Quality of Life Measurement for Children with Life-Threatening Conditions: Limitations and a New Framework

About 500,000 children are coping with life-threatening conditions (LTC) in the United States every year. Different service programs such as an integrated pediatric palliative care program may benefit health-related quality of life (HRQOL) which is a great concern of this children population and their families. However, evidence is limited about the appropriate HRQOL instruments for use. This study aims to validate psychometric properties of a generic HRQOL instrument, the Pediatric Quality of Life (PedsQL) 4.0, for children with LTC. The parent proxy-report was used. We conducted a telephone interview to collect data of 257 parents whose children had LTC and were enrolled in Medicaid. We used standard psychometric methods to validate the PedsQL: scale reliability, item-domain convergent/discriminant validity, and known-groups validity. We also conducted Rasch analysis to assess construct validity. Results suggest that the PedsQL did not demonstrate valid psychometric properties for measuring HRQOL in this population. Rasch analysis suggests that the contents of the items in all domains did not appropriately cover the latent HRQOL of children with LTC. We document several methodological challenges in using a generic instrument to measuring HRQOL and propose a new framework to improve HRQOL measures for children with LTC. The strategies include revising the content of existing items, designing new items, adding important themes (e.g., financial challenge), and applying computerized adaptive test to better select appropriate items for individual children with LTC.     

Reliability and validity of health status measurement by the TAPQOL.

BACKGROUND: In addition to clinical measures in the evaluation of paediatric interventions, health related quality of life (HRQoL) is an important outcome. The TAPQOL (TNO-AZL Preschool children Quality of Life) was developed to measure HRQoL in preschool children. It is a generic instrument consisting of 12 scales that cover the domains physical, social, cognitive, and emotional functioning. AIMS: To evaluate the feasibility, score distribution, internal consistency, test-retest reliability, and discriminative and concurrent validity of the TAPQOL multi-item scales in preschool children, aged 2-48 months. Also to evaluate the feasibility, reliability, and validity separately for infants (2-12 months old) and toddlers (12-48 months old). METHODS: Parents of a random general population sample of 500 preschool children were sent a questionnaire by mail. A random subgroup of 159 parents who participated received a retest after two weeks. RESULTS: The response rate was 83% at the test and 75% at the retest. There were few missing answers. Six scales showed ceiling effects. Nine scales had Cronbach's alphas >0.70. In general, score distributions and Cronbach's alphas were comparable for infants and toddlers. Test-retest showed no significant differences in mean scale scores; two scales had intra-class correlations <0.50. Five scales showed significant differences between children with no conditions versus children with two or more parent reported chronic conditions. CONCLUSION: Results showed that the TAPQOL is a feasible instrument to measure HRQoL and support the reliability and discriminative validity of the majority of its scales for infants as well as toddlers.     

Reliability and validity of health status measurement by the TAPQOL

Background: In addition to clinical measures in the evaluation of paediatric interventions, health related quality of life (HRQoL) is an important outcome. The TAPQOL (TNO-AZL Preschool children Quality of Life) was developed to measure HRQoL in preschool children. It is a generic instrument consisting of 12 scales that cover the domains physical, social, cognitive, and emotional functioning. Aims: To evaluate the feasibility, score distribution, internal consistency, test-retest reliability, and discriminative and concurrent validity of the TAPQOL multi-item scales in preschool children, aged 2–48 months. Also to evaluate the feasibility, reliability, and validity separately for infants (2–12 months old) and toddlers (12–48 months old). Methods: Parents of a random general population sample of 500 preschool children were sent a questionnaire by mail. A random subgroup of 159 parents who participated received a retest after two weeks. Results: The response rate was 83% at the test and 75% at the retest. There were few missing answers. Six scales showed ceiling effects. Nine scales had Cronbach''s alphas >0.70. In general, score distributions and Cronbach''s alphas were comparable for infants and toddlers. Test-retest showed no significant differences in mean scale scores; two scales had intra-class correlations <0.50. Five scales showed significant differences between children with no conditions versus children with two or more parent reported chronic conditions. Conclusion: Results showed that the TAPQOL is a feasible instrument to measure HRQoL and support the reliability and discriminative validity of the majority of its scales for infants as well as toddlers.     

The Minneapolis-Manchester Quality of Life instrument: Reliability and validity of the Youth Form

OBJECTIVE: Improvements in survival after childhood cancer have increased emphasis on health-related quality of life (HRQoL) of survivors. We developed the Minneapolis-Manchester Quality of Life-Youth Form (MMQL-YF) as a standardized patient self-report instrument designed to assess HRQoL in childhood cancer survivors between the ages of 8 and 12 years. STUDY DESIGN: To validate the instrument, the MMQL-YF was administered to 643 children (481 healthy, 162 with cancer). Factor analysis was conducted to refine the instrument, and Cronbach's alpha coefficient was used to measure its internal reliability. Known-groups validity was determined by comparing healthy children with those with cancer. Construct validity was studied by a comparison of similar domains in the MMQL-YF and the Child Health Questionnaire (CHQ). Stability was tested by re-administration of the MMQL-YF 2 weeks later. RESULTS: Internal consistency reliability was in the acceptable range for this instrument. The MMQL-YF was able to distinguish between known groups, and its scales correlated highly with similar CHQ domains. Test-retest reliability showed that the instrument was extremely stable in all scales tested. CONCLUSION: Data provide evidence for the validity and reliability of the MMQL-YF as a comprehensive, multidimensional, self-report instrument for measuring HRQoL among childhood cancer survivors.     

A comparison of pain scales in Thai children.

Three commonly used pain scales, the visual analogue scale, the Wong-Baker Faces Pain Scale, and the Faces Pain Scale Revised were administered to 122 Thai children, of whom half were HIV infected, in order to assess their validity. These scales presented moderate to good correlation and moderate agreement, sufficient for valid use in Thai children.     

A comparison of pain scales in Thai children

Three commonly used pain scales, the visual analogue scale, the Wong-Baker Faces Pain Scale, and the Faces Pain Scale Revised were administered to 122 Thai children, of whom half were HIV infected, in order to assess their validity. These scales presented moderate to good correlation and moderate agreement, sufficient for valid use in Thai children.     

Measuring quality of life in Japanese children: Development of the Japanese version of PedsQL

Background:  Health-related quality of life (HRQL) is perceived as an important health-care outcome. There are several systems for measuring the HRQL in adults but there are few such systems for children in Japan. Pediatric Quality of Life Inventory (PedsQL) is valid and demonstrates excellent reliability in the USA, Europe, and Asian countries. The aim of the present study was therefore to develop the Japanese version of PedsQL.
Methods:  A two-step procedure was performed: translation of PedsQL, followed by examination of the psychometric properties in a cross-sectional study. The feasibility, reproducibility, internal consistency reliability, factor structure, and concurrent and clinical validity were examined.
Results:  The internal consistency reliability of the Child Self-Reports of young children was slightly low, but that of the Child Self-Reports of school children and adolescents was good. Further, all the Parent Proxy-Reports had excellent alphas. The Japanese version had satisfactory feasibility for all age ranges. The intercorrelation of subscales supported the multidimensional factor structure. Clinical validity was examined by analysis of variance performed for four groups with different health conditions (healthy, chronic needs only, mental condition only, and chronic needs and mental condition). The scores of each functioning scale differed among the four groups, with the healthy group having the highest scores for all functioning scales.
Conclusions:  The Japanese version of PedsQL can be applied in community and school health settings in Japan. Because children with chronic health needs and mental conditions were included, the Japanese version of PedsQL is expected to be useful in clinical settings.     

Health-related quality of life of children and adolescents prior to hematopoietic progenitor cell transplantation: diagnosis and age effects

BACKGROUND: The health-related quality of life (HRQOL) may vary among children before undergoing hematopoietic progenitor cell transplantation (HPCT). This study examined the HRQOL of children scheduled for HPCT, the effects of diagnosis and age on HRQOL, and the convergent validity of one generic and two disease-specific measures of HRQOL. PROCEDURE: The sample consisted of 111 children (mean age = 10.4 years) diagnosed with acute lymphoblastic leukemia (ALL; 22%), other leukemias (26%), neuroblastoma (19%), other solid tumors (18%), and hematologic disorders (15%). Convergence validity was tested with 67 children (mean age = 10.3 years) who had an equivalent distribution of diagnoses except for neuroblastoma (12%). The Child Health Questionnaire (CHQ), a generic measure, and the Pediatric Oncology Quality of Life Scale (POQOL) and the Play Performance Scale (PPS), disease-specific measures, were completed by one parent prior to HPCT. RESULTS: Compared to the norms for healthy children, the CHQ Physical summary scores for every diagnostic subgroup and the CHQ Psychosocial summary scores for ALL were poorer. Compared to the cancer norms for Total POQOL and PPS scores, scores for ALL and neuroblastoma were the poorest. These measures also revealed that adolescents' HRQOL was perceived to be worse than children's. Total POQOL scores showed strong convergent validity with CHQ Physical and Psychosocial scores and moderate convergent validity with the PPS scores. CONCLUSIONS: Based on parental reports, children treated for ALL and neuroblastoma appear to be at the greatest risk for poor HRQOL before undergoing HPCT, and adolescents seem to be more compromised than younger children, based on parental reports. The POQOL measure seems to be the best predictor of HRQOL. These results have clinical implications for the care of children undergoing HPCT.     

The reliability of the Health Related Quality Of Life questionnaire PedsQL 3.0 Diabetes Module™ for Swedish children with type 1 diabetes

Aim: The overall aim of the study was to assess reliability and accomplish a limited validation of the Pediatric Quality of Life Inventory 3.0 Diabetes Module Scales (PedsQL 3.0), Swedish version in a sample of Swedish children diagnosed with Type 1 diabetes (T1DM). A secondary aim was to assess whether the children’s Health Related Quality of Life (HRQOL) was associated with children’s gender and age and whether the child self‐ and parent proxy reports were consistent. Methods: One hundred and thirty families from four diabetes centres participated in this study. The Pediatric Quality of Life Inventory 4.0 Generic Core Scales (PedsQL 4.0) and the PedsQL 3.0 were administered to 108 children (aged 5–18 years) with T1DM and 130 parents (of children with T1DM aged 2–18 years). Results: The internal consistency of the PedsQL 3.0, Swedish version, reached or exceeded Cronbach’s alpha values of 0.70 for both child self‐ and proxy reports‐ and parent proxy‐reports. The PedsQL 4.0 and PedsQL 3.0 were highly correlated (r = 0.76), indicating convergent validity. The parents reported lower diabetes‐specific HRQOL than the children themselves (p < 0.01). The girls in the study reported lower psychological functioning and treatment adherence compared with the boys (p < 0.05). The oldest children (between 13 and 18 years of age) reported significantly lower diabetes‐specific HRQOL, as compared with younger children (p < 0.05). Conclusions: PedsQL 3.0 Diabetes Module can be used as a valuable tool for measuring diabetes‐specific HRQOL in child populations, both in research and in clinical practice.     

The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity.

BACKGROUND: The application of health-related quality of life (HRQOL) as a pediatric population health measure may facilitate risk assessment and resource allocation, the tracking of community health, the identification of health disparities, and the determination of health outcomes from interventions and policy decisions. OBJECTIVE: To determine the feasibility, reliability, and validity of the 23-item PedsQL 4.0 (Pediatric Quality of Life Inventory) Generic Core Scales as a measure of pediatric population health for children and adolescents. DESIGN: Mail survey in February and March 2001 to 20 031 families with children ages 2-16 years throughout the State of California encompassing all new enrollees in the State's Children's Health Insurance Program (SCHIP) for those months and targeted language groups. METHODS: The PedsQL 4.0 Generic Core Scales (Physical, Emotional, Social, School Functioning) were completed by 10 241 families through a statewide mail survey to evaluate the HRQOL of new enrollees in SCHIP. RESULTS: The PedsQL 4.0 evidenced minimal missing responses, achieved excellent reliability for the Total Scale Score (alpha =.89 child;.92 parent report), and distinguished between healthy children and children with chronic health conditions. The PedsQL 4.0 was also related to indicators of health care access, days missed from school, days sick in bed or too ill to play, and days needing care. CONCLUSION: The results demonstrate the feasibility, reliability, and validity of the PedsQL 4.0 as a pediatric population health outcome. Measuring pediatric HRQOL may be a way to evaluate the health outcomes of SCHIP.     

Health related quality of life in disorders of defecation: the Defecation Disorder List.

BACKGROUND: Constipation and encopresis frequently cause problems with respect to emotional wellbeing, and social and family life. Instruments to measure Health Related Quality of Life (HRQoL) in these disorders are not available. METHODS: A disease specific HRQoL instrument, the "Defecation Disorder List" (DDL) for children with constipation or functional non-retentive faecal soiling (FNRFS) was developed using accepted guidelines. For each phase of the process, different samples of patients were used. The final phase of development included 27 children. Reliability was assessed in two ways: internal consistency of domains with Cronbach's alpha, and test-retest reliability with intra-class correlation coefficients (ICC). To assess validity, comparable items and domains were correlated with Tacqol, a generic HRQoL instrument for children (TNO-AZL). RESULTS: In the final phase of the development, 27 children completed the instrument. It consisted of 37 items in four domains. The response rate was 96%. Reliability was good for all domains, with Cronbach's alpha values ranging from 0.61 to 0.76. Measures of test-retest stability were good for all four domains with ICCs ranging from 0.82 to 0.92. Validity based on comparison with the Tacqol instrument was moderate. CONCLUSION: The DDL is promising as a measure of HRQoL in childhood defecation disorders.     

Pediatric asthma quality of life questionnaire (PAQLQ): Validation among asthmatic children in Thailand

Quality of life (QoL) is an important consideration among asthma sufferers. The Pediatric Asthma Quality of Life Questionnaire (PAQLQ) is one of the most widely used instruments for measuring health-related QoL in children with asthma. The standardized version of PAQLQ contains 23 questions in three domains, i.e., activity limitation, symptoms and emotional function. The objective of this study was to validate the Thai-translated version of the PAQLQ. The study design consisted of a five-week single cohort study. Patients recorded symptoms, and peak expiratory flow rate (PEFR) each morning and evening during the first and fifth week of the study in asthma diary. At each clinic visit, a trained-interviewer administered the PAQLQ and performed spirometric measurements. Fifty-one children, ages between 7 and 17 yr participated in the study. Scores from the asthma diary were used to classify patients into stable vs. unstable groups. The construct validity of the questionnaire was confirmed in both cross-sectional and longitudinal studies by demonstrating correlations between various PAQLQ domains with clinical asthma parameters (asthma diary, beta-agonist use and PEFR). There was high internal consistency for scores of the three domains (Cronbach's alpha-coefficient = 0.83-0.95). For those with stable asthma, the reliability of PAQLQ was good for the rating scale (intra-class correlation coefficient--ICC = 0.84) and for total score (alpha = 0.97) indicating high reproducibility of the PAQLQ. The significant difference of changes QoL scores between stable and unstable groups was observed in all domains. We conclude that the Thai version of PAQLQ is valid and reliable for implementing in Thai children with asthma.     

Health related quality of life in disorders of defecation: the Defecation Disorder List

Background: Constipation and encopresis frequently cause problems with respect to emotional wellbeing, and social and family life. Instruments to measure Health Related Quality of Life (HRQoL) in these disorders are not available. Methods: A disease specific HRQoL instrument, the "Defecation Disorder List" (DDL) for children with constipation or functional non-retentive faecal soiling (FNRFS) was developed using accepted guidelines. For each phase of the process, different samples of patients were used. The final phase of development included 27 children. Reliability was assessed in two ways: internal consistency of domains with Cronbach''s alpha, and test-retest reliability with intra-class correlation coefficients (ICC). To assess validity, comparable items and domains were correlated with Tacqol, a generic HRQoL instrument for children (TNO-AZL). Results: In the final phase of the development, 27 children completed the instrument. It consisted of 37 items in four domains. The response rate was 96%. Reliability was good for all domains, with Cronbach''s alpha values ranging from 0.61 to 0.76. Measures of test-retest stability were good for all four domains with ICCs ranging from 0.82 to 0.92. Validity based on comparison with the Tacqol instrument was moderate. Conclusion: The DDL is promising as a measure of HRQoL in childhood defecation disorders.     

Adaptation of the Manchester-Minneapolis Quality of Life instrument for use in the UK population.

INTRODUCTION: The availability of health-related quality of life (HRQL) measures that are reliable, valid, brief and comprehensible and appropriate for use with UK children is limited. We report the validation of a HRQL measure suitable for UK use in healthy children, children with chronic disease conditions and socially disadvantaged children. PATIENTS: A total of 1238 children took part in the study, including healthy children as controls (n = 824) and five exemplar groups: children diagnosed with asthma (n = 87), diabetes (n = 103) or inflammatory bowel disease (IBD; n = 69), children in remission from cancer (n = 68) and children in public care (n = 87). METHODS: In phase I, the Manchester-Minneapolis Quality of Life instrument (MMQL) Child Form was translated into UK English. In phases II and III, the questionnaire was shortened and validated. RESULTS: MMQL was anglicised and shortened to five components comprising 29 items. Good internal reliability was found with alpha reaching at least 0.69 for all subscales. Construct validity was established through moderate correlations with comparable PedsQL subscales (Pearson's r ranged from 0.38 to 0.58, p<0.01). Discriminant validity was also demonstrated in children with asthma and IBD, children in remission from cancer and children in public care, all of whom reported significantly lower HRQL than healthy children. Children with diabetes showed similar HRQL to their healthy peers. Good reproducibility and moderate responsiveness were demonstrated for the new measure. CONCLUSIONS: The anglicised and shortened MMQL was shown to be valid and reliable and could be a valuable new tool for the assessment of HRQL in children.     

Pediatric Functional Constipation Questionnaire-Parent Report (PedFCQuest-PR): development and validation

ObjectiveDeveloping and validating a disease-specific instrument in the Brazilian Portuguese language to assess the Health-Related Quality of Life of children with functional constipation, applied to parents/caregivers.MethodsThe process of developing the questionnaire was carried out in the following steps: items generation concerning functional constipation; elaboration of the preliminary questionnaire; assessment by health professionals; identifying problems or inconsistencies by the researchers; improvement of the questions; obtaining a final questionnaire named Pediatric Functional Constipation Questionnaire-Parent Form (PedFCQuest-PR) with 26 questions divided into four domains. Responses options use a Likert scale based on the events of the last four weeks. The process of validation was an observational, cross-sectional study in a sample of 87 parents/caregivers of children from 5 to 15 years of age diagnosed with Functional constipation according to the Rome IV Criteria. The questionnaire was applied simultaneously to the Pediatric Quality of Life Inventory 4.0 (PedsQL TM 4.0) as a control.ResultsThe questionnaire validation included 87 parents/caregivers. The children's median age was 8.2 years, with a long time of constipation symptoms associated with fecal incontinence in approximately two-thirds. Internal consistency reliability for the Total Scale Score of PedFCQuest-PR by Coefficient Alpha of Cronbach score was 0.86. Convergent and divergent validity of PedFCQuest-PR was demonstrated by correlating the domains of both questionnaires.ConclusionThis study provides evidence that PedFCQuest-PR is a reliable instrument. The results showed a high degree of internal consistency and validity of the instrument for future applications.     

Psychometric properties of the Swedish PedsQL, Pediatric Quality of Life Inventory 4.0 generic core scales

Aim:  To study the psychometric performance of the Swedish version of the Pediatric Quality of Life Inventory (PedsQL) 4.0 generic core scales in a general child population in Sweden.
Methods:  PedsQL forms were distributed to 2403 schoolchildren and 888 parents in two different school settings. Reliability and validity was studied for self-reports and proxy reports, full forms and short forms. Confirmatory factor analysis tested the factor structure and multigroup confirmatory factor analysis tested measurement invariance between boys and girls.
Results:  Test-retest reliability was demonstrated for all scales and internal consistency reliability was shown with α value exceeding 0.70 for all scales but one (self-report short form: social functioning). Child-parent agreement was low to moderate. The four-factor structure of the PedsQL and factorial invariance across sex subgroups were confirmed for the self-report forms and for the proxy short form, while model fit indices suggested improvement of several proxy full-form scales.
Conclusion:  The Swedish PedsQL 4.0 generic core scales are a reliable and valid tool for health-related quality of life (HRQoL) assessment in Swedish child populations. The proxy full form, however, should be used with caution. The study also support continued use of the PedsQL as a four-factor model, capable of revealing meaningful HRQoL differences between boys and girls.     

中文版儿童生命质量哮喘特异性量表的信度和效度评价

目的：评价简体中文版儿童生命质量（PedsQLTM）哮喘特异性量表家长报告的信度和效度。方法：采用 PedsQLTM 哮喘特异性量表家长报告量表及自设一般情况问卷,对重庆医科大学附属儿童医院哮喘中心就诊的 233 例哮喘患儿及其家长进行问卷调查。用克朗巴赫α系数考核其信度,分别用探索性因子分析、相关分析考核其效度。结果：中文版 PedsQLTM 哮喘特异性量表家长报告全量表、症状相关问题、治疗相关问题、担心相关问题、沟通相关问题的α系数分别为0.86、0.80、0.78、0.89和0.93,表明量表有较好的内部一致性。通过因子分析共提取7个公因子,与量表基本结构一致,主成分累积贡献率接近 66%。4个维度得分与所含条目得分间均有较强的相关关系(r=0.41~0.92, P＜0.01)。结论：中文版PedsQLTM 哮喘特异性量表家长报告有良好的信度和效度,与原语言版本一致,可适用于中国哮喘患儿健康相关生命质量评价。［中国当代儿科杂志,2010,12(12)：943-946］     

The performance of the PedsQL generic core scales in children with sickle cell disease

The objective of this study was to determine the feasibility, reliability, and validity of the Pediatric Quality of Life Inventory generic core scales (PedsQL questionnaire) in children with sickle cell disease. This was a cross-sectional study of children from an urban hospital-based sickle cell disease clinic and an urban primary care clinic. The study participants were children of ages 2 to 18 years who presented to clinic for a routine visit. Health-related quality of life (HRQL) was the main outcome. HRQL of children with sickle cell disease were compared with children without disease to test validity. Missing items were used to determine feasibility and Cronbach's alpha was used to determine reliability. Parents of 178 children (104 with sickle cell disease and 74 without disease) and 118 children (78 with sickle cell disease and 40 without disease) completed HRQL questionnaires. The PedsQL questionnaire was feasible and reliable. The parent proxy and child self-report questionnaire differentiated between children with and without sickle cell disease. The parent proxy-report differentiated well between children with mild and severe sickle cell disease. The questionnaire performed well in children with sickle cell disease and is a feasible, reliable, and valid tool to measure HRQL in children with sickle cell disease.     

Family Quality of Life Survey (FQOLS-2006): Evaluation of Internal Consistency,Construct, and Criterion Validity for Socioeconomically Disadvantaged Families

Aims: The purpose of this study was to test the psychometric properties of the Family Quality of Life Survey (FQOLS-2006) when used with urban families predominantly from socioeconomically disadvantaged backgrounds. Methods: Data gathered from 193 family caregivers using the FQOLS-2006 were subjected to reliability analyses, confirmatory factor analyses, and correlational analyses to test the internal consistency of the scales (reliability), factor structure of the scales (construct validity), and convergence between the long and short versions of the tool (criterion validity). Results: Internal consistency of the 54-item total FQOL scale was excellent (α = .89), while that of the six-item domain subscales ranged from moderate to strong (α = .46–.81). Although the subscale-level FQOL factor structure demonstrated good fit, some of the item-level factor loadings within each of the domains were low. Correlations between scores derived from the long and short versions ranged from moderate to strong (r = .37–.73). Conclusions: Although the internal consistency of the scales ranged from moderate to strong, the FQOLS-2006 had only a moderate degree of construct and criterion validity when used with a sample consisting predominantly of minorities from socioeconomically disadvantaged backgrounds.     

Swedish translation and validation of the Pediatric Allergic Disease Quality of Life Questionnaire (PADQLQ)

Aim: The aim of the study was to translate and validate the PADQLQ (Pediatric Allergic Disease Quality of Life Questionnaire), a disease‐specific quality of life questionnaire for the assessment of quality of life in children with pollen allergy. Methods: The PADQLQ was translated into Swedish according to guidelines. Children aged 7–18 with grass pollen allergy were included. Quality of life was assessed in parallel with ordinary symptom scales (VAS) before, during and after the pollen season. Results: A total of 98 children were included and 89 (91%) completed the study. The results for PADQLQ showed good cross‐sectional and longitudinal validity. The retrospective estimation after the season showed good consensus with the assessment during pollen season. Conclusion: Quality of life in children assessed with the PADQLQ (Pediatric Allergic Disease Quality of Life Questionnaire) is a reliable strategy for evaluating the burden of disease in children with pollen allergy and for the evaluation of treatment.