatic conditition compare to that of a reference group?
Course of life questionnaire (LVJV) 102
2 (Chapter 3 and 4)
How does the Health Related Quality of Life (HRQoL) and anxiety and depression of young adults with disability benefits as a result of a somatic condition compare to that of reference groups?
RAND-36 103
Hospital Anxiety and Depression Scale (HADS) 104
3 (Chapter 5) What are the Illness Cognitions (IC) of young adults with disability benefits as a result of a somatic condition? And is there an association of IC with HRQoL, anxiety and depression in this study population?
Illness Cognition Questionnaire (ICQ)
92 RAND-36
Hospital Anxiety and Depression Scale (HADS)
4 (Chapter 6) Is there a difference in the psychosocial development trajectory of young adults with and without a disability benefit?
Course of life questionnaire (LVJV)
5 (Chapter 7) What are the disease characteristics, work characteristics, and work motivation of young adults with disability benefits as a result of chronic conditions and the association of those factors and IC with the likelihood of being employed?
Illness Cognition Questionnaire (ICQ) Subscale of Work and Life Attitude Scale (WLAS) 105
our study group with that of peers from the general Dutch population. Subse-quently, the course of life and the HRQoL of young adult women with a Wajong benefit as a result of Juvenile Idiopathic Arthritis compared with the general Dutch population is described in chapter 4. We examined the illness cognitions (IC) of young adults with Wajong benefits because of chronic health conditions in chapter 5 and examined whether there is an association of IC with Health Re-lated Quality of Life (HRQoL), anxiety and depression in this study population. In chapter 6 we investigate possible differences between the psychosocial de-velopment trajectory of young adults with childhood cancer with and without a disability benefit. Finally, we examined the disease characteristics, work char-acteristics, and work motivation of young adults with disability benefits as a re-sult of chronic conditions and the association of those factors and IC with the likelihood of being employed in chapter 7. This thesis ends with the general dis-cussion in chapter 8 of the main results, a reflection of the methodology, clini-cal and societal implications and key messages.
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