Evidence map›Paper›PMID 40826120›Full record

ArticleOrphanet journal of rare diseases2025

Experiences of symptom burden among young children born with esophageal atresia-tracheoesophageal fistula: a US focus group study.

Michaela Dellenmark-Blom, John Bennett, Rosella Micalizzi, Lianne Cole, Kaylee Woods, Lauren Cardoni, Leah Frain, Abdimajid Mohamed, Jessica Yasuda, Peter Ngo and 3 more

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In one paragraph

Article in Orphanet journal of rare diseases, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

13 authors.

Michaela Dellenmark-BlomDepartment of Pediatrics, Institute of Clinical Sciences, University of Gothenburg, 416 85, Gothenburg, Sweden. michaela.dellenmark.blom@gu.se.ORCID 0000-0002-1467-9427
John BennettEsophageal and Airway Treatment Center, Department of Pediatric General Surgery, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.
Rosella MicalizziEsophageal and Airway Treatment Center, Department of Pediatric General Surgery, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.
Lianne ColeEsophageal and Airway Treatment Center, Department of Pediatric General Surgery, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.
Kaylee WoodsEsophageal and Airway Treatment Center, Department of Pediatric General Surgery, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.
Lauren CardoniEsophageal and Airway Treatment Center, Department of Pediatric General Surgery, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.
Leah FrainEsophageal and Airway Treatment Center, Department of Pediatric General Surgery, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.
Abdimajid MohamedEsophageal and Airway Treatment Center, Department of Pediatric General Surgery, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.
Jessica YasudaDivision of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital, Boston, MA, USA.
Peter NgoDivision of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital, Boston, MA, USA.
Anke WidenmannEAT (Esophageal Atresia Global Support Groups), Sommerrainstr. 61, 70374, Stuttgart, Germany.
Graham SlaterEAT (Esophageal Atresia Global Support Groups), Sommerrainstr. 61, 70374, Stuttgart, Germany.
Benjamin ZendejasEsophageal and Airway Treatment Center, Department of Pediatric General Surgery, Boston Children's Hospital, Harvard Medical School, Boston, MA, USA.

Funding

ALF Grants from Region of Västra Götaland ALF GBG-942815ALF Grants from Region of Västra Götaland ALFGBG-978335
6 · The paper itself

Abstract

backgroundChildren born with esophageal atresia-tracheoesophageal fistula (EA-TEF) can suffer from aerodigestive morbidity that impairs their quality of life and can persist into adulthood. Ameliorating their symptom burden requires a thorough understanding of the symptom experiences that children have early in life. We aimed to explore parents' experiences of their children's aerodigestive symptom burden during the first years of life after being born with EA-TEF. This exploration also aimed to help determine whether a disease-specific measurement of symptom burden is needed.

methodFive standardized focus groups (FGs) with 22 parents of children with EA-TEF aged 6 months-7 years treated at a US tertiary pediatric surgical center were used to explore the children's symptom experiences. The FGs were audio-recorded, transcribed, content analyzed into what symptoms were expressed, together with their stated frequency, severity and relation to child distress.

resultsTwenty-two parents made 450 unique statements about their children's aerodigestive symptom experiences. The respiratory symptoms (n = 170 statements, n = 21 parents) included the following unique symptom expressions; Breathing difficulties (n = 21), Breathing sounds (n = 6), Cough (n = 17), Mucus problems (n = 22), Prone to frequent or severe respiratory infections (n = 20) and Reduced physical capacity/strength (n = 8). The digestive symptoms (181 statements, n = 21 parents) encompassed symptom expressions of Acid reflux/heartburn (n = 7), Hiccups (n = 1), Nausea (n = 2), Reflux/food coming up (n = 10), Stomach problems (n = 4), Swallowing difficulties (n = 24) and Vomiting/throw-up (n = 6). The descriptions of respiratory and digestive symptom experiences included a variation of symptom frequency, severity and child distress. Furthermore, feeding difficulties (99 statements, n = 22 parents) included the children's Food refusal (n = 8), Need for mealtime adjustment (n = 7), Selective/restrictive eating (n = 14) and Upset/stress with feeds (n = 10). Most parents (n = 20, 91%) described that their children had symptom experiences that spanned all three categories (respiratory and digestive symptoms, feeding difficulties).

conclusionsYoung children born with EA-TEF experience a significant symptom burden that can be reflected as a summative composite of the dimensions respiratory and digestive symptom frequency, severity and distress, in addition to feeding difficulties. This supports the need for a disease-specific measurement of symptom burden that is guided by the content and wording obtained directly from the parents' descriptions to help establish its content validity.

Indexed as

Esophageal AtresiaTracheoesophageal FistulaChildChild, PreschoolFemaleFocus GroupsHumansInfantMaleParentsQuality of LifeSymptom BurdenEsophageal atresiaPatient-reported outcomeRare diseaseSymptom burdenTracheobronchomalaciaTracheomalacia

Identifiers

PMID40826120
PMCPMC12362997

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.