Evidence map›Paper›PMID 35937981›Full record

ArticleFrontiers in genetics2022

Clinically available testing options resulting in diagnosis in post-exome clinic at one medical center.

Elizabeth K Baker, Elizabeth A Ulm, Alyce Belonis, Diana S Brightman, Barbara E Hallinan, Nancy D Leslie, Alexander G Miethke, Marissa Vawter-Lee, Yaning Wu, Loren D M Pena

Open access · goldAbstract read
In one paragraph

Article in Frontiers in genetics, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
0.6field-weighted citation impact, top 28% 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

0 citing papers in PubMed, 3 citations in OpenAlex.

No citing paper in PubMed yet.

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 1 institution in 1 country.

Elizabeth K BakerDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.
Elizabeth A UlmDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.
Alyce BelonisDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.
Diana S BrightmanDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.
Barbara E HallinanDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH, United States.
Nancy D LeslieDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.
Alexander G MiethkeDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH, United States.
Marissa Vawter-LeeDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH, United States.
Yaning WuDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.
Loren D M PenaDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.
Cincinnati Children's Hospital Medical Center · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Exome sequencing (ES) became clinically available in 2011 and promised an agnostic, unbiased next-generation sequencing (NGS) platform for patients with symptoms believed to have a genetic etiology. The diagnostic yield of ES has been estimated to be between 25-40% and may be higher in specific clinical scenarios. Those who remain undiagnosed may have no molecular findings of interest on ES, variants of uncertain significance in genes that are linked to human disease, or variants of uncertain significance in candidate genes that are not definitively tied to human disease. Recent evidence suggests that a post-exome evaluation consisting of clinical re-phenotyping, functional studies of candidate variants in known genes, and variant reevaluation can lead to a diagnosis in 5-15% of additional cases. In this brief research study, we present our experience on post-exome evaluations in a cohort of patients who are believed to have a genetic etiology for their symptoms. We have reached a full or partial diagnosis in approximately 18% (6/33) of cases that have completed evaluations to date. We accomplished this by utilizing NGS-based methods that are available on a clinical basis. A sample of these cases highlights the utility of ES reanalysis with updated phenotyping allowing for the discovery of new genes, re-adjudication of known variants, incorporating updated phenotypic information, utilizing functional testing such as targeted RNA sequencing, and deploying other NGS-based testing methods such as gene panels and genome sequencing to reach a diagnosis.

Indexed as

exome sequencinggenome sequencingpost-exome clinicsequencing reanalysisundiagnosed disease

Identifiers

PMID35937981
PMCPMC9355124
OpenAlexW4286559811

What OpenQuestion holds

Textmetadata
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.