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Children and adolescents with VACTERL association: health-related quality of life and psychological well-being in children and adolescents and their parents
Uppsala universitet, Medicinska och farmaceutiska vetenskapsområdet, Medicinska fakulteten, Institutionen för kvinnors och barns hälsa, Forskargrupper (Inst. för kvinnor och barns hälsa), Barnkirurgisk forskning.
Department of Pediatrics, Institute of Clinical Sciences, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden; Department of Paediatric Surgery, The Queen Silvia Children's Hospital SU/Östra, Gothenburg, Sweden.
Department of Clinical Neuroscience, Karolinska Institutet, Stockholm, Sweden; The Multidisciplinary Pain Centre and Rehabilitation Medicine, Uppsala University Hospital, Uppsala, Sweden.
Uppsala universitet, Medicinska och farmaceutiska vetenskapsområdet, Medicinska fakulteten, Institutionen för kvinnors och barns hälsa, Forskargrupper (Inst. för kvinnor och barns hälsa), Barnneurologi/Barnonkologi.
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2019 (Engelska)Ingår i: Quality of Life Research, ISSN 0962-9343, E-ISSN 1573-2649Artikel i tidskrift (Refereegranskat) Epub ahead of print
Abstract [en]

PURPOSE: VACTERL association is a rare and complex condition of congenital malformations, often requiring repeated surgery and entailing various physical sequelae. Due to scarcity of knowledge, the study aim was to investigate self-reported health-related quality of life (HRQoL), anxiety, depression and self-concept in children and adolescents with VACTERL association and self-reported anxiety and depression in their parents.

METHODS: Patients aged 8-17 years with VACTERL association and their parents were recruited from three of four Swedish paediatric surgical centres during 2015-2019. The well-established validated questionnaires DISABKIDS, Beck Youth Inventories, Beck Anxiety Inventory and Beck Depression Inventory were sent to the families. Data were analysed using descriptives, t tests and multivariable analysis. Results were compared with norm groups and reference samples.

RESULTS: The questionnaires were returned by 40 patients, 38 mothers and 33 fathers. The mean HRQoL was M = 80.4, comparable to children with asthma (M = 80.2) and diabetes (M = 79.5). Self-reported psychological well-being was comparable to the norm group of Swedish school children, and was significantly higher than a clinical sample. Factors negatively influencing children's HRQoL and psychological well-being were identified. The parents' self-reports of anxiety and depression were comparable to non-clinical samples.

CONCLUSIONS: Although children and adolescents with VACTERL association reported similar HRQoL to those of European children with chronic conditions, their psychological well-being was comparable to Swedish school children in general. Nevertheless, some individuals among both children and parents were in need of extra support. This attained knowledge is valuable when counselling parents regarding the prognosis for children with VACTERL association.

Ort, förlag, år, upplaga, sidor
2019.
Nyckelord [en]
Congenital malformations, VACTERL association, Health-Related Quality of Life, psychological well-being, children and adolescents, parents
Nationell ämneskategori
Pediatrik
Identifikatorer
URN: urn:nbn:se:uu:diva-390576DOI: 10.1007/s11136-019-02364-wPubMedID: 31741214OAI: oai:DiVA.org:uu-390576DiVA, id: diva2:1343262
Tillgänglig från: 2019-08-16 Skapad: 2019-08-16 Senast uppdaterad: 2020-01-08Bibliografiskt granskad
Ingår i avhandling
1. Living with VACTERL association: From the perspectives of children, adolescents and their parents
Öppna denna publikation i ny flik eller fönster >>Living with VACTERL association: From the perspectives of children, adolescents and their parents
2019 (Engelska)Doktorsavhandling, sammanläggning (Övrigt vetenskapligt)
Abstract [en]

VACTERL association is a rare and complex congenital condition often requiring repeated surgery and entailing various physical sequelae. Knowledge is scarce regarding experiences of the health condition and health care, need of support at school, health-related quality of life (HRQoL), and psychological well-being in children, adolescents and their parents.

This thesis aims to investigate various aspects of living with VACTERL association, from the perspectives of children, adolescents and their parents.

Ten children aged five to eight years were interviewed using the computer-assisted interview technique In My Shoes (Study I). They expressed awareness of their health history and felt proud but also different due to physical dysfunction. While happy to meet familiar staff in the hospital they voiced worries about medical procedures.

The nineteen parents interviewed described crisis reactions on the discovery of malformations in their child (Study II). Parental involvement in care was reported from the initial hospital admission until taking responsibility for treatments at home. Eventually the health condition became integrated in everyday life. Insufficient emotional and limited medical support were reported. Various levels of professionalism among healthcare professionals and discrepancies concerning knowledge and experience between the tertiary and local hospitals were described.

In ten evaluated pre-school children (Study III), intelligence measured by Wechsler-scales was within the normal range. Eight children had attention difficulties and two were later diagnosed with attention deficit hyperactivity disorder (ADHD). All children had physical dysfunctions affecting their nutrition, bowel or bladder functions. All needed extra support and adjustments at school.

Forty children and adolescents responded to validated questionnaires of DISABKIDS and Beck inventories (Study IV). The HRQoL was comparable to European children with chronic conditions. Their psychological well-being was similar to that of Swedish school children and significantly better than that of a clinical sample. Self-reported anxiety and depression in 38 mothers and 33 fathers were comparable to non-clinical samples.

In conclusion, regular follow-up by multi-professional team with continuity is crucial to optimise the physical function in children with VACTERL, to identify those in need of extra support at school and to detect reduced psychological well-being in children and parents. Fear of medical procedures may be reduced by carefully providing information and individual care strategies. For the parents psychological processing, support from medical experts and peers is essential to achieve self-confidence and adaptation. Transfer of knowledge and information between multi-professional teams at the local and tertiary hospitals could be improved by the use of video sessions.

Ort, förlag, år, upplaga, sidor
Uppsala: Acta Universitatis Upsaliensis, 2019. s. 72
Serie
Digital Comprehensive Summaries of Uppsala Dissertations from the Faculty of Medicine, ISSN 1651-6206 ; 1591
Nyckelord
Congenital malformations, Experiences of health care, Health-related quality of life, Neurodevelopmental function, Physical dysfunction, Psychological well-being, VACTERL association
Nationell ämneskategori
Medicin och hälsovetenskap
Forskningsämne
Barnkirurgi
Identifikatorer
urn:nbn:se:uu:diva-389781 (URN)978-91-513-0731-2 (ISBN)
Disputation
2019-10-11, Rosénsalen, entrance 95/96, NBV, Akademiska sjukhuset, Uppsala, 13:15 (Svenska)
Opponent
Handledare
Tillgänglig från: 2019-09-19 Skapad: 2019-08-21 Senast uppdaterad: 2019-10-15

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