AIM: To evaluate the factors involved in the impairment of health-related quality of life (HRQOL) in patients with celiac disease.
METHODS: A multicenter, cross-sectional prospective study was performed in patients with celiac disease who completed two HRQOL questionnaires: the gastrointestinal quality of life index (GIQLI) and the EuroQol-5D (EQ).
RESULTS: Three hundred and forty patients (163 controlled with a gluten-free diet, and 177 newly diagnosed with a normal diet) were included. The GIQLI score was significantly better in patients on a gluten-free diet (GFD) than in non-treated patients on their usual diet, both in terms of the overall score (3.3 vs 2.7, respectively; P < 0.001), as well as on the individual questionnaire dimensions. Both the preference value of the EQ as the visual analogue scale were significantly better in treated than in non-treated patients (0.93 vs 0.72, P < 0.001 and 80 vs 70, P < 0.001, respectively). Variables significantly associated with a worse HRQOL score were female gender, failure to adhere to a GFD, and symptomatic status.
CONCLUSION: In untreated celiac disease, the most important factors that influence patient perception of health are the presence of symptoms and a normal diet. HRQOL improves to levels similar to those described in the general population in celiac disease patients well controlled with a GFD.
Celiac disease; Health status; Quality of life; Gluten-free diet
Celiac Disease (CD) is a chronic autoimmune disease triggered by dietary gluten. Gluten avoidance, which is the only available treatment for CD, could impact on quality of life of children with CD. We present the results of a qualitative study on the emotional impact of gluten free diet (GFD) on the everyday life of children affected with CD. We investigated 76 celiac patients aged 2–18 years (average age: 9.5 years). By using the Critical Incident Technique (CIT), we defined emotions related to difficulties and awkward situations experienced by the patients. Written answers to open-ended questions from either children (older than 8 years) and parents (children younger than 8 years) were analyzed qualitatively. We found 80 dilemmas experienced in three different arenas (food situations at school, meals at home, meals outside) and characterized lived experiences of children with CD in everyday life (specific emotions, difficulties in relationships and in management of daily life). Children with CD experience strong emotions related to the GFD, permeating several aspects of everyday life. These dilemmas may be missed by a conventional, questionnaire-based approach to the psycho-social consequences of CD treatment.
quality of life; celiac disease; gluten free diet; children; lived experiences; psycho-social aspects
International guidelines on type 1 diabetes advocate routine screening of health-related quality of life (HRQOL). DISABKIDS questionnaires are the first instruments developed across cultures and nations to provide age-appropriate measures of HRQOL in children with chronic diseases. DISABKIDS includes a Chronic Generic Module 37 (DCGM-37) and disease-specific modules. The purpose of this study was to examine reliability and validity of the Norwegian versions of the DISABKIDS questionnaires in children and adolescents with type 1 diabetes.
The DCGM-37 and the Diabetes Specific Module-10 (DDM-10) were translated into Norwegian using standard forward-backward translation. Eight to 19 year old children and adolescents with type 1 diabetes scheduled for routine follow-up at three diabetic clinics in Norway and one of their parents were invited to complete the DCGM-37 and the DDM-10. Internal consistency was determined using Cronbach's alpha. Results were compared with those of the Child Health Questionnaire Children Form-87 (CHQ-CF87) and Child Health Questionnaire Parent Form-50 which are established generic questionnaires. DISABKIDS results were related to age, gender, duration of diabetes, mode of insulin delivery and metabolic control. Clinical data were obtained from the Norwegian Childhood Diabetes Registry.
Of 198 eligible child-parent dyads, 103 (52%) completed the questionnaires. Mean age was 13.6 (2.6), range 8-19 yrs, 52% were boys. Cronbach's alpha was > 0.70 for all the DISABKIDS sub-scales except two (physical ability and social inclusion). There were moderate to high correlations (0.65-0.81) between the DISABKIDS scales and mental/emotional sub-scales of CHQ-CF87. Increasing age and higher HbA1c were significantly associated with reduced HRQOL scores. Parents tended to score their child's HRQOL lower than the children/adolescents themselves.
The study shows that the DISABKIDS instruments are applicable to a Norwegian childhood diabetes population. They seem to be a relevant supplement to other clinical indicators in medical practice and research.
Health-related quality of life; Type 1 diabetes; Children; Adolescents; Psychometrics; Reliability; Validity; DISABKIDS
Celiac disease (CD) is a chronic disorder in genetically predisposed individuals in which a small intestinal immune-mediated enteropathy is precipitated by dietary gluten. It can be difficult to diagnose because signs and symptoms may be absent, subtle, or not recognized as CD related and therefore not prompt testing within routine clinical practice. Thus, most people with CD are undiagnosed and a public health intervention, which involves screening the general population, is an option to find those with unrecognized CD. However, how these screening-detected individuals experience the diagnosis and treatment (gluten-free diet) is not fully understood. The aim of this study is to investigate the health-related quality of life (HRQoL) of adolescents with screening-detected CD before and one year after diagnosis and treatment.
A prospective nested case-referent study was done involving Swedish adolescents who had participated in a CD screening study when they were in the sixth grade and about 12 years old. Screening-detected adolescents (n = 103) and referents without CD who participated in the same screening (n = 483) answered questionnaires at the time of the screening and approximately one year after the screening-detected adolescents had received their diagnosis that included the EQ-5D instrument used to measure health status and report HRQoL.
The HRQoL for the adolescents with screening-detected CD is similar to the referents, both before and one year after diagnosis and initiation of the gluten-free diet, except in the dimension of pain at follow-up. In the pain dimension at follow-up, fewer cases reported problems than referents (12.6% and 21.9% respectively, Adjusted OR 0.50, 95% CI 0.27-0.94). However, a sex stratified analysis revealed that the significant difference was for boys at follow-up, where fewer screening-detected boys reported problems (4.3%) compared to referent boys (18.8%) (Adjusted OR 0.17, 95% CI 0.04-0.73).
The findings of this study suggest that adolescents with unrecognized CD experience similar HRQoL as their peers without CD, both before and one year after diagnosis and initiation of gluten-free diet, except for boys in the dimension of pain at follow-up.
Adolescents; Celiac disease; EQ-5D; Health-related quality of life; Screening; Screening-detected celiac disease
Celiac Disease (CD) is an increasingly common autoimmune disorder. It requires a strict lifelong adherence to a gluten-free diet (GFD) which can influence health-related quality of life (HRQOL). This study assesses HRQOL in children and adolescents with CD and explores how several demographic and clinical characteristics and GFD adherence affect their perceived health status.
We recruited 140 consecutive children and adolescents with CD confirmed by small bowel biopsy. HRQOL was assessed using the SF-12 questionnaire plus some CD-specific questions exploring wellbeing and lifestyle. Patients, aged 10 to 18 years, were identified by pediatric gastroenterologists and guided in filling out the questionnaire by trained psychologists. Parametric or non-parametric tests were applied to analyze continuous variables and frequencies as appropriate.
The SF-12 mean mental component summary score (MCS12) was lower than in the general Italian population (p < 0.001), whereas differences in terms of physical health were not significant (p = 0.220). More than one third of those interviewed reported feeling angry “always” or “most of the time” about having to follow the GFD, and nearly 20% reported feeling different from others and misunderstood because of CD “always” or “most of the time”.
Our findings highlight the need for health professionals to identify adolescents with major disease-related problems. The food industry should improve its range of gluten-free food products and public bodies and institutions should promote informative campaigns and help promote the overall quality of life of children and adolescents with CD.
Background & Aims
Celiac crisis is a life-threatening syndrome in which patients with celiac disease have profuse diarrhea and severe metabolic disturbances. Celiac crisis is rare among adults and not well documented. To improve awareness of this condition and to facilitate diagnosis, we reviewed cases of celiac crisis to identify presenting features, formulate diagnostic criteria, and develop treatment strategies.
Cases of biopsy-proven celiac disease were reviewed. Celiac crisis was defined as acute onset or rapid progression of gastrointestinal symptoms that could be attributed to celiac disease and required hospitalization and/or parenteral nutrition, along with signs or symptoms of dehydration or malnutrition.
Twelve patients met preset criteria for celiac crisis; 11 developed celiac crisis before they were diagnosed with celiac disease. Eleven patients had increased titres of tTG and 1 had immunoglobulin A deficiency. Results of biopsy analyses of duodenum samples from all patients were consistent with a Marsh 3 score (33% with total villous atrophy). Patients presented with severe dehydration, renal dysfunction, and electrolyte disturbances. All patients required hospitalization and intravenous fluids, 6 required corticosteroids, and 5 required parenteral nutrition. All patients eventually had a full response to a gluten-free diet.
Celiac crisis has a high morbidity and, although rarely described, occurs in adults and often has a clear precipitating factor. Patients that present with severe unexplained diarrhea and malabsorption should be tested for celiac disease; treatment with systemic steroids or oral budesonide should be considered. Nutritional support is often required in the short term but most patients ultimately respond to gluten avoidance.
steroids; treatment; tissue transglutaminase; enteropathy
A gluten-free camp allows children with celiac disease (CD) to enjoy a camp experience without concern and preoccupation with foods they eat or the stigma of their underlying disease. The objective of this study was to evaluate the impact of gluten-free camp on quality-of-life indicators for children and adolescents with CD.
Children aged 7 to 17 years with CD were administered a 14-question survey at the beginning and the end of a 7-day gluten-free camp. Surveys used a Likert scale to examine general well-being, emotional outlook, and self-perception for the week before each survey. Differences between the time points were compared. Data were analyzed by paired t test.
Of the 104 campers who attended camp, 77 (21 male) completed the survey at both time points. Most (70%) had been on a glutenfree diet (GFD) for <4 years. All seemed to benefit from camp, no longer feeling different from other kids or feeling frustrated with a restricted diet. A more beneficial impact was found for campers who were on a GFD for <4 years. Overall, campers reported an improvement in 11 of 14 questions, statistically significant (P <.05) for 8 of those 11 questions. Improvement was observed in each of the 3 categories of questions: well-being, self-perception, and emotional outlook.
Children who had CD and attended a week-long glutenfree camp demonstrated improvement in well-being, self-perception, and emotional outlook. The positive effects of camp were more apparent among campers who had been on a GFD for <4 years compared with those who had been on a GFD for ≥4 years, suggesting an adaptation to CD with time. A gluten-free camp that provides an environment of unrestricted foods can at least temporarily alleviate stress and anxiety around food and social interactions. Durability of these observations on return to daily life requires additional study.
pediatric; sprue; quality of life
Celiac disease is one of the malabsorption syndromes leads to growth and development retardation in children. There is no test lonely can definitely diagnose celiac; however, the collection of clinical findings, serologic tests, intestinal biopsy, and response to treatment may diagnose it. Although diagnostic value is variable in different studies, they are used a non-invasive and appropriate screening methods today. This study aimed to evaluate diagnostic value of celiac serologic tests in children less than 15-year-old.
During two years, this study conducted on children referred to Al-Zahra hospital (Isfahan, Iran). All the children who had duodenal biopsy tests were evaluated in terms of serologic tests and clinical symptoms due to suspected celiac. The results were analyzed through descriptive statistics, chi-square and Fisher's exact tests using SPSS software.
15.8 percent of children were under 2 years, 37.3 percent between age range of 2 to 12 years and 10.5 percent were above 12 years. 8.1 percent of children with negative anti-endomysial antibody (EMA) suffered from celiac; while 20.0 percent of children with positive EMA suffered from celiac. 15.4 percent of children with negative anti-gliadin antibody (AGA) had celiac; while 11.6 percent of those with positive AGA suffered from it. 11.1 percent of those with negative tissue transglutaminase antibody (tTG) and 37.5 percent with positive tTG suffered from celiac.
According to our study results, there is no correlation between gastrointestinal symptoms such as vomiting diarrhea, anorexia, bulimia, and failure to thrive (FFT) with celiac. TTG was the best screening test method to diagnose celiac disease and other tests such as AGA and EMA do not have high diagnostic value.
Celiac; Malabsorption; Anti-gliadin antibody; Anti-endomysial antibody; Tissue transglutaminase antibody
Hydroxyurea is a safe and efficacious medication for children with sickle cell disease (SCD). Our objective was to compare health related quality of life (HRQL) between children taking hydroxyurea and those not taking hydroxyurea. We conducted a retrospective cohort study of children with SCD who had completed the PedsQL 4.0 at Duke University Medical Center or the Midwest Sickle Cell Center. Our primary outcome was HRQL in children receiving hydroxyurea therapy compared to those not receiving hydroxyurea. One hundred ninety-one children with SCD were included in the study. Children in the hydroxyurea group had higher self-report Total Peds QL median scores than children in the no hydroxyurea group; p=0.04. Child self-report physical functioning scores were significantly higher for children in the hydroxyurea group; p=0.01. In conclusion, children with SCD who are receiving hydroxyurea therapy report better overall HRQL and better physical HRQL than children not receiving this therapy despite disease severity. Further research assessing the impact of hydroxyurea therapy on HRQL, such as prospective assessment over time, would aid in our understanding of the effectiveness of hydroxyurea for individual children. Ultimately, this may aid in decreasing the barriers to the use of hydroxyurea.
sickle cell disease; hydroxyurea; health related quality of life
The European DISABKIDS project aims to enhance the Health Related Quality of Life (HRQoL) of children and adolescents with chronic medical conditions and their families. We describe the development of the seven cross-nationally tested condition-specific modules of the European DISABKIDS HRQoL instrument in a population of children and adolescents. The condition-specific modules are intended for use in conjunction with the DISABKIDS chronic generic module.
Focus groups were used to construct the pilot version of the DISABKIDS condition-specific HRQoL modules for asthma, juvenile idiopathic arthritis, atopic dermatitis, cerebral palsy, cystic fibrosis, diabetes and epilepsy. Analyses were conducted on pilot test data in order to construct field test versions of the modules. A series of factor analyses were run, first, to determine potential structures for each condition-specific module, and, secondly, to select a reduced number of items from the pilot test to be included in the field test. Post-field test analyses were conducted to retest the domain structure for the final DISABKIDS condition-specific modules.
The DISABKIDS condition-specific modules were tested in a pilot study of 360 respondents, and subsequently in a field test of 1152 respondents in 7 European countries. The final condition-specific modules consist of an 'Impact' domain and an additional domain (e.g. worry, stigma, treatment) with between 10 to 12 items in total. The Cronbach's alpha of the final domains was found to vary from 0.71 to 0.90.
The condition-specific modules of the DISABKIDS instrument were developed through a step-by-step process including cognitive interview, clinical expertise, factor analysis, correlations and internal consistency. A cross-national pilot and field test were necessary to collect these data. In general, the internal consistency of the domains was satisfactory to high. In future, the DISABKIDS instrument may serve as a useful tool with which to assess HRQoL in children and adolescents with a chronic condition. The condition-specific modules can be used in conjunction with the DISABKIDS chronic generic module.
Asthma quality of life questionnaires are not readily incorporated into clinical care. We therefore computerised the Paediatric Asthma Quality of Life Questionnaire (standardised) (PAQLQ(S)) and the Paediatric Asthma Caregivers Quality of Life Questionnaire (PACQLQ), with a colour‐coded printed graphical report.
To (a) assess the feasibility of the electronic questionnaires in clinical care and (b) compare the child's PAQLQ scores with the parent's score, physician's clinical score and spirometry.
Children with asthma were given a clinical severity score of 1–4 (increasing severity) and then completed the PAQLQ(S) electronically (scores 1–7 for increasing quality of life in emotional, symptoms and activity limitation domains) followed by spirometry and physician review. Parents completed the PACQLQ. Inclusion criteria required fluent Hebrew and reliable performance of spirometry. Children with additional chronic diseases were excluded.
147 children with asthma aged 7–17 years completed PAQLQs and 115 accompanying parents completed PACQLQs, taking 8.3 (4.3–15) and 4.4 (1.5–12.7) min, respectively (mean (range)). Graphical reports enabled physicians to address quality of life during even brief visits. Children's (PAQLQ) and parents' (PACQLQ) total scores correlated (r = 0.61, p<0.001), although the children's median emotional score of 6.3 was higher than their parents' 5.7 (p<0.001), whereas median activity limitation score was lower than their parents': 5.0 and 6.8, respectively (p<0.001). No correlation was found with physician's clinical score or spirometry.
Electronic PAQLQs are easy to use, providing additional insight to spirometry and physician's assessment, in routine asthma care. Future studies must assess impact on asthma management.
asthma; quality of life; questionnaire; electronic; computerised
A decade ago celiac disease was considered extremely rare outside Europe and, therefore, was almost completely ignored by health care professionals. In only 10 years, key milestones have moved celiac disease from obscurity into the popular spotlight worldwide. Now we are observing another interesting phenomenon that is generating great confusion among health care professionals. The number of individuals embracing a gluten-free diet (GFD) appears much higher than the projected number of celiac disease patients, fueling a global market of gluten-free products approaching $2.5 billion (US) in global sales in 2010. This trend is supported by the notion that, along with celiac disease, other conditions related to the ingestion of gluten have emerged as health care concerns. This review will summarize our current knowledge about the three main forms of gluten reactions: allergic (wheat allergy), autoimmune (celiac disease, dermatitis herpetiformis and gluten ataxia) and possibly immune-mediated (gluten sensitivity), and also outline pathogenic, clinical and epidemiological differences and propose new nomenclature and classifications.
To assess health-related quality of life (HRQOL) in children and adolescents with sickle cell disease (SCD).
Design, Setting, and Participants
The PedsQL™ 4.0 Generic Scales, a multidimensional self-report instrument that has been shown to be valid and reliable for use in children and adolescents with chronic illness, consists of 23 items that assess physical, emotional, social, and school functioning. Questionnaires were administered to 124 children and adolescents (aged 8-18 years, child self-report) with SCD (100 sickle cell anemia [SS], 24 sickle beta zero thalassemia [Sβ0thal]) and their parents (parent-proxy report). Summary scores for children’s and parents’ ratings of overall HRQOL and psychosocial health and subscale scores for physical, emotional, social, and school functioning were compared to published data for healthy children. Both summary and subscale scores for children with SCD were also compared to those of their parents.
Children with SCD and their parents rated overall HRQOL and all sub-domains of HRQOL lower than ratings of healthy children and their parents (p < .001). Children with SCD rated their own HRQOL significantly better than their parents for overall HRQOL and all sub-domains (p <.001) except emotional functioning (p = .06).
Children with SCD and their parents perceived overall HRQOL and all HRQOL sub-domains to be lower than scores reported in healthy children. Therefore, successful therapeutic efforts to improve HRQOL could represent important advances in the health of children with SCD.
Quality of Life; Health Related Quality of Life; Sickle Cell Disease; Chronic Illness; Children and Adolescents
Background and Aims:
The psychological stress associated with type 1 diabetes (T1D) may be higher in children from developing world due to limited health resources. The aims of the study were to assess the quality of life (QoL), emotional well-being, behavioral, and cognitive profile of children/adolescents with T1D diagnosed at least 6 months prior.
Materials and Methods:
Forty-nine children with T1D, aged 6−18 years were assessed using DAWN Youth QoL questionnaire, WHO-5 Well-Being Index, Child Behavior Checklist (CBCL), and Malin's Intelligence Scale for Indian children (MISIC). The association of the scores was studied with age, gender, socioeconomic status (SES), frequency of hypoglycemia, HbA1c, and age of onset and duration of T1D.
The mean (standard deviation (SD)) for DAWN QoL, WHO-5, CBCL, and MISIC scores was 24.7 (16.7), 74.6 (19.4), 52.6 (8.8), and 96.0 (11.2), respectively. The significant associations noted were: Elevated HbA1c with poorer emotional well-being; higher negative impact on ‘symptoms of disease’ and ‘future prospects’ sub-areas of QoL; shorter duration of disease with more behavioral issues; lower maternal education with more ‘withdrawn/depressed’ behaviors and ‘worry about future prospects’; and lower SES with lower MISIC scores. Earlier onset (age <5 years) was associated with fewer behavioral problems and less negative impact on QoL.
Children with recent diagnosis, older age at onset, lower maternal educational level, elevated HbA1c, or belonging to lower SES were identified to have higher prevalence of various psychological and cognitive problems. In resource-limited settings, these children should be prioritized for behavioral and cognitive evaluation.
Behavior; children; cognition; quality of life; type 1 diabetes
The clinical presentation of celiac disease in children is very variable and differs with age. The prevalence of atypical presentations of celiac disease has increased over the past 2 decades. Several studies in adults and children with celiac disease indicate that obesity/overweight at disease onset is not unusual. In addition, there is a trend towards the development of overweight/obesity in celiac patients who strictly comply with a gluten-free diet. However, the pathogenesis and clinical implications of the coexistence of classic malabsorption (e.g., celiac disease) and overweight/obesity remain unclear. This review investigated the causes and main clinical factors associated with overweight/obesity at the diagnosis of celiac disease and clarified whether gluten withdrawal affects the current trends of the nutritional status of celiac disease patients.
celiac disease; overweight; obesity; gluten free diet
This study extends prior research on family caregiving in mental illness by investigating late-life parenting of adult children with bipolar disorder using a randomly selected community-based sample. The health and mental health, psychological well-being, marriage, work-life, and social resources of 145 parents of adult children with bipolar disorder were examined when parents were in their mid-60s. Stratified random sampling procedures were used to select a comparison group whose children did not have disabilities. Results indicate that parents of adult children with bipolar disorder had a more compromised profile of health and mental health, and experienced more difficulties in marriage and work-life than comparison parents. Furthermore, parents of adult children with bipolar disorder who were diagnosed with mental illness before the onset of their child’s symptoms were more vulnerable on multiple dimensions of mental health, psychological well-being, and work-life than parents without a preexisting mental illness.
Bipolar disorder; family caregiving; stratified random sampling
To determine within one tertiary care center: 1) the variation between providers in testing for celiac disease in children with chronic abdominal pain; 2) the characteristics of those children who were more likely to be tested, and 3) the prevalence of celiac disease in those evaluated.
Retrospective review of children with a primary complaint of chronic abdominal pain referred to a tertiary care children’s hospital for pediatric gastroenterology evaluation over a 2-year period was conducted. Children with at least two visits and without an identified organic etiology for the pain were included.
160 children were evaluated by 16 pediatric gastroenterologists and one nurse practitioner. Celiac serologic testing was completed in 63 (39.4%) children. There was no significant variance in the frequency of celiac serologic testing between providers. Child age, gender, body mass index, and baseline gastrointestinal symptoms did not predict whether celiac serologic testing occurred, though Caucasians (P < 0.01) were more likely to be tested. Eighty-two (51.3%) children underwent either serologic testing and/or esophagogastroduodenoscopy. Four (4.9%, 95% CI: 1.6-11.3%) of the 82 tested were diagnosed with celiac disease.
Though interprovider variation for celiac disease testing in children with chronic abdominal pain did not occur, a large number of these children were not evaluated for celiac disease. Children’s race/ethnicity but not their associated gastrointestinal symptoms predicted whether celiac testing was undertaken. In those tested, celiac disease was identified in a higher percentage than that expected in the general population.
Celiac disease; Children; Abdominal pain; Serology; Variation
This study examined the relationship between temperament and internalising and externalising problems among children of parents diagnosed with cancer, beyond the effects of socio-demographics, illness-related variables and life events.
Materials and methods
Three hundred and forty adolescent children and their 212 parents diagnosed with cancer participated. Children and parents completed the Youth Self Report and the Child Behaviour Checklist, respectively. Children completed also the Early Adolescent Temperament Questionnaire.
Daughters of parents with cancer were reported as having more internalising problems than their counterparts did. Prevalence of problems did not depend on children’s and parents’ age or educational level. Recurrent disease and number of life events experienced by children and parents affected the problems reported. The most important temperament dimensions in the prediction of internalising problems in children were shyness and fear/worry, to a lesser extent, frustration and perceptual sensitivity (children only) and lower scores on pleasure intensity (parents only). Externalising problems were associated with effortful control and in children’s reports with frustration. Temperament seemed to be a more important predictor of problems reported by children than parents.
Findings suggest that temperament is useful in determining the relative vulnerability of children of parents who have been diagnosed with cancer. Social workers may help parents to recognise individual differences between children and to support children by using techniques that are compatible with the temperament of children.
Parental cancer; Internalising and externalising problems; Adolescents; Temperament; Life events
To describe (1) anticipated health-related quality of life during different strategies for febrile neutropaenia (FN) management and (2) attributes of those preferring inpatient management.
Respondents were parents of children 0–18 years and children 12–18 years receiving cancer treatment. Anticipated health-related quality of life was elicited for four different FN management strategies: entire inpatient, early discharge, outpatient oral and outpatient intravenous (i.v.) therapy. Tools used to measure health-related quality of life were visual analogue scale (VAS), willingness to pay and time trade off.
A total of 155 parents and 43 children participated. For parents, median VAS scores were highest for early discharge (5.9, interquartile range 4.4–7.2) and outpatient i.v. (5.9, interquartile range 4.4–7.3). For children, median scores were highest for early discharge (6.1, interquartile range 4.6–7.2). In contrast, the most commonly preferred strategy for parents and children was inpatient in 55.0% and 37.2%, respectively. Higher current child health-related quality of life was associated with a stronger preference for outpatient management.
Early discharge and outpatient i.v. management are associated with higher anticipated health-related quality of life, although the most commonly preferred strategy was inpatient care. This data may help with determining more cost-effective strategies for paediatric FN.
health-related quality of life; child; febrile neutropaenia; willingness to pay; visual analogue scale; time trade-off technique
The quality of life of children with leukemia is reduced by fear and anxiety of parents after diagnosis, lack of information about the disease, treatments, and care of the child. This study aims to evaluate the effect of educating parents of leukemic children on the patients’ quality of life.
In this interventional study, sixty parents of ALL children who met inclusion criteria were selected using simple random sampling method, and assigned to the experimental and control groups. The study tool included a valid and reliable questionnaire (TNO-AZL), that was filled in through interview by parents before and two months after the intervention for both groups. The first part of the questionnaire included demographic items and the second part (7 dimensions, each with 8 sections) contained questions related to the quality of life. The scores could range between 56 and 280 and a higher score represented a better quality of life. The intervention included three one-hour classes composed of lecture and question-answer sessions which were held for groups of 4-6 participants, accompanied by a booklet.
Before the intervention, the quality of life score in the experimental and control groups was 180.83±14.43 and 174.28±20.72, respectively; after the intervention, these figures changed to 226.9±11.76 and 174.41±20.42 respectively. Paired samples T-test proved a significant increase in the quality of life in the experimental group.
Parent education successfully increased the quality of life of leukemic children; therefore, parental consultation sessions and educational programs are recommended.
Leukemia; Children; Education; Parent; Iran
‘Celiac crisis’ is a rare presentation of celiac disease with manifestations that include severe diarrhea, and severe metabolic and electrolyte abnormalities. It is most frequently seen in children younger than two years of age and has been rarely described in adults. A case of a 50-year-old woman who presented with diarrhea, severe dehydration, hypokalemia and metabolic acidosis is described. Based on positive serology and small bowel biopsy, she was diagnosed with celiac disease. She also had histological evidence of lymphocytic colitis. Microscopic colitis has not previously been described in association with celiac crisis, but it may have contributed to the presentation of celiac crisis in the current case. The patient was on corticosteroids and azathioprine for autoimmune hepatitis at the time of her presentation. The current case demonstrates that modest immunosuppression does not prevent a celiac crisis, although previous reports have shown that patients may respond rapidly to high-dose corticosteroids.
Acidosis; Celiac crisis; Celiac disease
A gluten-free diet is the treatment for celiac disease, but pharmaceutical agents are being developed. The level of interest amongst patients in using a medication to treat celiac disease is unknown. This study examined the level of interest amongst patients in medication to treat celiac disease.
A questionnaire was distributed to celiac disease patients and data were collected on demographics, presentation, and interest in medication. Three validated celiac disease-specific instruments were incorporated: Celiac Disease Associated Quality of Life, the Celiac Symptom Index, and the Celiac Dietary Adherence Test.
Responses were received from 365 individuals with biopsy-proven celiac disease. Respondents were 78% (n = 276) female, 48% (n = 170) over 50 years of age, and experienced a classical (diarrhea predominant) presentation in 44% (n = 154). Of the 339 individuals answering the question regarding use of a medication to treat celiac disease, 66% were interested. Interest was greatest in older individuals (71% >50 years of age versus 60% <50 years of age, p = 0.0415), men (78% men versus 62% women, p = 0.0083), frequent restaurant customers (76% versus 58%, p = 0.0006), those dissatisfied with their weight (73% versus 51%, p = 0.0003) and those concerned with the cost of a gluten-free diet (77% versus 64%, p = 0.0176). Length of time since diagnosis, education, presentation, and symptoms with gluten exposure did not demonstrate any effect. Interest in medication was associated with a worse quality of life (CD-QOL 69.4 versus 80.1, p < 0.0001).
Most individuals with celiac disease are interested in using a medication. Interest was highest among men, older individuals, frequent restaurant customers, individuals dissatisfied with their weight or concerned with the cost of a gluten-free diet, and those with a worse quality of life.
celiac disease; survey; therapy
Celiac disease often goes undiagnosed. Mass screening might be an option to reduce the public health burden of untreated celiac disease. However, mass screening is still controversial since it is uncertain whether the benefits of early detection outweigh the possible negative consequences. Before implementation of screening programs, the experiences of those being identified as cases should be considered. The aim of our study was to explore how screening-detected celiac disease impacts adolescents' quality of life, as perceived by themselves and their parents.
All adolescents (n = 145) with screening-detected celiac disease found in a Swedish screening study, and their parents, were invited to share their experiences in a qualitative follow-up study. In total, we have information on 117 (81%) of the adolescents, either from the adolescents themselves (n = 101) and/or from their parent/s (n = 125). Written narratives were submitted by 91 adolescents and 105 parents. In addition, 14 focus group discussions involving 31 adolescents and 43 parents were conducted. Data was transcribed verbatim and analyzed based on a Grounded Theory framework.
The screening-detected celiac disease diagnosis had varying impact on quality of life that related both to changes in perceived health and to the adolescents' experiences of living with celiac disease in terms of social sacrifices. Changes in perceived health varied from "healthy as anyone else with no positive change" to "something was wrong and then changed to the better", whereas experiences of living with celiac disease ranged from "not a big deal" to "treatment not worth the price". Perceptions about living with celiac disease and related coping strategies were influenced by contextual factors, such as perceived support from significant others and availability of gluten-free products, and were developed without a direct relation to experiencing changes in perceived health.
Screening-detected celiac disease has varying impact on adolescents' quality of life, where their perceived change in health has to be balanced against the social sacrifices the diagnosis may cause. This needs to be taken into account in any future suggestion of celiac disease mass screening and in the management of these patients.
Discrepancy between self-reports and parent-proxy reports of adolescent health-related quality of life (HRQoL) has been repeatedly acknowledged in the literature as the proxy problem. However, little is known about the extent and direction of this discrepancy. The purpose of this study is to explore to what extent and in what direction HRQoL self-reports of adolescents with chronic conditions and those of their parents differ.
A cross-sectional survey was conducted among adolescents suffering from chronic conditions and their parents. Socio-demographic and disease-related characteristics were collected and information about consequences of the chronic condition was assessed. HRQoL was measured with KIDSCREEN-10 and DISABKIDS condition generic measure (DCGM-10). Agreement was analysed through defining a threshold of agreement based on half of the standard deviation of the HRQoL score with the highest variance. Agreement occurred if the difference between adolescent and parent scores was less than or equal to half of the standard deviation. Intra-class correlation coefficients and Bland-Altman plots were also computed. The characteristics associated with direction of disagreement were statistically tested with one-way ANOVA and Chi-square tests.
584 paired HRQoL scores were obtained. Ratings from both adolescents and parents were high, compared to European norm data. Differences between adolescents and parents were statistically significant, yet relatively small. Disagreement existed in both directions: in 24.5% (KIDSCREEN-10) and 16.8% (DCGM-10) of the cases adolescents rated their HRQoL lower than did their parent, while in 32.2% (KIDSCREEN-10) and 31.7% (DCGM-10) of the cases the opposite was true. Adolescent's age, educational level and type of education, parent's educational level, number of hospital admissions and several other disease-related factors influenced direction of disagreement.
In a reasonable proportion of cases the adolescent and parent agreed on the adolescent's HRQoL (43-51% of the cases) and most disagreement tended to be minor. Thus, the proxy problem may be smaller than presented in the literature and its extent may differ per population. As adolescents are expected to become partners in their own health care, it is recommended to focus on adolescents' own perceptions of HRQoL.
Adolescent; Chronic Illness; Self Report; Quality of Life; Parent; Proxy Report; KIDSCREEN-10; DCGM-10
The current study was conducted to evaluate the effects of overweight, hyperglycemia symptoms, Hispanic ethnicity, and language barriers on health-related quality of life (HRQoL) among children and adolescents.
Parents'/guardians of a population based sample of 5530 children between ages 3 and 18 were administered the parents' version of the KINDL® survey instrument to assess HRQoL in children and adolescents. Multiple linear regression analysis was used to assess relationships between HRQoL, body mass index, and hyperglycemia symptoms categories.
The mean age of children was 10.6 (SD = 4.3). The mean KINDL® total score was 79.7 (SD = 11.6) and the mean physical functioning score was 81.9 (SD = 20.3). Male children exhibited better physical health as compared to the female children (p < 0.001). Overweight children had lower overall HRQoL (p = 0.008). However, the association was not significant for the four of the six subscales including the physical health domain. Children with hyperglycemia symptoms and a family history of diabetes also had significantly lower overall and physical health HRQoL (p < 0.05). Children diagnosed with diabetes and in lower income strata also had significantly lower overall HRQoL (p < 0.05). No significant association between the Hispanic ethnicity and HRQoL was observed. However, those who reported mostly speaking Spanish exhibited significantly lower overall HRQoL (p = 0.001).
Results suggest that overweight may reduce overall quality of life among children, though it does not directly influence physical functioning. However, hyperglycemia symptoms may affect both overall health and physical functioning. Findings also suggest the need for developing programs directed at overcoming language barriers that may face Spanish-speaking children or their parents. Furthermore, targeting children who have hyperglycemia symptoms with public information campaigns may be more appropriate than targeting overweight children.