Explaining parent-child (dis)agreement in generic and short stature-specific health-related quality of life reports: do family and social relationships matter?

Background: In the context of health-related quality of life (HrQoL) assessment in pediatric short stature, the present study aimed to examine the levels of agreement/disagreement between parents’ and children’s reports of generic and condition-specific HrQoL, and to identify socio-demographic, clinical and psychosocial variables associated with the extent and direction of parent-child discrepancies. Methods: This study was part of the retest phase of the QoLISSY project, which was a multicenter study conducted simultaneously in France, Germany, Spain, Sweden and UK. The sample comprised 137 dyads of children/adolescents between 8 and 18 years of age, diagnosed with growth hormone deficiency (GHD) or idiopathic short stature (ISS), and one of their parents. The participants completed child- and parent-reported questionnaires on generic (KIDSCREEN-10 Index) and condition-specific HrQoL (QoLISSY Core Module). Children/adolescents also reported on social support (Oslo 3-items Social Support Scale) and parents assessed the parent-child relationships (Parental Role subscale of the Social Adjustment Scale) and burden of short stature on parents (QoLISSY- additional module). Results: The parent-child agreement on reported HrQoL was strong (intraclass correlation coefficients between .59 and .80). The rates of parent-child discrepancies were 61.5 % for generic and 35.2 % for condition-specific HrQoL, with the parents being more prone to report lower generic (42.3 %) and condition-specific HrQoL (23.7 %) than their children. The extent of discrepancies was better explained by family and social relationships than by clinical and socio-demographic variables: poorer parent-child relationships and better children’s social support were associated with larger discrepancies in generic HrQoL, while more parental burden was associated with larger discrepancies in condition-specific HrQoL reports. Regarding the direction of discrepancies, higher parental burden was significantly associated with parents’ underrating, and better children’s social support was significantly associated with parents’ overrating of condition-specific HrQoL. Conclusions: Routine assessment of pediatric HrQoL in healthcare and research contexts should include child- and parent-reported data as complementary sources of information, and also consider the family and social context.This study is part of the “Quality of Life in Short Stature Youth” (QoLISSY) project, a joint initiative between the University Medical Centre Hamburg-Eppendorf and ©Pfizer Limited

Evaluation of the American-English Quality of Life in Short Stature Youth (QoLISSY) questionnaire in the United States.

BACKGROUND: The European Quality of Life in Short Stature Youth (QoLISSY) is a novel condition-specific instrument developed to assess health related quality of life (HrQoL) in children/adolescents with short stature from patient and parent perspectives. Study objective was to linguistically validate and psychometrically test the American-English version of the QoLISSY instrument. METHODS: Upon conversion of the British-English version to American-English, content validity and acceptance of the questionnaire were examined through focus group discussions with cognitive debriefing in 28 children/adolescents with growth hormone deficiency (GHD) or idiopathic short stature (ISS) and their parents. In the subsequent field test with 51 families and a re-test with 25 families the psychometric performance of the American-English version was examined and compared with the original European dataset. RESULTS: Pilot test results supported the suitability of the American-English version. Good internal consistency with Cronbach\u27s Alpha ranging from 0.84 to 0.97 and high test-re-test reliabilities were observed in the field test. The QoLISSY was able to detect significant differences according to the degree of short stature with higher HrQoL for taller children. Correlations with a generic HrQoL tool support the QoLISSY\u27s concurrent validity. The scale\u27s operating characteristics were comparable to the original European data. CONCLUSION: Results support that the QoLISSY American-English version is a psychometrically sound short stature-specific instrument to assess the patient- and parent- perceived impact of short stature. The QoLISSY instrument is fit for use in clinical studies and health services research in the American-English speaking population

Assessing the quality of life of health-referred children and adolescents with short stature: development and psychometric testing of the QoLISSY instrument

10.1186/1477-7525-11-76Health and Quality of Life Outcomes1117

Quality of Life of Short-Statured Children Born Small for Gestational Age or Idiopathic Growth Hormone Deficiency Within 1 Year of Growth Hormone Treatment

Aside from clinical endpoints like height gain, health-related quality of life has also become an important outcome indicator in the medical field. However, the data on short stature and health-related quality of life is inconsistent. Therefore, we examined changes in health-related quality of life in German children with idiopathic growth hormone deficiency or children born small for gestational age before and after 12 months of human growth hormone treatment. Children with idiopathic short stature without treatment served as a comparison group. At baseline, health-related quality of life data of 154 patients with idiopathic growth hormone deficiency (n = 65), born small for gestational age (n = 58), and idiopathic short stature (n = 31) and one parent each was collected. Of these, 130 completed health-related quality of life assessments after 1-year of human growth hormone treatment. Outcome measures included the Quality of Life in Short Stature Youth questionnaire, as well as clinical and sociodemographic data. Our results showed that the physical, social, and emotional health-related quality of life of children treated with human growth hormone significantly increased, while untreated patients with idiopathic short stature reported a decrease in these domains. Along with this, a statistically significant increase in height in the treated group can be observed, while the slight increase in the untreated group was not significant. In conclusion, the results showed that human growth hormone treatment may have a positive effect not only on height but also in improving patient-reported health-related quality of life of children with idiopathic growth hormone deficiency and children born small for gestational age

Caregiving burden and special needs of parents in the care of their short-statured children – a qualitative approach

PurposeTo explore caregiving burden, health-related quality of life (HRQOL), stress, and individual resources of parents in the care of children with isolated growth hormone deficiency (IGHD) or idiopathic short stature (ISS).MethodsFocused interview analysis of previously, within the Quality of Life in Short Stature Youth (QoLISSY) project, conducted structured focus group discussions (n=7) with parents (n=33) of children with IGHD/ISS aged 4 to 18 years were performed.Results26 out of the 33 parents reported mental stress due to their child’s growth disorder. Social pressure and stigmatization were also mentioned as being demanding. Some parents reported having trouble with human growth hormone (hGH) treatment. Several parents wished for parent support groups with other like-minded parents of short-statured children.ConclusionFor physicians, it is essential to understand the parents’ caregiving burden, stress, and individual resources in caring for IGHD/ISS children. If an impaired HRQOL is detected, psychological intervention for these parents may be scheduled, and coping mechanisms may be discussed. Furthermore, it seems essential for parents to be educated by their healthcare provider about the possible side effects of hGH treatment or to know where to find evidence-based information about it

Associations between psychological problems and quality of life in pediatric short stature from patients’ and parents’ perspectives

Short stature has been associated with psychosocial impairments, but whether treatments and achieved height impact on health-related quality of life (HrQoL) and psychological functioning of children/adolescents is still controversial. This study aimed to examine the effects of height deviation and treatment status on psychosocial adaptation outcomes and to identify clinical and psychosocial determinants of internalizing/externalizing problems in a large cohort of short statured children/adolescents from seven European countries. Participants were 345 children aged 8–18 years with a clinical diagnosis of short stature and 421 parents of 4–18 year-old patients. Children and parents reported on psychological problems (Strengths and Difficulties Questionnaire), generic (KIDSCREEN) and condition-specific HrQoL (QoLISSY). According to analyses of covariance, children/adolescents with current short stature presented more parent-reported internalizing problems and lower self- and parent-reported condition-specific HrQoL, compared to patients with an achieved height above -2SD. Treated children self-reported better HrQoL than the untreated group. Hierarchical regression analysis showed that, rather than height–related clinical variables, children’s sex, younger age and poorer HrQoL were the best predictors of psychological problems, explaining 39% of the variance in patient- and 42% in parent-reported internalizing problems, and 22% of the variance in patient- and 24% in parent-reported externalizing problems. Treatment status also moderated the negative links between patient-reported HrQoL and internalizing problems, explaining 2% of additional variance. These results suggest that children with current short stature are at greater risk for internalizing problems. Routine assessment of HrQoL in pediatric healthcare may help identify children for referral to specialized psychological assessment and intervention

HRQoL of European children and adolescents with short stature as assessed with generic (KIDSCREEN) and chronic-generic (DISABKIDS) instruments

Introduction. Short stature may be associated with impairments in health-related quality of life (HrQoL). This study compares the HrQoL of children/adolescents diagnosed with short stature to population norms and examined the effects of height deviation and treatment status on HrQoL. Method. We cross-sectionally assessed 110 children/adolescents aged 8-18, with current short stature (height deviation equal to or below -2 SD) or normal height achieved since diagnosis, and 98 parents, using the generic KIDSCREEN and the chronic-generic DISABKIDS instruments. Results. Generic HrQoL of patients was similar to population norms. Patients with achieved normal height reported better chronic-generic HrQoL when untreated, while patients with current short stature reported better HrQoL when receiving treatment. Parents reported better HrQoL for treated patients, especially for girls. Conclusion. Although their HrQoL is not significantly compromised, patients diagnosed with short stature may profit from growth-hormone treatment. Specific instruments are needed to adequately assess the effectiveness of treatment.The development of the QoLISSY questionnaire was funded through a research grant by Pfizer Ltd. MB has received honoraria and acted as consultant. AP and HW (members of the QoLISSY group) are Pfizer Employees. All other authors have no involvement with Pfizer ltd. nor have financial interests. This includes employment, consultancies, honoraria, stock ownership or options, expert testimony, grants or patents received or pending, or royalties. NS was supported by a PhD Grant from the Portuguese Foundation for Science and Technology (SFRH/BD/69885/2010)

Parent Perceptions of a Pretend Play Intervention for Their Children With Cancer

BackgroundAlthough the rate of survival in childhood cancer today is close to 85%, a cancer diagnosis can still turn the world upside down for both children and parents. Often, children in oncology care are frustrated about their inability to control events and activities around them. Therapeutic pretend play has been suggested as a means to encourage children to express and handle emotions in a safe environment.PurposeThis study was developed to describe and explore parents' experiences of a pretend play intervention that consisted of six to eight play sessions with a play facilitator administered to their children undergoing cancer treatment.MethodsA descriptive qualitative method was used, including individual interviews with 15 parents.ResultsThree main categories were developed, including (a) experiences of joining the project, (b) perceptions of the play intervention, and (c) reflections on effects and implications, with subcategories evolved for each category. The parents experienced that the play sessions helped improve their children's communication skills and made them more capable of participating in their care. They appreciated that the intervention focused on the child's well-being and saw it as a positive break in their child's cancer treatment. It also helped them better reflect on their own situation.Conclusions/Implications for PracticeAccording to the parents' experiences, pretend play can be a helpful tool for improving children's participation in their cancer care that strengthens their autonomy, emotional repertoire, and communication skills. However, the results also highlighted that some of the children did not fully understand the information provided about this study, which weakened the validity of their consent to participate. Thus, more work is needed on developing age-appropriate information to obtain participation consent from children. In addition, more knowledge is needed regarding how to appropriately include children with cancer in research in an ethically acceptable way