Support Care Cancer (2010) 18:1207–1217 DOI 10.1007/s00520-009-0751-y
ORIGINAL ARTICLE
Quality of life questionnaires for children with cancer and childhood cancer survivors: a review of the development of available measures Anne F. Klassen & Sonya J. Strohm & Heleen Maurice-Stam & Martha A. Grootenhuis
Received: 13 February 2009 / Accepted: 23 September 2009 / Published online: 16 October 2009 # Springer-Verlag 2009
Abstract Objective To identify and appraise all published quality of life (QOL) measures developed for use with children with cancer and childhood cancer survivors. Methods MEDLINE, CINAHL, EMBASE, PsycINFO, CancerLit, and Sociological Abstracts were searched from the inception of each database to 15 June 2009. Included articles were ones that described the development and/or psychometric evaluation of a QOL measure developed for use with children with cancer or childhood cancer survivors. Articles were appraised for adherence to internationally recommended guidelines for item generation, item reduction, and psychometric evaluation. This study was funded by a grant from the Canadian Cancer Society. Anne Klassen is a recipient of Canadian Institute of Health Research career award. We acknowledge the contribution of Maureen Rice for her help with the literature search.
Results Thirteen QOL questionnaires were identified by our search. Eleven measures are applicable to measuring QOL in children with any type of cancer, and two are specific to children with brain cancer. Four measures can be used to measure QOL in children undergoing cancer treatment, six can be used with children on or off treatment, and three are specific to childhood cancer survivors. While all measures underwent some degree of formal development and validation, item generation often did not involve children with cancer or their parents, and a number of measures did not describe or utilize recommended methods for item reduction and psychometric evaluation. Conclusions Most of the measures identified in this review were designed to measure QOL concerns of children with any type of cancer and at any time, during treatment or survivorship. Our findings can help researchers and clinicians identify scientifically sound measures.
A. F. Klassen (*) Department of Pediatrics, McMaster University, 3A, 1200 Main Street W., Hamilton, ON L8N 3Z5, Canada e-mail:
[email protected]
Keywords Cancer . Oncology . Quality of life . Psychometrics . Systematic review . Childhood survivors
S. J. Strohm Department of Pediatrics, McMaster University, IAHS Bldg, Room 408, 1400 Main St. W., Hamilton, ON L8S 1C7, Canada e-mail:
[email protected]
Introduction
H. Maurice-Stam : M. A. Grootenhuis Psychosocial Department, Pediatrics, Academic Medical Centre G8 224, University Hospital of Amsterdam, Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands H. Maurice-Stam e-mail:
[email protected] M. A. Grootenhuis e-mail:
[email protected]
There is growing interest in assessing quality of life (QOL) in children with cancer, especially now that most children survive their disease [1]. An increasing number of diseasespecific QOL questionnaires are available and can be used to determine the immediate and long-term effects of cancer treatments on the health and well-being of children with cancer. However, QOL questionnaires vary in terms of the QOL domains assessed as well as their adherence to recommended standards for development and psychometric rigor. Researchers and clinicians, therefore, need a way to choose among the various QOL tools available for use.
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Since the early 1990s, there has been increasing international consensus with respect to appropriate methods for the development and validation of QOL instruments, culminating with the 2002 report by The Scientific Advisory Committee of the Medical Outcomes Trust [2]. While the content and psychometric properties of QOL instruments developed for children and youth have been extensively reviewed [3–17], no review provides the level of information summarized within these guidelines for pediatric oncology QOL instruments. The only relevant published review was limited to English language articles, described instruments published between 1975 and 2000, and focused on instruments that could be used to assess QOL only in children with one kind of cancer (acute lymphoblastic leukemia) [18]. To address these various limitations, we performed a systematic literature review to identify and appraise all published QOL measures developed for use with children with cancer and childhood cancer survivors. Such a review is needed to help researchers and clinicians identify scientifically sound measures.
QOL questionnaire contained no information regarding instrument development or validation, the corresponding author was contacted by email. Eligible questionnaires were appraised for adherence to guidelines and criteria for the development and validation of health outcomes instrument as described by The Scientific Advisory Committee of the Medical Outcomes Trust [2] and the USA Food and Drug Administration (FDA) [19] (see Table 1). For each instrument, we examined published psychometric papers to determine whether recommended procedures for item generation, item reduction, and psychometric evaluation were used in the development process [2, 19]. In addition, we examined whether minimum standards for internal reliability and reproducibility were achieved (i.e., reliability coefficients of at least 0.70 for group level comparisons [2]) and findings for construct validity and responsiveness. Information about each instrument was extracted from articles by two independent reviewers (SS and HM) using a data extraction form. Discrepancies were resolved by a third reviewer (AK).
Methodology
Results
Medical Literature Analysis and Retrieval System Online (MEDLINE), Cumulative Index to Nursing and Allied Health Literature (CINAHL), Excerpta Medica Database (EMBASE), PsycINFO, CancerLit, and Sociological Abstracts were searched from the inception of each database to 15 June, 2009. The search strategy, developed by a medical research librarian, involved searching for a subject heading or text word for the following terms: “quality of life,” “health related quality of life,” “quality adjusted life years,” “health status,” “functional status,” “well-being,” and “patient reported outcome” along with specifically named generic and cancer-specific QOL questionnaires the team was familiar with (search strategy available upon request). These terms were combined with a subject heading or text word for cancer and limited to children. No limitation was made on language. Included articles described the development and/or psychometric evaluation of a QOL measure developed for use with children with cancer or childhood cancer survivors. Articles that focused exclusively on one specific QOL domain (e.g., fatigue, eating disorders) were excluded because they did not assess overall QOL. Articles that did not provide evidence of any development or validation process (i.e., ad hoc instruments) were excluded. The titles and abstracts of all identified articles were examined independently by two reviewers. The full text of all potentially relevant papers were obtained and examined by two reviewers. Citations for included articles were examined to identify additional articles. If a paper about a
A total of 5,993 articles were identified by our search strategy, of which 276 articles on the topic of QOL were examined in detail, and 26 [20–45] were found to meet our inclusion criteria. A citation review of the reference lists from these articles and other articles found by the authors added one additional paper [46]. In addition, one author we emailed provided an additional citation [47] resulting in a total of 28 articles that discussed the development and psychometric evaluation of 16 disease-specific QOL instruments. Eleven instruments [22–36, 39–47] are applicable to measuring QOL in children with any type of cancer, and two [37, 38] are specific to children with brain cancer. Four instruments [22–25, 46] can be used to measure QOL in children undergoing cancer treatment (one is specific to stem cell transplantation (SCT) [23], and another is specific to bone marrow transplantation (BMT) [22, 46]), six [20, 21, 30–37, 39, 40, 42–45, 47] can be used to measure QOL of children on or off treatment, and three [26–29, 38, 41] are specific to childhood cancer survivors. Table 2 shows detailed information for each QOL measure, including whether there is a child and/or proxy version available along with the total number of items and specific domains measured by each questionnaire. Table 3 summarizes procedures adhered to by authors in the development and validation of an instrument. Below, we highlight key information pertaining to each instrument, including the procedures undertaken for item generation and item reduction, and findings about the following attributes of each questionnaire: reliability, validity, and responsiveness.
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Table 1 Evaluating the development and modification process of a patient-reported outcome measure Phase
Description
Input and psychometric properties
Ia
Identify concepts and develop conceptual framework
Ib
Create instrument
II
Field testing: assessment of measurement properties
Concepts from: Patient interviews Expert opinion Literature review Item generation Pre testing Acceptability Internal consistency Item total correlations Test–retest reliability Validity within scale
Validity comparison with other measures Validity hypothesis testing Responsiveness III
Definition
Data quality: assess completeness of data and score distributions The extent to which items within a scale measure the same construct The correlation of an item with the remainder of the scale The stability of a scale when administered on two separate occasions Evidence that a scale measures a single construct, items can be combined to create a summary score, and subscales measure distinct but related constructs The comparison of the scale to known and validated instruments or a “gold standard” The ability of the scale to differentiate between known groups The ability of the scale to detect clinically significant change
Modify instrument
Adapted from FDA Guidance for Industry Patient-Reported Outcome Measures [19] and Scientific Advisory Committee of the Medical Outcomes Trust [2]
Adolescent Quality of Life Instrument The Adolescent Quality of Life Instrument (AQoL) [20, 21] is a 16-item child-report measure of QOL for adolescents with cancer. Items were generated from established QOL instruments but did not involve child or parent input or expert opinion. Formal item reduction strategies were not described. In a population of 75 participants aged 9– 20 years, internal consistency reliability was reported for the total score (Cronbach’s α=0.77), but not for the six domain scores [20, 21]. No other psychometric analyses were reported.
Behavioral, Affective, and Somatic Experiences Scale The Behavioral, Affective, and Somatic Experiences Scale (BASES) [22, 46] is a measure of health-related QOL designed for the study of response to BMT. Parent- and nurse-report versions are 38 items in length, differing only in the use of lay versus technical language. A 14-item child report is available for use by children. All versions measure five domains and provide a total score. Item generation involved a review of existing instruments and input from BMT nurses but not patient or parent involvement. Item reduction involved discarding items with low incidence and redundancy [46]. Development of the child-report version
is not described. Psychometric analysis, performed using data from studies of 119 nurses and 45 children, showed internal consistency reliability (Cronbach’s α) ≥0.74 for four of the five nurse-report domains and for one childreport domain. The nurse form was able to discriminate between children undergoing different BMT procedures and by age and gender. All three versions showed sensitivity to change over time at the domain and individual item level [22].
Child Health Rating Inventories-Hematopoietic Stem Cell Transplantation The Child Health Rating Inventories-Hematopoietic Stem Cell Transplantation (CHRIs-HSCT) [23] is a measure of health-related QOL specific to children undergoing SCT. Parent-, adolescent-, and child-report versions each consist of 10 items that measure three domains. Item generation involved literature review and interviews with patients, parents, and health-care providers. Item reduction strategies were not described. Psychometric analysis with 122 children and 74 of their parents showed adequate internal consistency reliability in the two scales where this could be computed (Cronbach’s α>0.70). A subanalysis of internal consistency reliability for groups of children between 5– 7 years and 8–18 years found α≥0.70 on all but one
Patient group On or off treatment
Undergoing BMT
Undergoing SCT
Undergoing treatment
Undergoing treatment
Survivors
On or off treatment
On or off treatment
Brain tumor survivors
Measure
AQoL [20, 21]
BASES [22, 46]
CHRIs-HSCT [23]
ECVNO [24]
MPQOLQ [25]
MMQL [26–29]
PedsQL™ Cancer Module [30–36]
PedsQL™ Brain Tumor Module [37]
Peds-FACT-Brs [38]
Child; 5–7; Child; 8–12; Child; 13–18; Parent; 2–4; Parent; 5–7; Parent; 8–12; Parent; 13–18 Child; 7–12;
Child; 5–7; Child; 8–12; Child; 13–18; Parent; 2–4; Parent; 5–7; Parent; 8–12; Parent; 13–18
Child; 8–12; Child; 13–20
Parent; 1–18
34; 37;
23; 24; 24; 17; 23; 24; 24
26; 27; 27; 25; 26; 27; 27
28; 41
39
19
10
14; 38; 38
Child; ≥5; Parent; 2–20; Nurse; 2–20
Child; 5–12; Child; 13–18; Parent; 5–12; Parent; 13–18 Child; 6–18
16
Number of items
Child; 9–20
Versions; age range (years)
Table 2 Characteristics for each included pediatric oncology quality of life instrument
Additional concerns
Emotional function Desire to play with other children Lack of adult caregiving Social isolation Emotional stability Self-competence Social competence Body image Cognitive functioning Intimate relations Outlook on life Physical functioning/symptoms Psychological functioning Social functioning Cognitive problems Communication Nausea Pain and hurt Perceived physical appearance Procedural anxiety Treatment anxiety Worry Cognitive problems Movement and balance Nausea Pain and hurt Procedural anxiety Worry
Body Image Hassle Worry
Health status Mood Meaning of being ill Normal activities Social/family interactions Activity Compliance Mood disturbance Quality of interactions Somatic distress
Domains
U.S.
U.S.
U.S.
U.S./U.K.
U.S.
Spain
U.S.
U.S.
U.S.
Country of origin
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On or off treatment
Survivors
On or off treatment
On or off treatment
POQOLS [39, 40, 47]
PEDQOL® [41]
QOLCC [42–44]
RMH-PQLQ [45]
Parent; 2–19
Child; 7–12; Child; 13–18; Parent; 7–18
Child; 8–18
Child; 12–18; Parent; 7–12; Parent; 12–18 Parent; 5–17
78
34
34
21
34; 37
Emotional well-being and illness experience Physical well-being Social and family well-being Emotional distress Physical functioning and restriction from normal activity Response to current medical treatment Autonomy Body image Cognition Emotional functioning Physical functioning Social functioning/friends Social functioning/family Cognitive Communication Physical Psychosocial Social Treatment/disease Understanding Behavioral problems Cognitive functioning Emotional status Functional status Global health Global quality of life Physical symptoms Social functioning U.K.
Taiwan
Germany
U.S.
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• • • • • •
Item–total correlations Test–retest reliability Validity within scale Validity comparison with other measures Validity hypothesis testing Responsiveness
Item generation • Patient interviews • Parent interviews • Literature • Expert opinion • Developed conceptual model Item reduction • Expert opinion • Item redundancy • Endorsement frequencies • Missing data • Factor analysis • Testing of scales assumptions Psychometric analyses • Acceptability • Internal consistency
Method/evaluation
X
X
AQoL
X X
X X
X X
X X X X
CHRIsHSCT
X
X
X
X X X
X X
BASES
X
ECVNO
X
X X
X X
X
X
X X
MPQOLQ
Table 3 Pediatric oncology quality of life instruments: development and validation criteria
X X X X
X
X X
X
X X
MMQL
X X X
X
X X
X
X
X
X X X X X
PedsQL Cancer
X X X
X
X X X X
PedsQL Brain
X
X
X
X
X
X X
PedsFACT-Brs
X X X
X
X
X
X
X X X X
POQOLS
X
X
X
X
X X
PEDQOL
X X X
X
X X
X
X X X
QOLCC
X X
X
X
X
X X
RMH PQLQ
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domain (child 5–7 years; worry=0.69). For the parent form, hypothesized differences were observed in QOL of children with more recent SCTs versus ≥6 months after SCT, and the measure was able to discriminate between least and most severe groups (as rated by health-care providers).
Scale of Quality of Life for Children with Cancer The Scale of Quality of Life for Children with Cancer (ECVNO) [24] is a 19-item Spanish self-report measure of QOL for use in children undergoing treatment for cancer. No information is provided about item generation or reduction other than that the authors describe that they started with 28 items and eliminated nine that seemed less reliable. This instrument was administered to 43 children aged 3–18 years recruited from four different hospitals in Spain. A factor analysis identified four domains. Internal consistency reliability was adequate for the total score (Cronbach’s α=0.76) but was not reported for the domains.
Miami Pediatric Quality of Life Questionnaire: Parent Scale The Miami Pediatric Quality of Life Questionnaire: Parent Scale (MPQOLQ) [25] is a parent-completed measure of health-related QOL in children undergoing cancer treatment. Item generation involved qualitative interviews with 30 families of children being treated or within 1 year of completion of therapy for cancer. Item reduction involved having a pediatric oncologist, and ten parents review the instrument. The questionnaire was administered to 132 parents. Psychometric analysis showed acceptable internal consistency reliability (Cronbach’s α≥0.76) for total score and all three domain scores. The MPQOLQ demonstrated the ability to detect differences among children with different cancers and on different treatments.
Minneapolis–Manchester Quality of Life Instrument The Minneapolis–Manchester Quality of Life Instrument (MMQL) [26–29] assesses health-related QOL in childhood cancer survivors. Item generation involved input from children with cancer, parents, and health-care professionals. A 32-item questionnaire was administered to 643 children aged 8–12 years (162 with cancer) and a 46-item questionnaire to 397 aged 13–20 years (268 with cancer). Item reduction involved deleting items with low corrected item–total correlations and factor loadings
