Evidence map›Paper›PMID 27756070›Full record

ArticleNeonatology2017

Long-Gap Esophageal Atresia Is a Unique Entity within the Esophageal Atresia Defect Spectrum.

Sigrid Bairdain, David Zurakowski, Sara O Vargas, Nicole Stenquist, Molly McDonald, Meghan C Towne, David T Miller, Russell W Jennings, David B Kantor, Pankaj B Agrawal

Open access · greenAbstract read
In one paragraph

Article in Neonatology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed, 1 pooled it
1.3field-weighted citation impact, top 18% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

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

14 citing papers in PubMed, 1 synthesis or guideline pooled it, 41 citations in OpenAlex.

  1. Pooled it
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  4. Management of long-gap esophageal atresia.Translational pediatrics · 2024
    Review
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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

10 authors at 4 institutions in 2 countries.

Sigrid BairdainDepartment of Pediatric Surgery, Boston Children's Hospital, Harvard Medical School, Boston, Mass., USA.
David Zurakowski
Sara O Vargas
Nicole Stenquist
Molly McDonald
Meghan C Towne
David T Miller
Russell W Jennings
David B Kantor
Pankaj B Agrawal
Pain and Rehabilitation Medicine · USCenter for Discovery · USHarvard University · USInstitut thématique Génétique, génomique et bioinformatique · FR

Funding

Pediatric Critical Care Scientist Development ProgramK12HD047349 · NICHD · UTAH STATE HIGHER EDUCATION SYSTEM--UNIVERSITY OF UTAH · PI HEATHER T. KEENAN, Kevin M Watt · 2004 to 2026
$17.0M
Genome Sequence-Based Screening for Childhood Risk and Newborn IllnessU19HD077671 · NICHD · BRIGHAM AND WOMEN'S HOSPITAL · PI BEGGS, ALAN H., GREEN, ROBERT C. · 2013 to 2018
$6.1M
RESEARCH TRAINING IN PEDIATRIC EMERGENCY MEDICINET32HD040128 · NICHD · CHILDREN'S HOSPITAL BOSTON · PI Kenneth D. Mandl · 2001 to 2026
$5.0M
SPEG is Critical in Skeletal Muscle Development and FunctionR01AR068429 · NIAMS · BOSTON CHILDREN'S HOSPITAL · PI AGRAWAL, PANKAJ B · 2015 to 2019
$1.9M
NIAMS NIH HHS R01 AR068429NICHD NIH HHS K12 HD047349NICHD NIH HHS T32 HD040128NICHD NIH HHS U19 HD077671
6 · The paper itself

Abstract

backgroundLong-gap esophageal atresia (LGEA) may have clinical and syndromic presentations different from those of esophageal atresia (EA) that affects shorter segments of the esophagus (non-LGEA). This may suggest unique underlying developmental mechanisms.

objectivesWe sought to characterize clinical differences between LGEA and non-LGEA by carefully phenotyping a cohort of EA patients, and furthermore to assess molecular genetic findings in a subset of them.

methodsThis is a retrospective cohort study to systematically evaluate clinical and genetic findings in EA infants who presented at our institution over a period of 10 years (2005-2015).

resultsTwo hundred twenty-nine EA patients were identified, 69 (30%) of whom had LGEA. Tracheoesophageal fistula was present in most non-LGEA patients (158 of 160) but in only 30% of LGEA patients. The VACTERL association was more commonly seen with non-LGEA compared to LGEA (70 vs. 25%; p < 0.001). Further, trisomy 21 was more common in LGEA than in non-LGEA. 25% of LGEA patients had an isolated EA diagnosis without other anomalies, compared to <1% for non-LGEA. Chromosomal microarray analysis showed copy number variations (CNV) in 4 of 39 non-LGEA patients and 0 of 3 LGEA patients. A review of the ClinGen database showed that none of those CNV have been previously described with EA.

conclusionsLGEA represents a unique type of EA. Compared to non-LGEA, it is more likely to be an isolated defect and associated with trisomy 21. Further, it is less commonly seen with VACTERL anomalies. Our findings suggest the involvement of unique pathways that may be distinct from those causing non-LGEA.

Indexed as

DNA Copy Number VariationsAnal CanalBostonDown SyndromeEsophageal AtresiaEsophagusFemaleHeart Defects, CongenitalHumansInfantInfant, NewbornKidneyLimb Deformities, CongenitalMaleMicroarray AnalysisRetrospective Studies

Identifiers

PMID27756070
PMCPMC5290190
OpenAlexW2538220025

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

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.