Health-related Quality of Life Working Group

  • Description
  • Description

    The Quality of Life working group examines the health or health-related quality of life (HrQoL), which according to the WHO includes physical, mental, social and emotional aspects.

    A special interest is the development and validation of questionnaires to measure HrQoL especially for children, adolescents and young adults with chronic and rare diseases. Within national and international projects (see below) these instruments have been and will be developed, validated and used to learn more about the health and functioning of patients and parents from their own perspective.

    Epidemiological studies are conducted to analyze HrQoL in representative populations and clinical studies focus on investigating the long-term effect of medical therapies on the patient’s quality of life. Finally, health service research focuses on optimizing the quality and access to medical care in terms of HrQoL.

  • Current projects:

    Accessibility and barriers to language diagnostics and therapy for children with a diagnosed language development disorder - ZuBa-US (03/2020 - 02/2021)

    Language acquisition is one of the most important stages of child development. If a child does not successfully complete a language acquisition developmental stage (i.e. the pace of development stagnates or slows down), it is referred to as a specific developmental language disorder (SDLD). The SDLD is initially diagnosed as a suspected "late talker" from the 24th month of life. 30-50% of children make up for this deficit by the age of three. After the 36th month of life, the SDLD is labeled as a chronic disorder. Speech diagnostics and early speech therapy lead to a significant improvement in the child's speech as well as better chances in reaching critical developmental stages. In this planned study, current accessibility to speech diagnostics and therapy, as well as barriers and obstacles in reaching these services will be recorded and analyzed in qualitative interviews using the perspectives of parents and experts from various areas of the health and social system. Using these results, measures to improve the accessibility to early speech diagnostic tools and therapy will be developed.

    ZSE-DUO - Dual specialist structure for clarifying unclear diagnoses in centers for rare diseases (10/2018 - 10/2022)

    The aim of this study is to evaluate a new form of care (NFC) for patients with suspected rare diseases (RD). This new form of care should, among other things, increase the diagnostic success rate, shorten the time to diagnosis and enable a successful transition of the affected patients into regular care. In order to achieve this, the rare disease centers participating in the project will create a dual contact point between a somatic and a psychiatric specialist. This prospective cohort study with a control and intervention group is divided into two phases. In the first phase, the centers for rare diseases (ZSE) will provide patients with only one somatic specialist (standard care, control group). In phase two, a psychiatric specialist will also be involved in the patients’ care (NFC, intervention group). Within the framework of the overall project, the Quality of Life research group will conduct qualitative telephone interviews with patients in the intervention and control groups at two measurement points, at the beginning and after twelve months of treatment.

    Children with rare diseases, their siblings and parents - Children affected by rare disease and their families - network - CARE-FAM-NET (10/2018 - 10/2022)

    In order to improve the mental health and quality of life of children with rare diseases and their families in the long term, the CARE-FAM-NET aims to implement and evaluate a psychosocial intervention for children and young adults (0-21 years) with rare diseases, their siblings and parents. This study is a prospective, randomized controlled multicenter study in factorial design with four groups: (1) CARE-FAM intervention (face to face); (2) WEB-CARE online intervention; (3) a combination of both interventions (CARE-FAM and WEB-CARE) and (4) a control group. The CARE-FAM intervention is a cross-sectoral, family-oriented psychosocial face-to-face intervention. It is based on a needs assessment from the perspective of the families concerned. The WEP-CARE intervention is web-based and is based on proven principles of cognitive-behavioral writing therapy. Within the framework of the overall project, the Quality of Life research group analyses the interviews and questionnaire responses regarding current access to psychosocial care and the barriers to such care from the perspective of those affected and professionals from various fields in the health and education sectors. The goal of this subproject is to analyze and improve new pathways-to-care (PTC) approaches to secondary and tertiary preventive psychosocial interventions.

    Quality of life and coping mechanisms of parents of children with adrenogenital syndrome diagnosed in neonatal screening (06/2018-12/2020)

    In a retrospective mixed-method approach, the aim of this study is to assess the parental quality of life, coping mechanisms, and the needs of parents with children diagnosed with adrenogenital syndrome (AGS) in neonatal screening to derive appropriate intervention options. Quantitative and qualitative data will be collected in order to provide a comprehensive picture of the psychosocial burden of the parents concerned. Quantitative data on health-related quality of life and psychosocial stress will be collected using validated questionnaires (ULQIE, CHIP, Parental needs scale for parents of chronically ill children). Qualitative data will be collected by telephone interviews to gain more knowledge about the parent’s particular situation, personal health status and specific reactions after experiencing the positive screening result. Upon completion of the 12-month study, in depth knowledge of the parent’s needs, interests and wishes of affected parents will be summarized and translated for use in target group-oriented psychosocial interventions.

    “Sicherer Hafen”/ “Safe port” - Scientific monitoring and evaluation of the effectiveness of a modularized prevention program for disadvantaged families (01/2017-03/2020)

    The aim of the study is to scientifically monitor and evaluate the effectiveness of the prevention program “Sicherer Hafen’’ (funded by the Berndtsteinkinder foundation). Based on the results of the pilot study, measures to optimize the prevention program can be generated. Furthermore, it will be assessed whether the modularized program contributes to an increase in parental competencies, an increase in child and parental quality of life and a positive parent-child interaction, which promotes a healthy child development. At several measurement points, the wellbeing, possible burdens, satisfaction with the program and individual counseling will be assessed using a written questionnaire. Along with the collection of quantitative data, the parent-child interaction will be qualitatively evaluated.

    Finished Projects

    Quality of life in children and adolescents with esophageal atresia - cultural adaptation and validation of a disease specific questionnaire (2015/1 -2017/1)

    The aim of this study is to develop and verify the psychometric properties of a questionnaire that aims to increase the knowledge of the health-related quality of life (QoL) of children and adolescents born with esophageal atresia (EA). In order to learn more about the specific situation of children and adolescents with EA, a disease-specific QoL questionnaire will be developed simultaneously in Sweden and Germany.In the first phase of the study, the subjective health-related quality of life, experiences and coping strategies in children and adolescents born with EA from the child’s and parents’ perspectives will be investigated in a qualitative approach via focus groups and cognitive interviewing. A field and re-test will be done in the second phase of the study to test the psychometric properties quantitatively and compare it to validated QoL questionnaires like the KIDSCREEN (generic) and DISABKIDS (chronic generic) questionnaire. Additionally, the PedsQL™ Family Impact Module will be utilized to measure the impact of the child’s condition on the parents and family life. This project will lead to the first disease-specific instrument for EA and hence contribute to the improved understanding of long-term disease-related consequences from the child’s and parents’ perspectives.

    Linguistic validation of the QoLISSY and APLES instruments for the cross cultural health-related Quality of Life and Functioning assessment in children and adolescents with Achondroplasia(2016/02 – 2016/12)

    Achondroplasia is the most common form of disproportionate short stature. It is a result of an autosomal dominant mutation in the fibroblast growth factor receptor 3 gene (FGFR3), which causes an abnormality of cartilage and bone formation. BioMarin Pharmaceutical is currently developing and testing a natural human peptide, BMN-111, to treat achondroplasia in children. Phase 1 and 2 clinical trials have already been completed in children aged 5-14 years. BioMarin Pharmaceutical is currently planning Phase 3 Trial in which Patient-Reported Outcome (PRO) instruments will be used. PROs are defined as “any report of the status of a patient’s health condition that comes directly from the patient, without interpretation of the patient’s response by the clinician or anyone else”. In another study funded by BioMarin, different instruments, such as the generic PedsQL and the disease specific QoLISSY questionnaire, were reviewed and tested for use in the clinical trial. A new disease-specific instrument, the Achondroplasia Life Experience Scale (APLES), has also been included in this study. After completion of Phase 1 of the above mentioned PRO study, a decision was made to include the PedsQoL, QoLISSY and APLES in the planned phase 3 trial in Australia, France, Germany, Japan, Spain, Turkey, United Kingdom and USA. Since the QoLISSY and APLES instruments are not available in the languages of these countries, a linguistic validation is the first needed step towards a cross-cultural validation procedure.

    APLES – The Achondroplasia Personal Life Experience Scale – Development of a Questionnaire to Assess Quality of Life, Burden of Disease and Functionality of Children with Achondroplasia (2015/1-2015/12)

    This study aimed to determine disease-specific and patient-reported outcome (PRO) instruments that are suitable in terms of content and psychometry for assessing the functionality, needs and well-being of children with achondroplasia. The existing QoLISSY questionnaire examines the health related quality of life (HrQoL) of young patients with achondroplasia, but does not take the disease-specific burden and resources into account. Therefore, a new instrument was developed, implemented and validated. This new instrument, APLES (Achondroplasia Life Experience Scale), examines the HrQoL, burden of disease and functionality of children and adolescents with achondroplasia, while taking into account the parental perspective.

    Patient-reported Outcome - Selection and Validation for Achondroplasia (2015/1-2015/12)

    This project aims to select the most appropriate Patient Reported Outcome (PRO) instrument that best captures the interests of children with achondroplasia and their parents, as well as provide insights of the impacts of treatment benefits on the patients’ quality of life. Furthermore, the psychometric properties of these tools in children with achondroplasia will be assessed.

    Health-related Quality of Life of short statured children in the course of growth hormone treatment (2013/1 – 2016/10)

    With the availability of the internationally developed QOLISSY (Quality of Life Questionnaire for Short Statured Youth), it is now possible to examine changes in the quality of life of children and adolescents on growth hormone treatment from the patient and parental perspective. Within a multicenter, longitudinal study in Germany, relevant clinical, psychosocial and quality of life-related data will be collected from children and examined before and 1 year after the start of the growth treatment. The study focuses on changes in the QOLISSY quality of life questionnaire measurements over time. Short statured children (GHD & SGA) who visit cooperating clinics and plan to undergo growth hormone are examined. Patients diagnosed with Idiopathic Short Stature (ISS), a group who are not normally treated with growth hormones, will serve as a control group in this prospective observational study.

  • Selected publications

    • Witt, S., Blömeke, J., Bullinger, M., Dingemann, J., & Quitmann, J. (2020). Basic Principles of Health-Related Quality of Life in Parents and Caregivers of Pediatric Surgical Patients with Rare Congenital Malformations-A Scoping Review. EUR J PEDIATR SURG, 30(3), 225-231.
    • Dellenmark-Blom, M., Quitmann, J., Dingemann, J., Witt, S., Ure, B. M., Bullinger, M., ... Dingemann, C. (2020). Clinical Factors Affecting Condition-Specific Quality-of-Life Domains in Pediatric Patients after Repair of Esophageal Atresia: The Swedish-German EA-QOL Study. EUR J PEDIATR SURG, 30(1), 96-103.
    • Feng, X., Lacher, M., Quitmann, J., Witt, S., Witvliet, M. J., & Mayer, S. (2020). Health-Related Quality of Life and Psychosocial Morbidity in Anorectal Malformation and Hirschsprung's Disease. EUR J PEDIATR SURG, 30(3), 279-286.
    • Bloemeke, J., Witt, S., Bullinger, M., Dingemann, J., Dellenmark-Blom, M., & Quitmann, J. (2020). Health-Related Quality of Life Assessment in Children and their Families: Aspects of Importance to the Pediatric Surgeon. EUR J PEDIATR SURG, 30(3), 232-238.
    • Dingemann, J., Dellenmark-Blom, M., & Quitmann, J. H. (2020). Health-Related Quality of Life in Pediatric Surgical Patients and their Caretakers. EUR J PEDIATR SURG, 2020(30), 223-224.
    • Witt, S., Heiden, J., & Quitmann, J. H. (2020). Lebensqualität und psychische Gesundheit von Kindern mit Adrenogenitalem Syndrom. Päd Praxis, 93(03), 385-397.
    • Witt, S., Bloemeke, J., Bullinger, M., Dingemann, J., Dellenmark-Blom, M., & Quitmann, J. (2019). Agreement between mothers', fathers', and children's' ratings on health-related quality of life in children born with esophageal atresia - a German cross-sectional study. BMC PEDIATR, 19(1), 330.
    • Bloemeke, J., Sommer, R., Witt, S., Bullinger, M., Nordon, C., Badia, F. J., ... Quitmann, J. (2019). Cross-cultural selection and validation of instruments to assess patient-reported outcomes in children and adolescents with achondroplasia. QUAL LIFE RES, 28(9), 2553-2563.
    • Witt, S., & Quitmann, J. H. (2019). Eine Diagnose überbringen? Warum gerade ich? Deutsche Hebammenzeitschrift, 71(10), 30-35.
    • Quitmann, J., Bloemeke, J., Dörr, H-G., Bullinger, M., Witt, S., & Silva, N. (2019). First-year predictors of health-related quality of life changes in short-statured children treated with human growth hormone. J ENDOCRINOL INVEST, 42(9), 1067-1076.
    • Flieder, S., Dellenmark-Blom, M., Witt, S., Dingemann, C., Quitmann, J. H., Jönsson, L., ... Dingemann, J. (2019). Generic Health-Related Quality of Life after Repair of Esophageal Atresia and Its Determinants within a German-Swedish Cohort. EUR J PEDIATR SURG, 29(1), 75-84.
    • Witt, S., Dellenmark-Blom, M., Flieder, S., Dingemann, J., Abrahamsson, K., Jönsson, L., ... Quitmann, J. H. (2019). Health-related quality of life experiences in children and adolescents born with esophageal atresia: A Swedish-German focus group study. CHILD CARE HLTH DEV, 45(1), 79-88.
    • Bloemeke, J., Sommer, R., Witt, S., Dabs, M., Badia, F. J., Bullinger, M., & Quitmann, J. (2019). Piloting and psychometric properties of a patient-reported outcome instrument for young people with achondroplasia based on the International Classification of Functioning Disability and Health: the Achondroplasia Personal Life Experience Scale (APLES). DISABIL REHABIL, 41(15), 1815-1825.
    • Bloemeke, J., Silva, N., Bullinger, M., Witt, S., Dörr, H-G., & Quitmann, J. (2019). Psychometric properties of the quality of life in short statured youth (QoLISSY) questionnaire within the course of growth hormone treatment. HEALTH QUAL LIFE OUT, 17(1), 49.
    • Witt, S., Kolb, B., Bloemeke, J., Mohnike, K., Bullinger, M., & Quitmann, J. (2019). Quality of life of children with achondroplasia and their parents - a German cross-sectional study. ORPHANET J RARE DIS, 14(1), 194.
    • Quitmann, J. H., Bloemeke, J., Silva, N., Bullinger, M., Witt, S., Akkurt, I., ... Dörr, H-G. (2019). Quality of Life of Short-Statured Children Born Small for Gestational Age or Idiopathic Growth Hormone Deficiency Within 1 Year of Growth Hormone Treatment. FRONT PEDIATR, 7, 164.

  • Qualifying theses (2019/2020)

    • Böhme, Paula (laufend). Behandlungszufriedenheit und gesundheitsbezogene Lebensqualität von primär undiagnostizierten Patienten mit Verdacht auf eine seltene Erkrankung nach 12-monatiger somatisch-fachärztlicher Betreuung an einem Zentrum für seltene Erkrankungen (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Schnieders, Miriam (laufend). Behandlungserwartungen und Zufriedenheit von PatientInnen mit unklarem komplexen Beschwerdebild mit Verdacht auf eine seltene Erkrankung (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
      Schütt, Katharina (laufend). Hindernisse und Barrieren von Kindern mit seltenen Erkrankungen und deren Familien in der psychosozialen Versorgung (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Leja, Anja (laufend). Zugang und Barrieren zur Sprachdiagnostik und -therapie bei Kindern mit umschriebener Sprachentwicklungsstörung aus Expertenperspektive (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Thielmann, Deborah (laufend). Zugang und Barrieren zur Sprachdiagnostik und -therapie bei Kindern mit umschriebener Sprachentwicklungsstörung aus Elternperspektive (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Korczanowski, Leonie (laufend). Externalisierende und internalisierende Auffälligkeiten von Vorschulkindern während der Covid-19 Pandemie aus Elternsicht (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Uekermark, Clarissa (laufend). Elterliches Stresserleben in Familien mit Kindern im Kindergartenalter in Pandemiezeiten – Zusammenhänge von elterlichem Stress, Depressivität und Angst und kindlichen Verhaltensauffälligkeiten (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Steinert, Maren (laufend). Entwicklung eines Fragebogens zur Erfassung der gesundheitsbezogenen Lebensqualität und diagnosespezifischen Belastungen von Eltern mit Kinder mit einem adrenogenitalen Syndrom (AGS) mit 21-Hydroxylase-Defekt (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Lackner, Lea (laufend). Belastungen von Eltern von Kindern mit Idiopathischem Kleinwuchs oder Wachstumshormonmangel (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Hagen, Marie (laufend). Lebensqualität und psychosoziale Belastungen der Eltern von Kindern mit der angeborenen Anomalie „Ösophagusatresie“ - Entwicklung eines Pilot-Fragebogens zur Erfassung von elterlichen Belastungen und ihrer Lebensqualität bei der Fürsorge und Betreuung ihrer erkrankten Kinder (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Schmidt, Eileen (laufend). Lebensqualität und psychosoziale Belastung der Eltern von Kindern mit Achondroplasie - Entwicklung eines Pilotfragebogens zur Erfassung der Auswirkungen auf die elterliche Lebensqualität (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • MacAllister, Margarita (laufend). Gesundheitsbezogene Lebensqualität von Patient*innen mit diagnostizierter Zystinose (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Fey, Mie-Ra (laufend). Untersuchung der Auswirkungen durch die Corona-Pandemie auf die Lebensqualität von Patienten und deren Bezugspersonen mit Cystinose in Deutschland (Doktorarbeit PhD Programm Mediziner, Medizinische Fakultät, UKE)
    • Suck, Isabell (laufend). HIV-PRO – Patientenberichtete Endpunkte (engl. patient-reported outcome; PRO) bei HIV-positiven Patient*innen – Eine qualitative Studie zur subjektiven Gesundheit, Lebensqualität und zum Krankheitsverständnis von Patient*innen mit einer HIV-Infektion (Master-Thesis, Gesundheitswissenschaften Hochschule für Angewandte Wissenschaften Hamburg, Fakultät Life Sciences)
    • Heiser, Maike (laufend). Mother-Infant interaction patterns within the course of an early parenting training (Master-Thesis, Gesundheitswissenschaften, Hochschule für Angewandte Wissenschaften Hamburg, Fakultät Life Sciences)
    • Degirmencioglu, Hacer (laufend). Verhaltensauffälligkeiten bei 2-jährigen Kindern mit umschriebener Sprachentwicklungsstörungen (USES) (Master-Thesis, Soziale Arbeit, Medical School Hamburg, Fakultät Art, Health and Social Science)
    • Könekamp, Leonie (laufend). Kindliche Lebensqualität bei Late-Talkern aus der Perspektive der Eltern (Bachelor-Thesis, Psychologie, Medical School Hamburg, Fakultät Humanwissenschaften)
    • Borcherding, Antonia, (laufend). Resilienz und gesundheitsbezogene Lebensqualität bei Kindern im Vorschulalter - Eine retrospektive qualitative Analyse des Präventionsprogramms SUNNI (Master-Thesis, Psychologie, TU Chemnitz, Fakultät für Human- und Sozialwissenschaften)
    • Blatt, Wilhelm (2021). Die Entwicklung eines Patient-Benefit-Index für Menschen mit seltenen Erkrankungen (Bachelor-Thesis, Psychologie, Fresenius Hochschule)