Evidence map›Paper›PMID 28451876›Full record

ArticleJournal of genetic counseling2017

An Exploration of Genetic Test Utilization, Genetic Counseling, and Consanguinity within the Inborn Errors of Metabolism Collaborative (IBEMC).

Quinn P Stein, Cate Walsh Vockley, Mathew J Edick, Shaohui Zhai, Sally J Hiner, Rebecca S Loman, Laura Davis-Keppen, Taylor A Zuck, Cynthia A Cameron, Susan A Berry and 1 more

Abstract read
In one paragraph

Article in Journal of genetic counseling, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
–field-weighted citation impact
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

3 citing papers in PubMed.

  1. Article
  2. Article
  3. 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

11 authors.

Quinn P SteinAugustana University, 2001 S Summit Ave, Sioux Falls, SD, 57117, USA. qstein@augie.edu.
Cate Walsh VockleyChildren's Hospital of Pittsburgh of UPMC, Pittsburgh, PA, USA.
Mathew J EdickMichigan Public Health Institute, Okemos, MI, USA.
Shaohui ZhaiMichigan Public Health Institute, Okemos, MI, USA.
Sally J HinerMichigan Public Health Institute, Okemos, MI, USA.
Rebecca S LomanAugustana University, 2001 S Summit Ave, Sioux Falls, SD, 57117, USA.
Laura Davis-KeppenSanford Children's Specialty Clinic, Sioux Falls, SD, USA.
Taylor A ZuckAugustana University, 2001 S Summit Ave, Sioux Falls, SD, 57117, USA.
Cynthia A CameronMichigan Public Health Institute, Okemos, MI, USA.
Susan A BerryUniversity of Minnesota, Minneapolis, MN, USA.
Inborn Errors of Metabolism Collaborative

Funding

Collaborative Defining the Natural History of Inborn errors of MetabolismR01HD069039 · NICHD · MICHIGAN PUBLIC HEALTH INSTITUTE · PI BERRY, SUSAN A., CAMERON, CYNTHIA A · 2011 to 2015
$4.3M
NICHD NIH HHS HHSN275201300011CNICHD NIH HHS R01 HD069039
6 · The paper itself

Abstract

The Inborn Errors of Metabolism Collaborative (IBEMC) includes clinicians from 29 institutions collecting data to enhance understanding of metabolic conditions diagnosable by newborn screening. Data collected includes hospitalizations, test results, services, and long-term outcomes. Through evaluation of this data, we sought to determine how frequently genetic counseling had been provided, how often genetic testing was performed, and also determine the consanguinity rate in this population. A data query was performed with the following elements abstracted/analyzed: current age, metabolic condition, whether genetic counseling was provided (and by whom), whether genetic testing was performed, and consanguinity. Genetic counseling was provided to families 95.8% of the time and in 68.6% of cases by a genetic counselor. Genetic testing was performed on 68.0% of subjects, with usage highest for fatty-acid-oxidation disorders (85.1%). The rate of consanguinity was 2.38%. Within this large national collaborative there is a high frequency of genetic counseling, though in one-third of cases a genetic counselor has not been involved. Additionally, while metabolic conditions have historically been diagnosed biochemically, there is currently high utilization of molecular testing suggesting DNA testing is being incorporated into diagnostic assessments - especially for fatty-acid-oxidation disorders where the underlying genotype helps predict clinical presentation.

Indexed as

ConsanguinityGenetic CounselingGenetic TestingHumansInfant, NewbornMaleMetabolism, Inborn ErrorsNeonatal ScreeningConsanguinityGenetic counselingGenetic testingInborn errors of metabolismNewborn screening

Identifiers

PMID28451876
PMCPMC5659965

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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.