Evidence map›Paper›PMID 36790509›Full record

ReviewPediatric cardiology2023

Sudden Cardiac Arrest During a Sedated Cardiac Magnetic Resonance Study in a Nonsyndromic Child with Evolving Supravalvar Aortic Stenosis Due to Familial ELN Mutation.

Dor Markush, Pedro A Sanchez-Lara, Katheryn Grand, Robert Wong, Ruchira Garg

Open access · hybridAbstract readCase ReportsReview
In one paragraph

Review in Pediatric cardiology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
1.0field-weighted citation impact, top 27% 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

2 citing papers in PubMed, 5 citations in OpenAlex.

  1. Article
  2. 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

5 authors at 2 institutions in 1 country.

Dor MarkushGuerin Family Congenital Heart Program, Smidt Heart Institute, Cedars-Sinai Medical Center, Los Angeles, CA, USA. Dor.Markush@cshs.org.
Pedro A Sanchez-LaraDepartment of Pediatrics, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Katheryn GrandDepartment of Pediatrics, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Robert WongDepartment of Anesthesiology, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Ruchira GargGuerin Family Congenital Heart Program, Smidt Heart Institute, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Cedars-Sinai Medical Center · USCedars-Sinai Smidt Heart Institute · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Supravalvar aortic stenosis (SVAS) is a less common but clinically important form of left ventricular outflow tract obstruction, and commonly associated with Williams syndrome (WS). SVAS outside of WS may also occur sporadically or in a familial form, often with identifiable mutations in the elastin (ELN) gene. While risk of sudden cardiac death in patients with SVAS has been extensively described in the context of WS, less is known about risk in patients with isolated SVAS. We report a case of a nonsyndromic two-year-old boy with evolving manifestations of SVAS who developed sudden cardiac arrest and death during a sedated cardiac magnetic resonance imaging study. A strong family history of SVAS was present and targeted genetic testing identified an ELN gene mutation in the boy's affected father and other paternal relatives. We review risk factors found in the literature for SCA in SVAS patients and utilize this case to raise awareness of the risk of cardiac events in these individuals even in the absence of WS or severe disease. This case also underscores the importance of genetic testing, including targeted panels specifically looking for ELN gene mutations, in all patients with SVAS even in the absence of phenotypic concerns for WS or other genetic syndromes.

Indexed as

Aortic Stenosis, SupravalvularWilliams SyndromeChildChild, PreschoolDeath, Sudden, CardiacElastinHumansMagnetic Resonance SpectroscopyMaleMutationElastinElastinElastin arteriopathySudden cardiac arrestSupravalvar aortic stenosisWilliams syndrome

Identifiers

PMID36790509
PMCPMC10063468
OpenAlexW4320856431

What OpenQuestion holds

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LicenceCC BY
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.