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Multicenter Study
. 2025 Jun 26;184(7):450.
doi: 10.1007/s00431-025-06270-x.

Reference models for individualized assessment of cardiorespiratory fitness in children and adolescents with congenital heart disease: a retrospective multicentre study

Vibeke Klungerbo  1   2 , Asle Hirth  3 , Per Morten Fredriksen  4   5 , René Holst  6 , Elisabeth Edvardsen  7 , Henrik Holmstrøm  8 , Thomas Möller  9
Affiliations
Multicenter Study

Reference models for individualized assessment of cardiorespiratory fitness in children and adolescents with congenital heart disease: a retrospective multicentre study

Vibeke Klungerbo et al. Eur J Pediatr. .

Abstract

Cardiopulmonary Exercise Testing (CPET) is considered the gold standard assessment of peak oxygen uptake ( V ˙ O 2 peak), and consequently, cardiorespiratory fitness (CRF). Common practice in children with congenital heart diseases (CHD) is to compare individual test results to reference values in healthy children. This approach does not account for the diversity of heart defects and may also be demotivating for children with CHD. This retrospective multicentre study aimed to establish individualized CRF reference models specific to children with CHD, to facilitate a better understanding and management of their condition. A total of 1475 CPETs until exhaustion were performed in 943 children and adolescents at two hospitals in Norway. The children and adolescents were categorized into three groups: simple defects (VSD, ASD, CoA, LVOTO; n = 497, 38% female), moderate defects (TGA, Fallot; n = 299, 41% female), and univentricular defects with a Fontan circulation (n = 147, 42% female). Multivariable mixed-effects models with individuals as random intercepts were used to develop group-specific reference models for CRF. Estimated predictive models including covariance matrices for each group and outcome are provided, allowing for the generation of reference values with confidence intervals for clinical and research applications. Conclusion: Treadmill-based reference models for maximal and submaximal CRF variables in the most prevalent types of CHD are presented. Using individualized reference values has the potential to improve clinical decision-making for children with CHD and to enhance their sense of mastery. A web-based calculator is available for quick and easy clinical use.

Keywords: Cardiopulmonary Exercise Test; Congenital heart disease; Reference values; Treadmill.

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Conflict of interest statement

Declarations. Ethics approval: The authors assert that this retrospective, multicentre study complies with the Helsinki Declaration of 1975, as revised in 2008. Due to its retrospective nature and inclusion of minors, the study was reviewed and approved as a quality assurance project by the Data Protection Officer at Oslo University Hospital (project number 2016/14332). Data were anonymized, and the need for informed consent was waived in accordance with institutional guidelines. Data from Haukeland University Hospital in Bergen were categorized and anonymized before being merged into the database. Consent to participate: Since data were previously collected, the need for informed consent was waived. Consent to publish: Since data were previously collected, the need for informed consent was waived. Competing interests: EE has received personal consulting fees, honoraria, and expenses for presentations at education meetings from GlaxoSmithKline, AstraZeneca Norway, Birk NPC AS, and Chiesi Pharma AB. None of the other authors have competing interest to declare.

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