Patient-driven healthcare recommendations for adults with esophageal atresia and their families

Kate, Chantal A. ten; Rietman, André B.; Kamphuis, Lieke; Gischler, Saskia J.; Lee, Demi; Fruithof, JoAnne; Wijnen, René; Spaander, Manon

doi:10.1016/j.jpedsurg.2020.12.024

Cited by 12 publications

(9 citation statements)

References 38 publications

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“…This team can help monitor and treat the child’s symptoms properly, but also provide practical and educational support to families of children with EA and facilitate a good adaptational process. This would be in line with patient-driven health-care recommendations for adults with EA and their families [ 48 ]. Moreover, since our study results imply that family impact in children with digestive symptoms and feeding difficulties is worse, these families may benefit from having targeted support early after the child is born with EA.…”

Section: Discussionmentioning

confidence: 54%

Factors of family impact in a Swedish–German cohort of children born with esophageal atresia

Dellenmark‐Blom

Abrahamsson

Dingemann

et al. 2022

Orphanet J Rare Dis

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Background After repair of esophageal atresia (EA), childhood survivors commonly present with digestive and respiratory morbidity, and around 55% have associated anomalies. Although it is known that these problems can reduce health-related quality of life in children with EA, less is understood about the impact on the family. We aimed to identify factors related to family impact in children with EA. Methods One parent each of a child with EA (2–18 years) in 180 families from Sweden and Germany answered the PedsQL™ Family Impact Module as the dependent variable. The independent variables were the child’s parent-reported health-related quality of life as measured by PedsQL™ 4.0, current symptoms, school situation, and parent/family characteristics together with child clinical data from the medical records. Results Stepwise multivariable regression analysis showed a multifactorial model of the total family impact scores (R2 = 0.60), with independent factors being the child’s overall generic health-related quality of life, school-absence ≥ 1/month, severe tracheomalacia, a family receiving carer’s allowance, and a parent with no university/college education, p < 0.05. Logistic regression analysis showed that an increased number of symptoms in the child the preceding 4 weeks lowered the family impact scores; however, the child’s feeding (R2 = 0.35) and digestive symptoms (R2 = 0.25) explained more in the variation of scores than the child’s respiratory symptoms (R2 = 0.09), p < 0.0001. Conclusions Family functioning may be a contributing factor to the maintenance of child health. The study findings suggest multifactorial explanations to family impact in children with EA, which are essential when optimizing the support to these families in clinical and psychosocial practice. Future research should explore experiences of family impact from all family members’ perspectives and multicenter studies are warranted to understand better the effectiveness of psychosocial-educational interventions to families of children with EA.

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Section: Discussionmentioning

confidence: 54%

Factors of family impact in a Swedish–German cohort of children born with esophageal atresia

Dellenmark‐Blom

Abrahamsson

Dingemann

et al. 2022

Orphanet J Rare Dis

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“…As with previous research, the current study found that parents and adults experienced a profound cultural shift during transition and difference between the paediatric and adult healthcare (Coyne et al, 2019; Fegran et al, 2014; Heath et al, 2017; Lugasi et al, 2011). The differences between the paediatric and adult healthcare services included a lack of follow‐up and condition‐specific expertise in adult health services (ten Kate et al, 2021; Rabone & Wallace, 2021; Rare Disease UK, 2014).…”

Section: Discussionmentioning

confidence: 99%

“…However, the NHS is struggling to balance access with expertise, and the quality of care for children and young people with chronic and long‐term conditions, including transition services, is often variable (Wolfe et al, 2006). There is a lack of research on transition in OA/TOF (Krishnan et al, 2016), and a recent study showed that transition to adult health care is typically experienced as a shift from comprehensive paediatric care to a simple follow‐up by a GP (ten Kate et al, 2021).…”

Section: Introductionmentioning

confidence: 99%

“…Once reaching adulthood, perception of general and disease‐specific quality of life is generally reported to be positive (Ijsselstijn et al, 2016). However, some adults born with OA/TOF will continue to experience negative consequences from their birth defect (Connor et al, 2015; Roberts et al, 2016; ten Kate et al, 2021). The most common health‐related issues that require ongoing management (Smith, 2014) are dysphagia (Mousa et al, 2017); gastro‐oesophageal reflux disease; recurrent lung infections (Connor et al, 2015; ten Kate et al, 2021); and other respiratory symptoms (Gatzinsky et al, 2011).…”

Section: Introductionmentioning

confidence: 99%

“…However, some adults born with OA/TOF will continue to experience negative consequences from their birth defect (Connor et al, 2015; Roberts et al, 2016; ten Kate et al, 2021). The most common health‐related issues that require ongoing management (Smith, 2014) are dysphagia (Mousa et al, 2017); gastro‐oesophageal reflux disease; recurrent lung infections (Connor et al, 2015; ten Kate et al, 2021); and other respiratory symptoms (Gatzinsky et al, 2011). There is also evidence that some adults born with OA/TOF and their family members can struggle with ongoing psychosocial difficulties throughout life (Caplan, 2013; ten Kate et al, 2021; Losty & Thursfield, 2020).…”

Section: Introductionmentioning

confidence: 99%

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The lived experience of adults and parents: Transitioning from paediatric to adult health care with oesophageal atresia and tracheo‐oesophageal fistula

Haig‐Ferguson

Wallace

Davis

2022

Journal of Clinical Nursing

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Aim and Objective: To explore the experience of healthcare transition from paediatric to adult health care for adults born with oesophageal atresia and tracheo-oesophageal fistula (OA/TOF) and parents.Background: OA/TOF is a rare and chronic health condition that can require lifelong medical follow-up and management. There is evidence to suggest that transitioning from paediatric to adult health care can be problematic for people with rare and chronic conditions, including OA/TOF. The previous literature suggests that the experience of transitioning with a rare condition is more complex than transitioning with a common chronic condition. Design:The current study was a qualitative, cross-sectional, survey-based study. Methods: Data were collected through an online survey. Parents of children born with OA/TOF (n = 23) and adults born with OA/TOF (n = 16) were recruited through a UKbased OA/TOF patient charity.Data from six open-ended questions were analysed using a hybrid approach combining elements of inductive and deductive thematic analyses. Throughout the research process, the SRQR were followed.Results: Five themes were constructed during the analysis, reflecting the experience of parents and adults transitioning from paediatric to adult health care: thrown into the unknown; a cultural shift; stepping back and stepping up; 'no transition as such'; and living with uncertainty. Conclusions:The findings suggested that a formalised, managed healthcare transition is not commonly experienced by people born with OA/TOF and parents.Relevance to Clinical Practice: We recommend a formalised healthcare transition process in OA/TOF, including preparation for transition and having a named key worker to manage the multidisciplinary transition process. The results also highlighted the need for adults born with OA/TOF to have access to a specialist health service with knowledge and understanding of issues related to OA/TOF.

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Clinical outcome, quality of life, and mental health in long-gap esophageal atresia: comparison of gastric sleeve pull-up and delayed primary anastomosis

et al. 2023

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Introduction Pediatric surgeons have yet to reach a consensus whether a gastric sleeve pull-up or delayed primary anastomosis for the treatment of esophageal atresia (EA), especially of the long-gap type (LGEA) should be performed. Thus, the aim of this study was to evaluate clinical outcome, quality of life (QoL), and mental health of patients with EA and their parents. Methods Clinical outcomes of all children treated with EA from 2007 to 2021 were collected and parents of affected children were asked to participate in questionnaires regarding their Quality of Life (QoL) and their child’s Health-Related Quality of Life (HRQoL), as well as mental health. Results A total of 98 EA patients were included in the study. For analysis, the cohort was divided into two groups: (1) primary versus (2) secondary anastomosis, while the secondary anastomosis group was subdivided into (a) delayed primary anastomosis and (b) gastric sleeve pull-up and compared with each other. When comparing the secondary anastomosis group, significant differences were found between the delayed primary anastomosis and gastric sleeve pull-up group; the duration of anesthesia during anastomosis surgery (478.54 vs 328.82 min, p < 0.001), endoscopic dilatation rate (100% vs 69%, p = 0.03), cumulative time spent in intensive care (42.31 vs 94.75 days, p = 0.03) and the mortality rate (0% vs 31%, p = 0.03). HRQoL and mental health did not differ between any of the groups. Conclusion Delayed primary anastomosis or gastric sleeve pull-up appear to be similar in patients with long-gap esophageal atresia in many key aspects like leakage rate, strictures, re-fistula, tracheomalacia, recurrent infections, thrive or reflux. Moreover, HrQoL was comparable in patients with (a) gastric sleeve pull-up and (b) delayed primary anastomosis. Future studies should focus on the long-term results of either preservation or replacement of the esophagus in children.

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Patient-driven healthcare recommendations for adults with esophageal atresia and their families

Cited by 12 publications

References 38 publications

Factors of family impact in a Swedish–German cohort of children born with esophageal atresia

Factors of family impact in a Swedish–German cohort of children born with esophageal atresia

The lived experience of adults and parents: Transitioning from paediatric to adult health care with oesophageal atresia and tracheo‐oesophageal fistula

Clinical outcome, quality of life, and mental health in long-gap esophageal atresia: comparison of gastric sleeve pull-up and delayed primary anastomosis

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