Julia Quitmann

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

BackgroundWhen evaluating the outcomes of treatment in paediatric endocrinology, the health-related quality of life (HrQoL) of the child is to be taken into consideration. Since few self–reported HrQoL instruments exist for children with diagnosed short stature (dSS), the objective of this study was to develop and psychometrically test a targeted HrQoL instrument for use in multinational clinical research.MethodsThe target population were short stature (height < −2 SDS) children and adolescents (age 8–12 and 13–18 years) with a diagnosis of growth hormone deficiency (GHD) or idiopathic short stature (ISS), differing in growth hormone treatment status. Focus group discussions for concept and item generation, piloting of the questionnaire with cognitive debriefing, and instrument field testing with a retest were conducted simultaneously in five countries. After qualitative and preliminary quantitative analyses, psychometric testing of field test data in terms of reliability and validity including confirmatory factor analyses (CFA) was performed.ResultsFollowing item generation from focus group discussions, 124 items were included in a pilot test with a cognitive debriefing exercise providing preliminary feedback on item and domain operating characteristics. A field test with 268 participants showed high internal consistency reliabilities (alpha 0.82 – 0.95), good correlations with generic measures (up to r = .58), significant known group differences (e.g. in height: F = 32, df 244, p < 0.001) and an acceptable CFA model fit suggesting construct validity of the three-domain core structure with 22 items, supplemented by three mediator domains with 28 items.ConclusionsThe QoLISSY questionnaire is a promising step forward in assessing the impact of dSS on HrQoL. It is based on items generated from the subjective experience of short stature children referred for endocrine investigation, is validated for use in five languages and it is easy to administer in clinical and research settings.

Gesundheitsbezogene Lebensqualität und psychische Gesundheit von kleinwüchsigen Kindern und Jugendlichen

Three percent of children and adolescents in Germany have short stature (SS) which also includes patients with growth hormone deficiency (GHD) and idiopathic short stature (ISS). Growth hormone substitution in these patients is expected to increase height, health-related quality of life (HrQoL) and mental health. The present cross-sectional study investigated healthrelated quality of life and mental health of children with SS in comparison to a representative German population sample and examined differences within patients with respect to treatment status, diagnosis and actual height. A total of 143 patients (4 ‐ 18 years) and their parents completed questionnaires for mental health (SDQ) and health-related quality of life (KIDSCREEN-52). Results showed that children and adolescents with short stature report significant HrQoL impairments in comparison to children with normal height, while differences in mental health were apparent at specific subscale levels. Within the clinical sample, no differences regarding age and gender were detected. Also, no significant differences in HrQoL and mental health were found according to diagnosis (GHD/ISS), treatment status and actual height. Parents rated their childrens’ HrQoL higher than the children themselves, whereas they considered their children’ psychological health problems more serious than the children themselves did. Results suggest that SS is associated with impairments in HrQol and mental health as compared to a population reference group, suggesting that these impairments should be addressed with condition-specific interventions. Generic tools such as KIDSCREEN did not yield differences within the patient group regarding clinical and socio-demographic characteristics. Clinical studies therefore should consider making use of condition specific measures.

Cross-Cultural Equivalence of the Patient- and Parent-Reported Quality of Life in Short Stature Youth (QoLISSY) Questionnaire

Background: Testing cross-cultural equivalence of patient-reported outcomes requires sufficiently large samples per country, which is difficult to achieve in rare endocrine paediatric conditions. We describe a novel approach to cross-cultural testing of the Quality of Life in Short Stature Youth (QoLISSY) questionnaire in five countries by sequentially taking one country out (TOCO) from the total sample and iteratively comparing the resulting psychometric performance. Methods: Development of the QoLISSY proceeded from focus group discussions through pilot testing to field testing in 268 short-statured patients and their parents. To explore cross-cultural equivalence, the iterative TOCO technique was used to examine and compare the validity, reliability, and convergence of patient and parent responses on QoLISSY in the field test dataset, and to predict QoLISSY scores from clinical, socio-demographic and psychosocial variables. Results: Validity and reliability indicators were satisfactory for each sample after iteratively omitting one country. Comparisons with the total sample revealed cross-cultural equivalence in internal consistency and construct validity for patients and parents, high inter-rater agreement and a substantial proportion of QoLISSY variance explained by predictors. Conclusion: The TOCO technique is a powerful method to overcome problems of country-specific testing of patient-reported outcome instruments. It provides an empirical support to QoLISSYs cross-cultural equivalence and is recommended for future research.

The capacity to see things from the child's point of view--assessing insightfulness in mothers with and without a diagnosis of depression.

UNLABELLED Insightfulness is seen as the mental capacity that provides the context for a secure child-parent attachment. It involves the ability to see things from the childs perspective and is based on insight into the childs motives, a complex view of the child and openness to new information about the child. To test our hypothesis that maternal insightfulness is related to maternal depression, we utilized the Insightfulness Assessment (IA) developed by Oppenheim and Koren-Karie to conduct and analyse interviews in which mothers discussed their perceptions of video segments of their interactions with their children. We compared the results of a control group of 30 mothers without a diagnosis of depression with a sample of 23 mothers diagnosed with depression (International Classification of Diseases, 10th Revision). As expected, depression was negatively related to maternal insightfulness. Oppenheim and Koren-Karie have argued that the IA might be used as a diagnostic instrument; our work suggests that the IA might show ways of aiding mothers in improving the quality of mother-infant interaction while they are being treated for depression. KEY PRACTITIONER MESSAGE Insightfulness is seen as the mental capacity that provides the context for a secure child-parent attachment. Maternal depression was negatively related to maternal insightfulness. The Insightfulness Assessment might be used as a diagnostic instrument. The Insightfulness Assessment might show ways of aiding mothers in improving the quality of mother-infant interaction while they are being treated for depression.

Understanding the impact of statural height on health-related quality of life in German adolescents: a population-based analysis

AbstractImproving health-related quality of life (HrQoL) is one aim of growth hormone treatment for short-statured patients. Yet, evidence of the relationship between height and HrQoL is inconclusive, especially for young people. This study investigates the association of height and HrQoL in the general German adolescent population. A total of 6646 adolescents and 6388 parents from the German Health Interview and Examination Survey for Children and Adolescents (KIGGS) were included in the analysis. An analysis of covariance was used to evaluate the effect of height deviation on HrQoL as assessed with the KINDL-R in adolescent self-report and parent-report, taking into account sociodemographic and health-related variables. Height was found to be a weak predictor of HrQoL in both adolescent-report and parent-report. Short and tall adolescents did not substantially differ from the normal statured. Sociodemographic and health-related variables failed to significantly interact with height. In contrast, psychosocial variables contributed to the explained HrQoL variance. Conclusion: Height does not appear to be a strong determinant of HrQoL on the population level. These results suggest that the height gain in short stature treatment may not be the most important objective. Contrary to prevalent stereotypes, short adolescents can experience a high HrQoL.

Insightfulness and later infant attachment in clinically depressed and nonclinical mothers.

The aim of this pilot study was to investigate the relationship between maternal insightfulness and sensitivity and subsequent infant attachment security and disorganization in clinically depressed and nonclinical mother-infant groups. Nineteen depressed mothers with infants ages 3 to 11 months participated in this study. Twenty nonclinical mother-infant dyads were matched to the clinical sample according to infant sex and age. Maternal depression was assessed using the Diagnostic and Statistical Manual of Mental Disorders, fourth edition (American Psychiatric Association, 1994), insightfulness using the Insightfulness Assessment (IA), and sensitivity using the Maternal Sensitivity Scales (M.D.S. Ainsworth, 1969). IA classifications and subscales were considered separately. Later infant attachment was assessed by the Strange Situation Procedure (M.D.S. Ainsworth, M.C. Blehar, E. Waters, & S. Wall, 1978). Depressed mothers tended to have less securely attached children than did nonclinical mothers. Within the clinical sample, the insightfulness categories correlated slightly moderately with attachment security, but were not related to attachment disorganization. Within the nonclinical sample, the IA categories were slightly moderately associated with attachment security and with disorganization. On IA subscales, relationship patterns differed in clinically depressed and nonclinical mother-infant dyads. These findings provide the first evidence of the predictive power of the IA categorization and subscales on subsequent infant attachment. They also may allow the development of different foci of intervention for enhancing insightful caregiving.

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

BackgroundThe 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.MethodsUpon 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.ResultsPilot test results supported the suitability of the American-English version. Good internal consistency with Cronbach’s 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’s concurrent validity. The scale’s operating characteristics were comparable to the original European data.ConclusionResults 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.

Leben mit Achondroplasie

OBJECTIVE Presently, little is known aqout the quality of life (QoL) as well as the strengths and difficulties of young people with achondroplasia. This study describes these patient-reported indicators and identifies possible correlates. METHOD At the invitation of a patient organization, a total of 89 short-statured patients aged 8 to 28 years and their parents participated in this study. QoL was assessed cross-sectionally with both generic and disease-specific instruments and the Strengths and Difficulties Questionnaire (SDQ) as a brief behavioral screening. In addition to descriptive analyses, patient data were compared with a reference population. Hierarchical regression analyses reflecting sociodemographic, clinical, and psychological variables were conducted to identify correlates of QoL. RESULTS QoL and the strengths and difficulties of young patients with achondroplasia did not differ substantially from a healthy norm sample. However, the participants reported more behavioral problems and limitations in their physical and social QoL compared to patients with another short stature diagnosis. Strengths and difficulties, height-related beliefs, and social support correlated significantly with QoL. Adding psychological variables to the regression model increased the proportion of variance explained in QoL. CONCLUSIONS Young persons with achondroplasia did not differ in their QoL and strengths and difficulties from healthy controls. Characteristics such as height appear less important for the self-perceived QoL than are strengths and difficulties and protective psychosocia~factors.

Development and pilot-testing of a condition-specific instrument to assess the quality-of-life in children and adolescents born with esophageal atresia

The survival rate of children with esophageal atresia has today reached 95%. However, children are at risk of chronic morbidity related to esophageal and respiratory dysfunction, and associated anomalies. This study describes the pilot testing of a condition-specific health-related quality-of-life instrument for children with esophageal atresia in Sweden and Germany, using a patient-derived development approach consistent with international guidelines. Following a literature review, standardized focus groups were conducted with 30 Swedish families of children with esophageal atresia aged 2-17 years. The results were used for item generation of two age-specific pilot questionnaire versions. These were then translated from Swedish into German with considerations of linguistic and semantical perspectives. The 30-item pilot questionnaire for children aged 2-7 years was completed by 34 families (parent report), and the 50-item pilot questionnaire for children aged 8-17 years was completed by 52 families (51 child report, 52 parent report), with an overall response rate of 96% in the total sample. Based on predefined psychometric criteria, poorly performing items were removed, resulting in an 18-item version with three domains (Eating, Physical health and treatment, Social isolation and stress,) for children aged 2-7 years and a 26-item version with four domains (Eating, Social relationships, Body perception, and Health and well-being) for children aged 8-17 years. Both versions demonstrated good internal consistency reliability and acceptable convergent and known-groups validity for the total scores. The study identified specific health-related quality-of-life domains for pediatric patients with esophageal atresia, highlighting issues that are important for follow-up care. After field testing in a larger patient sample, this instrument can be used to enhance the evaluation of pediatric surgical care.

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.