Health-Related Quality of Life in Pediatric Surgical Patients and their Caretakers

The primary aim of pediatric surgical care is to achieve operative solutions that last for lifetime and to ensure the best possible outcomes for the patient and his/her family. Traditionally, quality assessment in pediatric surgery has been based on unambiguous clinical and functional outcome parameters. Due to the recent advances of operative and medical care, infants with neonatal malformations commonly survive into adulthood. However, along with increased survival rates, long-term sequelae may persist, indicating the need for continuous follow-up of the patients. In the last decade, the focus of follow-up research has moved from clinical and functional parameters toward the patients' and parents' perception of the child's health condition and health-related treatment. Patient-reported outcomes (PROs) have become a major matter of academic interest, illuminating the patient's perspective in pediatric surgical research. PROs comprise various constructs like quality of life (QoL), health-related quality of life (HrQoL), or functional health status. A multitude of validated instruments are available to assess these outcomes at different patient ages.

We appreciate the opportunity to edit this special Review Block of the European Journal of Pediatric Surgery as a multidisciplinary team of guest editors, enlightening the concepts, applications, and results of PROs in pediatric surgical patients, from childhood into adulthood. We have chosen to focus on HrQoL as one of the most acknowledged PRO's in different congenital malformations, representing the pediatric surgical core entities. Fortunately, we were able to gather distinguished experts from different fields of pediatric surgery and related subspecialties for this review block, which therefore comprises seven high-quality peer-reviewed articles. We feel honored, to give the readers of the journal an interesting and fruitful outlook on what is known about HrQoL in pediatric surgical congenital malformations and what is recommended for future research.

The review block is initiated by Blömeke et al[1] who give a comprehensive review article on what aspects of HrQoL are important to the pediatric surgeon. They describe the concept of HrQoL, its methodological challenges, and the application of HrQoL instruments in pediatric surgical practice and health services research. The importance to accumulate and critically appraise knowledge about HrQoL, and to broaden the understanding of the child's health status, has been emphasized in this article in a clear way. The authors conclude that, from the perspective of a pediatric surgeon, knowledge about HrQoL of the children and their parents is essential, since it might influence treatment decisions and facilitate patient–physician communication.

In the second article of this issue, Witt et al[2] put a distinct focus on the HrQoL of parents, guardians, and other caregivers of patients with pediatric surgical congenital malformations. The diagnosis of a congenital malformation with necessary surgical treatment and possibly complex aftercare may represent an emotionally stressful phase in a parent's life which may impact on parental HrQoL. The authors review the available literature on this topic and conclude with the clear perspective that since studies are scarce, further research is needed. It is recommended to utilize both generic and disease-specific HrQoL instruments and to measure HrQoL of parents and siblings in longitudinal studies to better understand the family as a system. This implies not only looking at the patient but involving the whole family.

In addition to those two generic/chronic opening papers, five further articles have been successfully invited and completed. These investigate the recent literature regarding HrQoL in specific congenital malformations from the pediatric surgical repertoire that we all deal with every day. From our point of view, the articles are particularly valuable to, for example, counsel parents before and after birth of their affected children on how a certain malformation might influence the life of the patient and its family.

Dellenmark-Blom et al[3] have reviewed the current literature on HrQoL after repair of esophageal atresia (EA). They have found that specific clinical subgroups of children with EA present with impaired HrQoL and that digestive symptomology may influence HrQoL negatively in children and adults.

In the article of Feng et al[4] the recent literature on HrQoL after surgical correction of anorectal malformations (ARMs) and Hirschsprung’s disease (HD) was reviewed. The authors found that children with ARM/HD are at risk for impaired HrQoL, in both physical and psychosocial aspects. Age, gender, severity of disease, surgical technique, as well as their caregivers may influence HrQoL in children with ARMs/HD. With age, physical HrQoL seems to improve but psychosocial HrQoL may decrease.

Derraugh and colleagues[5] reviewed data on HrQoL of patients with congenital diaphragmatic hernia (CDH). It is concluded from the reviewed literature that CDH patients may have an HrQoL equivalent to the general population in the long-term, despite the severity of the malformation in the neonatal period.

In the review article of Holmdahl and coauthors,[6] the current literature on HrQoL following repair of bladder exstrophy–epispadias complex (BEEC) is reviewed in relationship to important clinical outcome parameters. The authors conclude that overall HrQoL in individuals born with the BEEC appears good, but urinary incontinence and sexual dysfunction may have a negative impact on HrQoL.

Finally, the article of Rodijk et al summarizes the recent data on HrQoL in patients with biliary atresia (BA) and contains a remarkable amount of original data from their own center.[7] It is concluded that children with BA are at risk of impaired HrQoL and that special attention is warranted for children with adverse medical events and special education.

We believe that this special issue of EJPS will give a decent overview of the current status of HrQoL in pediatric surgical patients and the scientific and clinical perspectives for future research. We are convinced that the high-quality articles will further promote the application of PRO's in the pediatric surgical community. We would like to thank the editorial board to have been given the opportunity to guest edit this review block, and we wish the esteemed readers of EJPS enlightening and motivating moments reading these articles.

• References

• 1 Bloemeke J, Witt S, Bullinger M, Dingemann J, Dellenmark-Blom M, Quitmann J. Health-related quality of life assessment in children and their families: aspects of importance to the pediatric surgeon. Eur J Pediatr Surg 2020; 30 (03) 232-238
• 2 Witt S, Blömeke J, Bullinger M, Dingemann J, Quitmann J. 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 2020; 30 (03) 225-231
• 3 Dellenmark-Blom M, Quitmann J, Dingemann C. Health-related quality of life in patients after repair of esophageal atresia: a review of current literature. Eur J Pediatr Surg 2020; 30 (03) 239-250
• 4 Feng X, Lacher M, Quitmann J, Witt S, Witvliet MJ, Mayer S. Health-related quality of life and psychosocial morbidity in anorectal malformation and Hirschsprung's disease. Eur J Pediatr Surg 2020; 30 (03) 279-288
• 5 Derraugh G, Lum Min SA, Keijzer R. Long-term health-related quality of life in survivors of congenital diaphragmatic hernia. Eur J Pediatr Surg 2020; 30 (03) 273-278
• 6 Holmdahl G, Dellenmark-Blom M, Nordenskjöld A, Sjöström S. Health-related quality of life in patients with the bladder exstrophy-epispadias complex and relationship to incontinence and sexual factors: a review of the recent literature. Eur J Pediatr Surg 2020; 30 (03) 251-260
• 7 Rodijk LH, Schins EMW, Witvliet MJ, Verkade HJ, de Kleine RH, Hulscher JBF, Bruggink JLM. Health-related quality of life in biliary atresia patients with native liver or transplantation. Eur J Pediatr Surg 2020; 30 (03) 261-272