Predictors of Parent-Reported Health-Related Quality of Life in Young Children with Early Brain Damage and Severe Motor Dysfunction
Abstract
1. Introduction
2. Materials and Methods
2.1. Study Design
2.2. Recruitment and Evaluation
2.3. Participants
2.4. Measures
2.5. The ICF-CY Framework and Selection of Outcome Measures
2.6. Statistical Analysis
2.7. Ethics
2.8. Use of GenAI in Writing
3. Results
3.1. Participants
3.2. Clinical Characteristics
3.3. Regression Analyses
3.3.1. Adaptive Skills
3.3.2. Gross Motor Function
3.3.3. Communicative Abilities
4. Discussion
4.1. Interpretation of Findings in Relation to Previous Research
4.1.1. Adaptive Skills
4.1.2. Gross Motor Function and Postural Control
4.1.3. Communicative Abilities
4.2. Underlying Mechanisms and Interpretations
4.3. Strengths and Limitations
4.4. Clinical Implications and Future Research
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Acknowledgments
Conflicts of Interest
Appendix A
Appendix A.1
Measurement | Included Total N = 65 | Excluded Total N = 24 |
---|---|---|
CPCHILD Total score | 65 54.4 (11.9) | |
PEDI-CAT Self-care | 65 42.2 (6.0) | 23 41.6 (4.5) |
PED-ICAT Mobility | 65 47.0 (7.0) | 23 48.0 (5.3) |
PEDI-CAT Soc/cog | 65 52.8 (7.1) | 23 51.2 (5.4) |
PEDI-CAT Responsibility | 56 33.1 (7.0) | 15 31.4 (5.4) |
FOCUS Total score | 65 102.8 (47.1) | 23 83.7 (27.26) |
GMFM-66 score | 65 34.9 (11.9) | 24 25.5 (10.7) |
Age | 65 45.6 (16.8) | 24 46.1 (12.5) |
Non-ambulant | 53/65 (81.5) | 21/24 (87.5) |
Appendix A.2
Regression Coefficient | ||||
---|---|---|---|---|
Covariate | N | Estimate | 95% CI | p-Value |
PEDI-CAT Self-care | ||||
Unadjusted | 65 | 1.15 | 0.75–1.56 | <0.001 |
Adjusted for | ||||
Age | 65 | 1.22 | 0.80–1.64 | <0.001 |
Ambulant | 65 | 1.09 | 0.65–1.63 | <0.001 |
Age and ambulant | 65 | 1.11 | 0.70–1.61 | <0.001 |
PEDI-CAT Mobility | ||||
Unadjusted | 65 | 0.97 | 0.62–1.32 | <0.001 |
Adjusted for | ||||
Age | 65 | 1.02 | 0.66–1.38 | <0.001 |
Ambulant | 65 | 1.03 | 0.60–1.66 | <0.001 |
Age and ambulant | 65 | 1.07 | 0.63–1.52 | <0.001 |
PEDI-CAT Social/Cognitive | ||||
Unadjusted | 65 | 0.68 | 0.30–1.07 | <0.001 |
Adjusted for | ||||
Age | 65 | 0.71 | 0.31–1.12 | <0.001 |
Ambulant | 65 | 0.59 | 0.20–0.99 | 0.004 |
Age and ambulant | 65 | 0.62 | 0.21–1.04 | 0.004 |
PEDI-CAT Responsibility | ||||
Unadjusted | 41 | 0.83 | 0.29–1.37 | 0.004 |
Adjusted for | ||||
Age | 41 | 0.83 | 0.28–1.38 | 0.004 |
Ambulant | 41 | 0.75 | 0.19–1.31 | 0.011 |
Age and ambulant | 41 | 0.75 | 0.17–1.33 | 0.013 |
FOCUS total score | ||||
Unadjusted | 65 | 0.16 | 0.06–0.17 | <0.001 |
Adjusted for | ||||
Age | 65 | 0.16 | 0.06–0.17 | <0.001 |
Ambulant | 65 | 0.11 | 0.05–0.16 | <0.001 |
Age and ambulant | 65 | 0.11 | 0.05–0.17 | <0.001 |
GMFM-66 | ||||
Unadjusted | 65 | 0.54 | 0.33–0.75 | <0.001 |
Adjusted for | ||||
Age | 65 | 0.55 | 0.34–0.77 | <0.001 |
Ambulant | 65 | 0.60 | 0.32–0.87 | <0.001 |
Age and ambulant | 65 | 0.61 | 0.32–0.89 | <0.001 |
ECAB Total score | ||||
Unadjusted | 65 | 0.45 | 0.21–0.70 | <0.001 |
Adjusted for | ||||
Age | 65 | 0.46 | 0.21–0.71 | <0.001 |
Ambulant | 65 | 0.39 | 0.11–0.68 | 0.008 |
Age and ambulant | 65 | 0.40 | 0.11–0.69 | 0.008 |
References
- Van Druten, V.P.; Bartels, E.A.; van de Mheen, D.; de Vries, E.; Kerckhoffs, A.P.M.; Venrooij, L.M.W.N.-V. Concepts of health in different contexts: A scoping review. BMC Health Serv. Res. 2022, 22, 389. [Google Scholar] [CrossRef]
- Dodds, C.B.; Bjornson, K.F.; Sweeney, J.K.; Narayanan, U.G. The effect of supported physical activity on parental-reported health-related quality of life in children with medical complexity. J. Pediatr. Rehabil. Med. 2015, 8, 83–95. [Google Scholar] [CrossRef]
- Vente, T. How Do you Focus on Quality of Life When Kids Cannot Go to School or Outside? Curr. Pediatr. Rep. 2021, 9, 37–45. [Google Scholar] [CrossRef]
- Colver, A. Quality of Life Outcomes. In Measures for Children with Developmental Disabilities; Majnemer, A., Ed.; MAC Keith Press: Montreal, QC, Canada, 2012; pp. 503–504. [Google Scholar]
- Narayanan, U.G.; Weir, S.; Fehlings, D. The CPCHILD Manual & Interpretation Guide. 2017. Available online: https://lab.research.sickkids.ca/pscoreprogram/wp-content/uploads/sites/72/2017/09/6573-CPCHILD_manual.pdf (accessed on 6 June 2025).
- Hollung, S.J.; Andersen, G.L. NORCP Årsrapport; Vestfold Hospital Norway: Tønsberg, Norway, 2024; pp. 23–36. Available online: www.siv.no (accessed on 6 June 2025).
- Majnemer, A.; Shevell, M.; Rosenbaum, P.; Law, M.; Poulin, C. Determinants of life quality in school-age children with cerebral palsy. J. Pediatr. 2007, 151, 470–475.e3. [Google Scholar] [CrossRef]
- Milićević, M. Functional and environmental predictors of health-related quality of life of school-age children with cerebral palsy: A cross-sectional study of caregiver perspectives. Child Care Health Dev. 2023, 49, 62–72. [Google Scholar] [CrossRef] [PubMed]
- World Health Organization. International Classification of Functioning, Disability, and Health: Children & Youth Version: ICF-CY; World Health Organization: Geneva, Switzerland, 2007. [Google Scholar]
- Elema, A.; Zalmstra, T.A.L.; Boonstra, A.M.; Narayanan, U.G.; Reinders-Messelink, H.A.; Putten, A.A.J.V.D. Pain and hospital admissions are important factors associated with quality of life in nonambulatory children. Acta Paediatr. 2016, 105, e419–e425. [Google Scholar] [CrossRef] [PubMed]
- Ramstad, K.; Jahnsen, R.; Skjeldal, O.H.; Diseth, T.H. Mental health, health related quality of life and recurrent musculoskeletal pain in children with cerebral palsy 8–18 years old. Disabil. Rehabil. 2012, 34, 1589–1595. [Google Scholar] [CrossRef]
- Ramstad, K.; Jahnsen, R.B.; Terjesen, T. Severe hip displacement reduces health-related quality of life in children with cerebral palsy. Acta Orthop. 2017, 88, 205–210. [Google Scholar] [CrossRef]
- Jon, S. Program intensive Habilitation (PIH) for Young Children With early Brain Damage (PIH Multi). 2025. Available online: https://clinicaltrials.gov/study/NCT05093777?term=NCT05093777&rank=1#more-information (accessed on 6 June 2025).
- Skranes, J.; Vestrheim, I. PIH Multi Study. 2025. Available online: https://www.sshf.no/kliniske-studier/pih-multi/ (accessed on 6 June 2025).
- Sørensen, K.; Johnsen, S. The PIH Multi Program. 2024. Available online: https://www.sshf.no/behandlinger/pih-multi/ (accessed on 6 June 2025).
- Narayanan, U.G.; Fehlings, D.; Weir, S.; Knights, S.; Kiran, S.; Campbell, K. Initial development and validation of the Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD). Dev. Med. Child Neurol. 2006, 48, 804–812. [Google Scholar] [CrossRef]
- Pettersson, K.; Bjerke, K.M.; Jahnsen, R.; Öhrvik, J.; Rodby-Bousquet, E. Psychometric evaluation of the Scandinavian version of the caregiver priorities and child health index of life with disabilities. Disabil. Rehabil. 2019, 41, 212–218. [Google Scholar] [CrossRef]
- Haley, S.M.; Coster, W.J.; Dumas, H.M.; Fragala-Pinkham, M.A.; Moed, R. PEDI-CAT: Development, Standardization and Administration Manual; Boston University: Boston, MA, USA, 2012; pp. 648–657. [Google Scholar]
- Dumas, H.M.; Fragala-Pinkham, M.A.; Rosen, E.L.; O’bRien, J.E. Construct validity of the pediatric evaluation of disability inventory computer adaptive test (PEDI-CAT) in children with medical complexity. Disabil. Rehabil. 2017, 39, 2446–2451. [Google Scholar] [CrossRef] [PubMed]
- Jahnsen, R.; Klevberg, G.L.; Frøslie, K.F.; Berg, M.; Hussain, A. Evaluation of Disability Inventory Computer Adaptive Test (PEDI-CAT) Norwegian Version; Pearson Clinical & Talent Assessment, 2017. [Google Scholar]
- Hansen, T.N.; Christensen, K.B.; Stahlhut, M.; Ketelaar, M.; Klevberg, G.L.; Laursen, L.B.; Kristensen, M.R.; Jahnsen, R.; Hansen, T. Use of the Pediatric Evaluation of Disability Inventory—Computer Adaptive Test in Denmark, the Netherlands, and Norway. Dev. Med. Child Neurol. 2025. [Google Scholar] [CrossRef] [PubMed]
- Russel, D.J.; Wright, M.; Rosenbaum, P.L.; Avery, L.M. Ross Motor Function Measure (GMFM-66 & GMFM-88) User’s Manual, Clinics in Developmental Medicine, 3rd ed.; Mac Keith Press: London, UK, 2021. [Google Scholar]
- Russell, D.J.; Avery, L.M.; Rosenbaum, P.L.; Raina, P.S.; Walter, S.D.; Palisano, R.J. Improved scaling of the gross motor function measure for children with cerebral palsy: Evidence of reliability and validity. Phys. Ther. 2000, 80, 873–885. [Google Scholar] [CrossRef] [PubMed]
- Shiggavi, A.; Kb, K.; Mahaveer, N. Effects of Treadmill Walking and Overground Walking in Young Children with Non-progressive Neurodevelopmental Disorders. Cureus 2024, 16, e71495. [Google Scholar] [CrossRef]
- McCoy, S.W.; Bartlett, D.J.; Yocum, A.; Jeffries, L.; Fiss, A.L.; Chiarello, L.; Palisano, R.J. Development and validity of the early clinical assessment of balance for young children with cerebral palsy. Dev. Neurorehabil. 2014, 17, 375–383. [Google Scholar] [CrossRef]
- LaForme Fiss, A.; McCoy, S.W.; Bartlett, D.; Avery, L.; Hanna, S.E.; On Track Study Team. Developmental Trajectories for the Early Clinical Assessment of Balance by Gross Motor Function Classification System Level for Children with Cerebral Palsy. Phys. Ther. 2019, 99, 217–228. [Google Scholar] [CrossRef]
- Inoue, T.; Izumi, H.; Nishibu, H.; Himuro, N. Reliability, validity, and minimal clinically important differences of the Japanese version of the early clinical assessment of balance in children with cerebral palsy. Disabil. Rehabil. 2022, 44, 7283–7289. [Google Scholar] [CrossRef]
- Thomas-Stonell, N.L.; Oddson, B.; Robertson, B.; Rosenbaum, P.L. Development of the FOCUS (Focus on the Outcomes of Communication Under Six), a communication outcome measure for preschool children. Dev. Med. Child Neurol. 2010, 52, 47–53. [Google Scholar] [CrossRef]
- Eadie, T.L.; Yorkston, K.M.; Klasner, E.R.; Dudgeon, B.J.; Deitz, J.C.; Baylor, C.R.; Miller, R.M.; Amtmann, D. Measuring communicative participation: A review of self-report instruments in speech-language pathology. Am. J. Speech-Language Pathol. 2006, 15, 307–320. [Google Scholar] [CrossRef]
- Washington, K.; Thomas-Stonell, N.; Oddson, B.; McLeod, S.; Warr-Leeper, G.; Robertson, B.; Rosenbaum, P. Construct validity of the FOCUS© (Focus on the Outcomes of Communication Under Six): A communicative participation outcome measure for preschool children. Child Care Health Dev. 2013, 39, 481–489. [Google Scholar] [CrossRef]
- Canchild.ca. Focus on the Outcomes of Communication Under Six. 2025. Available online: https://canchild.ca/shop/30-focus-34/ (accessed on 6 June 2025).
- Hollingshead, A.B. Four factor index of social status. Yale J. Sociol. 2011, 8, 21–51. [Google Scholar]
- Schiariti, V.; Klassen, A.F.; Cieza, A.; Sauve, K.; O’DOnnell, M.; Armstrong, R.; Mâsse, L.C. Comparing contents of outcome measures in cerebral palsy using the international classification of functioning (ICF-CY): A systematic review. Eur. J. Paediatr. Neurol. 2014, 18, 1–12. [Google Scholar] [CrossRef]
- Thompson, S.V.; Cech, D.J.P.; Cahill, S.M.P.; Krzak, J.J. Linking the Pediatric Evaluation of Disability Inventory-Computer Adaptive Test (PEDI-CAT) to the International Classification of Function. Pediatr. Phys. Ther. 2018, 30, 113–118. [Google Scholar] [CrossRef] [PubMed]
- Fayed, N.; Klassen, A.; Schiariti, V. Health Status Instruments. In Measures of Children with Developmental Disabilities—An ICF-CY Approach; Majnemer, A., Ed.; Mac Keith Press: Montreal, QC, Canada, 2012; pp. 487–488. [Google Scholar]
- Wang, H.-Y.; Yang, Y.H. Evaluating the Responsiveness of 2 Versions of the Gross Motor Function Measure for Children with Cerebral Palsy. Arch. Phys. Med. Rehabil. 2006, 87, 51–56. [Google Scholar] [CrossRef] [PubMed]
- Thomas-Stonell, N.; Robertson, B.; Walker, J.; Oddson, B.; Washington, K.; Rosenbaum, P. FOCUS©: Focus on the Outcomes of Communication Under Six-Focus Manual; Holland Bloorview Kids Rehabilitation Hospital: Toronto, ON, Canada, 2012. [Google Scholar]
- Kraus, T.; Gegenleitner, K.; Svehlik, M.; Novak, M.; Steinwender, G.; Singer, G. Long-term therapy with intrathecal baclofen improves quality of life in children with severe spastic cerebral palsy. Eur. J. Paediatr. Neurol. 2017, 21, 565–569. [Google Scholar] [CrossRef] [PubMed]
- Vaillant, E.; Oostrom, K.J.; Beckerman, H.; Vermeulen, J.R.; Buizer, A.I.; Geytenbeek, J.J.M. Convergent validity of functional communication tools and spoken language comprehension assessment in children with cerebral palsy. Int. J. Lang. Commun. Disord. 2022, 57, 963–976. [Google Scholar] [CrossRef]
- van Stralen, R.A.; Roelen, M.C.R.; Moerman, S.; Witbreuk, M.M.E.H.; Witlox, M.A.; Ten Ham, A.; Eygendaal, D.; Reijman, M.; Tolk, J.J. GUIDANCE study: Guided growth of the proximal femur to prevent further hip migration in patients with cerebral palsy-study protocol for a multicentre randomised controlled trial. BMJ Open 2024, 14, e091073. [Google Scholar] [CrossRef]
- Wong, C.; Westphall, I.; Michelsen, J.S. Measuring Effects on Pain and Quality of Life after Abobotulinum Toxin A Injections in Children with Cerebral Palsy. Toxins 2022, 14, 43. [Google Scholar] [CrossRef]
Children n = 65 | ||
---|---|---|
Age in months | Mean (SD) | 45.6 (16.8) |
Min/max | 15.8/83.5 | |
Sex | Boys | 33 (51) |
Girls | 32 (49) | |
Diagnosis | CP | 35 (54) |
Other (a) | 30 (46) | |
Mobility | Ambulant | 13 (20) |
Non-ambulant | 52 (80) | |
Child impairments, as reported by parents (b) | Epilepsy | 22 (34) |
Vision | 24 (37) | |
Hearing | 7 (11) | |
Respiration | 18 (28) | |
Communication | 54 (83) | |
Cognition | 33 (51) | |
Nutrition | 38 (59) | |
Sleep | 26 (40.0) | |
Pain | 6 (9) | |
Parents n = 65 | ||
Age | Mean (SD) | 34.6 (5.6) |
Education (c) | ≤10 years | 1 (2) |
11–13 years | 19 (29) | |
Diploma/ Bachelor | 21 (32) | |
Master | 20 (31) | |
Doctoral degree | 4 (6) | |
Employment (d) | Adm. Management | 1 |
Academics | 15 | |
Technicians, lower academics | 4 | |
Sales, service, office, care | 33 | |
Farming, forestry, fishing, craftsmen transport | 3 | |
Student/childcare | 11 | |
Other | 4 | |
Born outside Norway | 9 |
Measurement | N | Mean (SD) ** | Min–Max |
---|---|---|---|
CPCHILD Total score | 65 | 54.4 (11.9) | 26.7–83.3 |
PEDI-CAT Self-care | 65 | 42.3 (6.0) | 30.0–56.0 |
PEDI-CAT Mobility | 65 | 47.0 (7.0) | 33.0–63.0 |
PEDI-CAT Social/Cognitive | 65 | 52.8 (7.1) | 38.0–66.0 |
PEDI-CAT Responsibility | 41 * | 34.1 (7.0) | 25.0–49.0 |
GMFM-66 Total score | 65 | 34.9 (11.9) | 10.4–65.4 |
ECAB Total score | 65 | 25.5 (10.7) | 4.0–44.0 |
FOCUS Total score | 65 | 102.8 (47.1) | 52.5–228.0 |
Covariate | Regression Coefficient (Estimate and CI) | p-Value | Predicted Change * (Estimate and CI) |
---|---|---|---|
PEDI-CAT Self-care | 1.15 (0.75–1.56) | <0.001 | 6.90 (4.50–9.36) |
PEDI-CAT Mobility | 0.97 (0.62–1.32) | <0.001 | 6.79 (4.34–9.24) |
PEDI-CAT Social/Cognitive | 0.68 (0.20–1.07) | <0.001 | 4.83 (4.50–9.24) |
PEDI-CAT Responsibility | 0.83 (0.29–1.37) | 0.004 | 5.81 (2.13–7.60) |
GMFM-66 Total score | 0.54 (0.33–0.75) | <0.001 | 6.43 (3.93–8.93) |
ECAB Total score | 0.45 (0.21–0.70) | <0.001 | 4.82 (2.25–7.49) |
FOCUS Total score | 0.12 (0.07–0.17) | <0.001 | 5.65 (2.82–8.01) |
Disclaimer/Publisher’s Note: The statements, opinions and data contained in all publications are solely those of the individual author(s) and contributor(s) and not of MDPI and/or the editor(s). MDPI and/or the editor(s) disclaim responsibility for any injury to people or property resulting from any ideas, methods, instructions or products referred to in the content. |
© 2025 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
Share and Cite
Johnsen, S.; Sørensen, K.; Skranes, J.S.; Vestrheim, I.E.; Modahl, M.G.; Jahnsen, R.B.; Stadskleiv, K.; Hansen, G.; Lydersen, S.; Sæther, R. Predictors of Parent-Reported Health-Related Quality of Life in Young Children with Early Brain Damage and Severe Motor Dysfunction. J. Clin. Med. 2025, 14, 7054. https://doi.org/10.3390/jcm14197054
Johnsen S, Sørensen K, Skranes JS, Vestrheim IE, Modahl MG, Jahnsen RB, Stadskleiv K, Hansen G, Lydersen S, Sæther R. Predictors of Parent-Reported Health-Related Quality of Life in Young Children with Early Brain Damage and Severe Motor Dysfunction. Journal of Clinical Medicine. 2025; 14(19):7054. https://doi.org/10.3390/jcm14197054
Chicago/Turabian StyleJohnsen, Siri, Kristian Sørensen, Jon Sverre Skranes, Ida Eline Vestrheim, Mette Gro Modahl, Reidun Birgitta Jahnsen, Kristine Stadskleiv, Gry Hansen, Stian Lydersen, and Rannei Sæther. 2025. "Predictors of Parent-Reported Health-Related Quality of Life in Young Children with Early Brain Damage and Severe Motor Dysfunction" Journal of Clinical Medicine 14, no. 19: 7054. https://doi.org/10.3390/jcm14197054
APA StyleJohnsen, S., Sørensen, K., Skranes, J. S., Vestrheim, I. E., Modahl, M. G., Jahnsen, R. B., Stadskleiv, K., Hansen, G., Lydersen, S., & Sæther, R. (2025). Predictors of Parent-Reported Health-Related Quality of Life in Young Children with Early Brain Damage and Severe Motor Dysfunction. Journal of Clinical Medicine, 14(19), 7054. https://doi.org/10.3390/jcm14197054