Measuring Health-Related Quality of Life in Children and Adolescents

Anne Klassen

Veronica Schiariti

Jill Zwicker

Workshop Presenters

Anne Klassen, Associate Professor of Pediatrics at McMaster University. Interests include developing, validating and using HRQOL instruments in clinical and population-based studies.

Veronica Schiariti, Paediatrician & PhD student working as a Research Associate in the CYDiS research unit at BC Children’s Hospital. Interests are health services research and measuring HRQOL of high risk populations of infants and children.

Jill Zwicker, OT & PhD student in Rehabilitation Sciences at UBC & CIHR Quality of Life Strategic Training Fellow. Her interests include measuring HRQOL of children with developmental coordination disorder

Today’s Workshop

1) Issues specific to understanding and measuring HRQOL in children and adolescents

2) HRQOL of preschoolers admitted at birth in 1996–1997 to a tertiary NICU in BC

3) Systematic review of HRQOL studies of children, adolescents and young adults born preterm and/or at very low birth weight

Measuring HRQOL in Children and Adolescents

Anne Klassen, DPhilAssociate Professor, PediatricsMcMaster UniversityMay 2008

Child Welfare Officials to Make Sure Boy They Seized Gets

Chemo – CBCnews.ca, Friday May 9, 2008

Child welfare officials have taken temporary custody of an 11-year-old Ontario boy to ensure he undergoes chemotherapy after his father decided to take him off treatment for his aggressive form of leukemia. A father who cannot be identified says his son is being treated 'like a prisoner' at the hospital where he is being treated for leukemia. His father … told CBC News on Friday that the boy didn't want to continue with the treatments.

"I think about the first time around, what it did to him and how it almost killed him, and when he told me he doesn't want it anymore. He doesn't want to die this way. He would rather die at home in a peaceful, comfortable way.“

The dad, who lives in Hamilton, was briefly shackled by security when he arrived at McMaster Children's Hospital on Thursday with his son for what he believed was a routine appointment. Local Children's Aid Society officials then took custody of the boy due to the father's refusal to admit the son for another round of chemotherapy.

"He had a 50 per cent chance of survival if the treatment was carried out. If the treatment was not carried out, then in fact his chance of survival would be not good. In fact they estimated it would be fatal in six months.“

But the father said doctors told him the boy had a 20 per cent chance of making it through his chemotherapy treatments, then a 50 per cent chance after that, once he undergoes full body radiation and a bone marrow transplant.

… quality > quantity

“I watched a friend be tortured by the cancer treatment. It was the treatment, not the cancer, that was the torture. If the issue comes up for me, I'll refuse treatment. There are worse things than death.”

… quantity > quality

“I lost a cousin at age 28 to leukemia but I also have a friend who survived a year of intensive treatments. Yes, it almost killed him but he is doing OK now and has travelled the world since. He will never be 100% but he is very happy to be alive and able to carry on.”

As people today survive what used to be primarily fatal diseases, and learn to live with complex chronic conditions, the impact of treatment and disease on QOL has become increasingly important to clinicians, researchers and patients

Background

Measurement of QOL emerged in the 1970s when the focus of health care evaluation moved from traditional clinical outcomes (i.e. mortality, morbidity) to the measurement of broader outcomes, such as function (i.e. ability to perform daily activities of life).

Traditional clinical outcomes were seen to not capture the whole range of ways in which a patient is affected by disease and/or treatment

The inclusion of more holistic outcomes, such as quality-of-life (QOL) began to gain increasing interest

QOL is conceptualized as a broad assessment of well-being across various domains

HRQOL is considered to be a subdomain of QOL

Moons et al 2006

Adult QOL publications

Klassen et al 2007

Pediatric QOL publications

Definitional and conceptual issues

Despite increasing interest in QOL, there is no consensus on the definition and/or conceptualization of QOL

A wide spectrum of QOL definitions and conceptualizations exist in the literature

People use the term QOL to mean many different things – this can make comparison of findings difficult or impossible

QOL is often used as a generic label for an assortment of physical functioning and psychosocial variables

There is often no distinction made between measures of QOL and measures of HRQOL

WHO definition of QOL (1994)

“an individual’s perception of his/her position in life in the context of the culture and value systems in which he/she lives, and in relation to his/her goals, expectations, standards and concerns. It is a broad-ranging concept, incorporating in a complex way the person’s physical health, psychological state, level of independence, social relationships, and their relationship to salient features of their environment.”

QOL: The degree to which a person enjoys the important possibilities of his or her life -- Ctr for Health Promotion, U of Toronto

Being: who one is

Physical Being: physical health; personal hygiene; nutrition; exercise; grooming/clothing; physical appearance

Psychological Being: psychological health & adjustment; cognitions; feelings; self-esteem/self-concept/self-control

Spiritual Being: personal values ; personal standards of conduct; spiritual beliefs

Belonging: connections with one's environments

Physical Belonging: home; workplace/school; neighbourhood; community

Social Belonging: intimate others’; family; friends ; co-workers; neighbourhood and community

Community Belonging: adequate income; health & social services; employment; educational programs; etc

Becoming: achieving personal goals, hopes, and aspirations

Practical Becoming: domestic activities; paid work; school or volunteer activities; seeing to health or social needs.

Leisure Becoming: activities that promote relaxation and stress reduction

Growth Becoming: activities that promote the maintenance or improvement of knowledge/skills; adapting to change

What about pediatric QOL?

Many pediatric QOL measures have now been developed and several reviews have been published.

Review of Child QOL measures

Reviewed QOL measures for children aged 0 to 12 yr

Looked at conceptual frameworks & definitions

14 generic and 25 condition-specific identified

Reliability and validity tested for most

Only 3 based on a conceptual model/theory

Health-related QOL

Health is consistently included as an important aspect of QOL

HRQOL is seen as a subset of the overall concept of QOL and includes those parts of QOL that can directly relate to an individual’s health.

HRQOL, Health Status, Functional Status are terms that are often used interchangeably but they measure different things

Functional Status Health Status (HS) HRQOL

An individual’s ability to perform normal daily activities that are essential in order to meet basic needs, fulfill usual roles, and maintain health and well-being (Wilson & Clearly 1995)

“Within the skin” dimensions of health, e.g., Health Utilities Index – vision, hearing, speech, ambulation, dexterity, emotion, cognition, pain/discomfort (Feeny et al 1995)

Can be similar to health status, but differs in that it includes a subjective assessment of the impact of disease and treatment across domains of QOL

HRQOL

Based on the view that health is multidimensional and that it is subjective and therefore should be evaluated by asking a person directly

HRQOL / Health Status

HRQOL and HS were distinct constructs in a meta-analysis of 12 chronic disease studies (Smith 1999) – When rating HRQOL patients gave greater

emphasis to mental health – When rating HS patients gave greater emphasis to

physical health

QOL was only modestly correlated with HS in study of 203 adolescents with CP (Rosenbaum 2008)

Does a healthy life = a high QOL?

People with significant HS and functional status problems do not necessarily have low QOL score

“Disability paradox” -- physically disabled persons unexpectedly experience a good QOL even though most external observers may assume that these people live an undesirable life (Albrecht & Devlieger, 1999)

What measures are available to measure HRQOL in children?

Review findings

30 generic & 64 disease/condition-specific identified

Number & name of HRQOL domains varies substantially

Lower age limit for child-report measures = 5-6 yrs

Many instruments meet accepted psychometric standards

Generic vs. disease-specific

Generic questionnaires allow direct comparisons across disease groups or between sick and healthy groups

Disease or condition-specific instruments address problems specific to only one illness or disease.

– Such instruments, when developed through in-depth qualitative interviews, can help to identify QOL issues of importance to them.

Generic Disease-specific

QOL instruments 30 64 ( 27 different conditions) Asthma (10), cancer (8), epilepsy

(7)

Published > 2001 9 42

Respondents 13 child

4 proxy

13 both

28 child

17 proxy

19 both

1 nurse

16 generic HRQOL measures identified

Research example

HRQOL of Children with ADHD

Background

ADHD is a common psychiatric disorder, affecting approximately 3-5% of children

The aim of treatment is to decrease symptoms, enhance functionality and improve well-being for the child and his/her close contacts

Measurement of treatment response is often limited to measuring symptoms using behavior rating scales

Methods

Subjects: all referrals to the ADHD clinic at BC Children’s Hospital, Vancouver between Nov ’01 and Oct ’02

Data collection: CHQ included in package of symptom rating scales sent prior to outpatient appointment

Clinical data: psychiatric diagnoses extracted from the hospital charts

Measures

Child (CSI), Adolescent (ASI) & Youth (YSI) Symptom Inventory – Measure of symptoms for a wide range of DSM-IV diagnoses

including ADHD subtypes

Conners Rating Scale Hyperactivity Index – Measure of overall psychopathology for children presenting with

ADHD

Child Health Questionnaire (50-item parent form)– Multidimensional HRQOL measure for children 5-18

Interpretation of low and high scores for each CHQPF-50 concept

Domain/Scale Low Score High Score Physical function

Child is limited a lot in performing all physical activities, including self-care, due to health.

Child performs all types of physical activities including the most vigorous without limitations due to health.

Role/physical Child is limited a lot in school work or activities with friends as a result of physical health.

Child has no limitations in school work or activities with friends as a result of physical health.

Bodily pain Child has extremely severe, frequently and limiting bodily pain.

Child has no pain or limitations due to pain.

General behavior

Child very often exhibits aggressive, immature, delinquent behavior.

Child never exhibits aggressive, immature, delinquent behavior.

Mental health Child has feelings of anxiety and depression all of the time.

Child feels peaceful, happy and calm all of the time.

Self esteem Child is very dissatisfied with abilities, looks, family/peer relationships and life overall.

Child is very satisfied with abilities, looks, family/peer relationships and life overall.

General health Parent believes child’s health is poor and likely to get worse.

Parent believes child’s health is excellent and will continue to be so.

Family activities

The child’s health very often limits and interrupts family activities or is a source of family tension.

The child’s health never limits or interrupts family activities nor is a source of family tension.

Family cohesion

Family’s ability to get along is rated “poor”. Family’s ability to get along is rated “excellent”.

Response Rate

335 mailed

79 children ineligible

165 returned

64.5% response rate

Child characteristics

Male, n (%) 106 (80.9)

Child age, mean (s.d.) 10 (2.8)

Comorbidity

0 47 (35.9)

1 56 (42.7)

>2 28 (21.4)

Comorbidity Type

Learning disorder 50 (38.2)

Disruptive Behavior Disorder 45 (34.4)

Other disorder 26 (19.9)

Mean CHQ-PF50 item and domain scores comparing Australian and USA population norms with ADHD children

0

10

20

30

40

50

60

70

80

90

100

PFRP BP GH

REB BEM

H SE PE PTFA FC

Australia

USA

ADHD

Effect sizes comparing ADHD sample with USA population norms

Domain Effect Size

Family-Cohesion -0.66

Self-esteem -0.90

Mental Health -0.97

Parental Impact-Time -1.07

Role/Social Limitations Emotional-Behavioural -1.60

General Behaviour -1.73

Parental Impact-Emotional -1.87

Family Activities -1.95

Psychosocial Summary Score -1.98

Comparisons with other ADHD samples and population norms

Figure 1 Mean CHQ-PF50 scores for different clinical samples compared with USA and Australian normative data

0

20

40

60

80

100

PF RP BP GH REB BE MH SE PE PT FA

Australia

USA

Klassen

Perwien

Landgraf

Sawyer

PF = Physical Function; RP = Role Physical; BP = Bodily Pain; GH = General Health; REB = Role Emotional-Behavioral; BE =

Behavior; MH = Mental Health; SE = Self-esteem; PE = Parental Impact-Emotional; PT = Parental Impact-Time; FA = Family

Activities

Proxy-reported HRQOL

Patient self-report is considered the gold standard in HRQOL assessment

Riley (2004) reviewed research from a range of areas: – studies of cognitive abilities– cognitive interviewing studies of children’s ability to respond to

questionnaires and influences on their responses– psychometric studies of child-report questionnaires– longitudinal research on the value of child report

Children as young as 6 years are able to understand questions about their QOL and to give valid and reliable answers

Complementary perspectives

Self-report is not always feasible or possible for children– may be too ill, unwilling, lacking the necessary language skills,

attention, or cognitive abilities

Proxy may need to report HRQOL scores for child

Even when children are able to participate, proxy ratings may provide a different perspective that is complementary

What is important is what each reporter contributes to the overall understanding of child HRQOL rather than who is more accurate

Eiser & Morse review of 14 studies– Agreement was generally good for more

observable domains of HRQOL (e.g., physical function, behavior)

– Agreement generally poor for less observable domains of HRQOL (e.g., social function, emotional function)

Research example

Agreement between parent- and self-report of HRQOL in children with ADHD

58 children diagnosed with ADHD and their parents participated

CHQ permitted comparisons on 8 HRQOL domains and one single item

Sample characteristics

Child age n (%)

10-11 years 20 (34.5)

12-13 years 20 (34.5)

14-15 years 14 (24.1)

16-17 years 4 (6.8)

Child gender (male) 48 (82.8)

Type of comorbidity  

Learning disorder 25 (43.1)

ODD/CD disorder 16 (27.6)

Other disorder 12 (20.7)

Parent gender (female) 49 (86)

Biological parent 48 (92.3)

Married or common-law 39 (68.4)

Household income

< $40,000 23 (42.6)

$40,000 to $80,000 19 (35.2)

> $80,000 12 (22.2)

Agreement between parent and child

Mean CHQ domain scores comparing children with ADHD and their parents with Australian child and adult population norms

0

20

40

60

80

100

PF RP BP GH BE MH SE FA FC

Austrialian parent

Australian child

ADHD parent

ADHD child

Mean scores, p-value and effect sizes comparing self- and parent-report of CHQ domain scores

Concluding comments

It is crucial to inspect the HRQOL items and domains of any candidate generic or condition-specific measure to ensure that the content of the instrument is appropriate for the research about to be undertaken.

If a new HRQOL measure is required, how you develop a HRQOL measure is important and guidelines exist for the 3 key stages:– Item generation– Item reduction– psychometric evaluation

Caregivers’ Perspective of Health Outcomes of NICU Graduates

Veronica SchiaritiMD, MHSc, PhD (Candidate)Research Associate CYDiS, CCHRBC Children’s Hospital, UBCMay 2008

OUTLINE

Review health related quality of life (HRQOL) outcomes of particular subpopulations of neonatal intensive care unit (NICU) graduates

– Caregiver-reported health outcomes of preschool children born at 28-32 weeks’ gestation

– Perinatal characteristics and parents’ perspective of health status in NICU graduates born at term (≥ 38 weeks)

Rationale

Neonatal follow-up studies have helped to identify a range of negative health outcomes associated with NICU admissions.

Most research to date has focused on preterm infants, particularly the extremely low birth weight (≤ 28 weeks gestational age (GA).

Although many studies report on 1 outcome (i.e. rate of CP, intellectual disability, etc), a comprehensive assessment of the overall burden of morbidity is often lacking.

Rationale for 28-32 week study population

In most tertiary centers, extremely premature infants (≤ 28 weeks GA) and those who experienced major morbidities during NICU admission are enrolled in FU programs.

Most research has focused on the ≤ 28 weeks population, who represent a small proportion of NICU survivors.

Little is currently known about the HRQOL outcomes of the increasing numbers of more mature premature infants.

Rationale for ≥ 38 week study population

Few studies have examined long-term outcomes of term infants who required NICU. The majority were carried out more than 10 years ago.

Most studies of full term NICU graduates have focused on the outcome of specific disease entities (i.e asphyxia, sepsis, etc).

Term infants represent a significant proportion of NICU admissions but generally they are not seen in neonatal FU programs.

Objectives

To measure the HRQOL of preschoolers who were admitted to a BC NICU at birth following a GA of 28-32 weeks and ≥38 weeks.

For the ≥38 week, to investigate the differences in HRQOL in relation to main reason for admission to NICU.

Methods: Data collection

A questionnaire booklet was sent to each mother as her child turned 3.5 years of age including the following HRQOL measures:

Health Status Classification System Preschool Version (HSCS-PS) 14-item HS instrument assesses the following 12 health attributes,

each with 3 to 5 levels of severity:

– Seeing, hearing, speaking, getting around, using hands and fingers, taking care of self, feelings, learning and remembering, thinking and solving problems, pain and discomfort, general health, behaviour

Methods: Data collection

– Infant and Toddler Quality of Life Questionnaire (ITQOL)

8 child concepts – physical abilities (10 items),– growth and development (10),– pain and discomfort (3),– Temperament and moods (18), – general behaviour (13), getting along with others (15),– general health perception (12), – and change in health (1)– and 5 parental concepts

Methods: study sample

2221 surviving infants admitted >24 hrs to one of three tertiary NICUs in BC (March 1996 to June 1997).

718 Full term controls, from the 2 hospitals with a hospital-based primary care unit.

Excluded: – Healthy infants subsequently admitted >24 hs to NICU– language barriers– child/mother died– completion of the questionnaire on the wrong child

Methods: NICU sample

2221 NICU admissions

555 ≤ 33 wks

945 33-37 wks

721 ≥ 38 wks

Exclusions-Parent did not speak English -Baby/mother died -Family moved/could not be traced

251

50 ≤ 27 wks

Questionnaire completion and consent to link data to CNN341 261

201 28-32 wks

Methods: healthy full term sample

718 healthy full term infants

393

Exclusions-Parent did not speak English

-Baby/mother died -Family moved/could not be traced- Not applicable

Questionnaire completion

NICU Nonrespondents

• 28-32 wks GA:• older

• less likely to have major morbidity

• had less intensive interventions

• lower SNAP-II scores less likely to be outborn

≥ 38 wks GA:

• lower Birthweight

• had lower Apgar at 5 min

• had shorter NICU stay

Methods: analysis

HSCS-PS domains were grouped in 4 categories:– neurosensory (seeing & hearing)– motor (motor & self-care)– learning/remembering (speaking & learning) – QoL (feelings, pain, health & behavior)

The Chi-square was used to compare outcomes by type of HSCS-PS problem and severity of problem.

Effect sizes were calculated to estimate the importance of the differences in HRQOL between groups (ie, .20 is small, .50 is moderate, and .80 is large).

28-32 wks study

Results: 28-32 week GA

0

10

20

30

40

50

60

<28 weeks 28-32 weeks healthy controls

% Reported problems by GA for each HSCS-PS domains

Results: 28-32 week GA

Conclusion

Infants born at 28-32 wks GA at preschool age may have significant difficulties in:

– neurosensory, – motor, – learning/remembering, – quality of life– general health

This population is at higher risk for long-term health and developmental problems than has been previously recognized.

Routine and long term neurodevelopmental follow-up should be considered for this population.

≥ 38 wks study

Results: ≥ 38 week GA

0

5

10

15

20

25

30

35

Respira

tory

CNS

Gastrointestin

al

Cardio

vascular

Musculoskeletal

Other

conditions

NICU

Results: ≥ 38 week GA

0

2

4

6

8

10

12

14

Term NICU graduates Healthy infants

% Caregiver reported moderate/severe problems for each HSCS-PS domain

p-value< 0.001 overall comparison, Chi-square test

Results: ≥ 38 week GA

Conclusion

• Term infants represent a significant proportion of

level three NICU admissions in BC. • Term babies admitted to an NICU may have

significant health differences in health status in early childhood.

• Further study is needed to address whether NICU term survivors warrant secondary and/or tertiary level neonatal follow-up.

Relevance

These studies examine current HRQOL of infrequently studied NICU graduates population.

Our results are relevant to healthcare providers, health and education policy makers, and families.

Limitations

Potential non-response bias in postal survey

Exclusion of certain ethnic groups due to language barriers.

Non-random selection of healthy comparison group.

Acknowledgements

Co-investigators: Anne Klassen, Jill Houbé, Anne Synnes, Sarka Lisonkova, Shoo Lee, Canadian Neonatal Network

• The Hospital for Sick Children Foundation (Toronto) provided an operating grant for this study.

• Veronica Schiariti was the recipient of a trainee award from the Neonatal-Perinatal Interdisciplinary Capacity Enhancement (NICE) Team, funded by Canadian Institutes of Health Research (CIHR).

• From British Columbia Research Institute, Veronica Schiariti holds a Graduate Studentship.

References

Klassen AF, Lee SK, Raina P, Chan HW, Matthew D, Brabyn D. Health status and health-related quality of life in a population-based sample of neonatal intensive care unit graduates. Pediatrics 2004; 113(3 Part 1): 594–600.

Saigal S, Rosenbaum P, Stoskopf B, Hoult L, Furlong W, Feeny D et al. Development, reliability and validity of a new measure of overall health for pre-school children. Qual Life Res 2005; 14(1): 243–257.

Klassen AF, Landgraf JM, Lee SK, et al. Health related quality of life in 3 and 4 year old children and their parents: preliminary findings about a new questionnaire. Health Qual Life Outcomes. 2003;1:81

Cohen J. Statistical Power for the Behavioural Sciences. New York, NY:Academic Press; 1977 Achenbach TM, Rescorla LA. Manual for the ASEBA Preschool Forms and Profiles. Burlington,

VT: University of Vermont Department of Psychiatry; 2000 Abetz L. The Infant/Toddler Quality of Life Questionnaire: Conceptual Framework, Logic,

Content, and Preliminary Psychometric Results. Final Report to Schering-Plough Laboratories and Health Technology Associates. Boston, MA: New England Medical Center; July 1994

Klassen AF, Lee SK, Raina P, Chan H, Matthew D, Brabyn D. Reliability and validity of the Infant Toddler Quality of Life Questionnaire. Qual Life Res. 2002;11:684

Quality of Life of Former Preterm and Very Low Birth Weight Infants from

Preschool-Age to Adulthood: A Systematic Review

Jill Zwicker, PhD Candidate Rehabilitation Sciences

Susan Harris, PhD Department of Physical Therapy

University of British Columbia

Vancouver, BC Canada

Background and Rationale

Survival of very low birth weight (VLBW:<1500g) and extremely low birth weight (ELBW:<1000g) infants has increased

Although mortality has decreased, morbidity continues to be high

Long term studies indicate that these children have issues with health, growth, learning, and behaviour

Several authors have examined QOL of preterm (< 37 weeks gestation), VLBW and ELBW infants at older ages

Purpose of Systematic Review

The purpose of the systematic review was to answerthe following question:

In preschool children, school-age children, adolescents and young adults, does preterm birth and/or very low birth weight have an effect on their HRQOL?

Photo from: http://medicineworld.org/images/blogs/5-2007/premature-baby.jpg

Search Strategy

We searched the following databases up to September 2006: MEDLINE and PubMed, EMBASE, EBM Reviews, CINAHL, PsycINFO, and ERIC

We also attempted to locate additional, unpublished, and grey literature by searching SIGLE (System for Information on Grey Literature), ProQuest, PapersFirst, ProceedingsFirst, and other databases

Search Terms

a liberal search strategy was used initially to include terms related to quality of life, such as health status, well being, activity and/or participation limitation, functional status, or motor skill impairment

samples which included preterm, VLBW, or ELBW cohorts were considered for review

studies that focused primarily on low birth weight or small for gestational age infants were excluded

Study Selection

Comprehensive search resulted in 1845 articles Titles were independently reviewed by two

reviewers, JZ and SH Titles were included if either reviewer selected them,

resulting in 206 abstracts for further review Abstracts were independently reviewed using

checklist of inclusion and exclusion criteria We initially obtained 84.5% agreement of which

abstracts to include, followed by consensus after discussion

Study Selection con’t

49 full-text articles were obtained for review

Our initial inter-rater reliability of articles to proceed to quality assessment was 67.3%, with consensus reached after discussion

Articles were excluded: no QOL measure no separate analysis for preterm, VLBW, or ELBW review article, commentary, or editorial

foreign language*

Assessment of Study Quality

19 observational studies remained and were assessed independently by JZ and SH using an adapted checklist from the CRD

Papers received a score out of 10 based on factors such as # of drop-outs, % of cohort followed, control of confounding variables, blinded assessment, etc.

4 studies were excluded at this stage due to lack of comparison group of normal birth weight or full-term children, leaving 15 studies included in systematic review

HRQOL Outcome Measures

Preschool School Adolescent Adult

ITQOL (1) HUI2 (1) HUI2 (2) HUI2 (1)

PedsQL (1) CHQ (2) Informal QOL questionnaire (2)

TAPQOL (3) SF-36 (1)

Scheffzek’s Categories (1)

Summary of Findings

Preschool-Age (6 studies)

4 studies found significant differences between study and control

groups in physical functioning, esp. in motor function (Chien et al.,2006; Fekkes et al., 2000; Klassen et al., 2003, 2004;Theunissen et al., 2001)

Social functioning was also significantly lower for preterm and VLBW groups (Chien et al.; Eiser et al.; Fekkes et al.; Klassen et al.)

Some studies found no significant difference in emotional functioning (Eiser et al.; Klassen et al.) where as other reported that preterm/VLBW group were significantly more anxious (Chien et al.; Fekkes et al.; Theunissen et al.) than controls

Summary of Findings

School-Age (1 study)

HRQOL was significantly lower for the ELBW children compared to the general population based on mean utility scores on Health Utilities Index (Saigal

et al., 1994a,1994b)

Summary of Findings

Adolescents (4 studies) ELBW teens reported significantly lower utility scores than their

peers, but not necessarily lower HRQOL (Saigal et al., 1996)

VLBW teens did not rate themselves as significantly different than their peers, but parents reported significant differences in health and behaviour of their children and their own psychosocial health (Indredavik, 2005)

Parents of ELBW and ELGA teens reported that their children’s QOL was significantly different than their peers both on utility measures (Saigal et al.,2000) and generic health measures (Johnson et al.,2003)

Summary of Findings

Young Adults (4 studies)

no significant differences in QOL (Bjerager et al., 1995) or subjective QOL (Dinesen et al., 2001) reported by adults born at VLBW

significantly lower scores for VLBW adults in objective QOL, which is based on societal standards as opposed to individuals’ experiences and preferences (Dinesen et al., 2001)

significantly poorer performance in physical functioning of VLBW adults compared to NBW adults, but no other significant differences on the SF-36 (Cooke, 2004)

similar utility scores for ELBW and NBW young adults (Saigal et al., 2006)

Discussion

In preschool children, does preterm birthand/or VLBW have an effect on HRQOL?

Differences between the study and control groups were reported in all six studies, suggesting that many preschool children born preterm or at VLBW performed significantly more poorly than their peers in physical, emotional, and/or social functioning (2 studies of high quality, 4 moderate)

All studies involved parent-proxy report (influenced by feelings toward and expectations of child as well as cultural, social, and educational background)

Discussion

In school children, does preterm birthand/or VLBW have an effect on HRQOL?

Only one study, but it was a methodologically rigorous and well-designed retrospective cohort study with a quality rating of 10.

Insufficient information to answer this question from this study, but children born preterm or at VLBW experience academic, motor, psychosocial, and behavioral difficulties at school age (Bhutta et al., 2002; Foulder-Hughes et al.,2003; Grunau et al., 2004; Holsti et al., 2002; Taylor et al., 2000; Whitfield et al.,1997)

Preterm children are 2.6 times more likely than their full-term peers to develop ADHD (Bhutta et al.), a disorder that has been shown to affect QOL of children and youth (Klassen et al., 2004)

Discussion

In adolescents, does preterm birthand/or VLBW have an effect on HRQOL?

Evidence from the 4 (high quality) studies suggests parents of VLBW, ELBW, or ELGA teens perceive that their children had a lower HRQOL than their peers, but the teens themselves did not

These studies highlight the issue of self versus parent proxy report

Discussion

In young adults, does preterm birthand/or VLBW have an effect on HRQOL?

Overall, there was not a significant difference between preterm and full-term young adults, although measures of objective QOL and physical functioning were significantly lower for the preterm groups (3 high quality, 1 weak)

Discussion

Notwithstanding the limited information at school-age, it appears that the impact of preterm birth and/or VLBW on HRQOL diminishes over time

What is not known is whether this is a true change in HRQOL or a function of:– parent versus child report– different definitions and measures of HRQOL– adaptation and changing of expectations as

individuals grow and develop

Limitations

Heterogeneity of populations included in the review

Comparing QOL across age groups is confounded by changes in neonatal interventions over time, which may influence outcomes in different cohorts

By only including studies that used a QOL measure, we consequently excluded a large body of preterm literature reporting on QOL-related concepts

Conclusion

Results of this systematic review indicate that preterm birth and/or VLBW does have an effect on HRQOL at various age groups

Impact of low birth weight and gestational age is greatest during the younger years, but the influence also extends into adolescence and adulthood

Directions for Future Research

a consistent definition and measurement of HRQOL would help to consolidate findings of future follow-up studies of preterm and/or VLBW children, adolescents and adults

more studies on the HRQOL of preterm and/or VLBW children at school-age

qualitative studies exploring HRQOL from the perspectives of preterm and/or VLBW children and adults

Acknowledgments

Charlotte Beck, Health Science Reference Librarian, for her assistance in conducting the literature searches

CIHR Quality of Life Strategic Training Fellowship in Rehabilitation Research for providing me with funding and training