ArticleAmerican journal of medical genetics. Part A2013
Public attitudes regarding a pilot study of newborn screening for spinal muscular atrophy.
Article in American journal of medical genetics. Part A, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers, 1 of them a synthesis that pooled 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.
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.
Who cites it
18 citing papers in PubMed, 1 synthesis or guideline pooled it.
- A systematic review of approaches for engaging patients for research on rare diseases.Journal of general internal medicine · 2014Pooled it
- The Path to Screening US Newborns for Severe Combined Immunodeficiency, 1968-2018: A Narrative Review of a Successful Biomedical Research and Public Health Partnership.Public health genomics · 2026Review
- 25 years of the SMN genes: the Copernican revolution of spinal muscular atrophy.Acta myologica : myopathies and cardiomyopathies : official journal of the Mediterranean Society of Myology · 2020Review
- "I didn't take it too seriously because I'd just never heard of it": Experiential knowledge and genetic screening for thalassaemia in the UK.Journal of genetic counseling · 2019Article
- Pilot study of population-based newborn screening for spinal muscular atrophy in New York state.Genetics in medicine : official journal of the American College of Medical Genetics · 2018Article
- The role of experiential knowledge within attitudes towards genetic carrier screening: A comparison of people with and without experience of spinal muscular atrophy.Health expectations : an international journal of public participation in health care and health policy · 2018Article
- Newborn genetic screening for spinal muscular atrophy in the UK: The views of the general population.Molecular genetics & genomic medicine · 2018Article
- An Assessment of a Shortened Consent Form for the Storage and Research Use of Residual Newborn Screening Blood Spots.Journal of empirical research on human research ethics : JERHRE · 2017Article
- Newborn screening for spinal muscular atrophy: The views of affected families and adults.American journal of medical genetics. Part A · 2017Article
- A Review on Spinal Muscular Atrophy: Awareness, Knowledge, and Attitudes.Journal of genetic counseling · 2016Review
- Parental intentions to enroll children in a voluntary expanded newborn screening program.Social science & medicine (1982) · 2016Article
- New multiplex real-time PCR approach to detect gene mutations for spinal muscular atrophy.BMC neurology · 2016Article
- Deliberative Discussion Focus Groups.Qualitative health research · 2016Article
- Understanding the experiences and needs of individuals with Spinal Muscular Atrophy and their parents: a qualitative study.BMC neurology · 2015Article
- Framing optional genetic testing in the context of mandatory newborn screening tests.BMC medical informatics and decision making · 2015Article
- Applying public health screening criteria: how does universal newborn screening compare to universal tumor screening for Lynch syndrome in adults with colorectal cancer?Journal of genetic counseling · 2015Article
- The translational potential of research on the ethical, legal, and social implications of genomics.Genetics in medicine : official journal of the American College of Medical Genetics · 2015Review
- Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
5 authors.
Funding
Abstract
A population-based pilot study of newborns screening for a rare genetic condition, spinal muscular atrophy (SMA), is being conducted with funding from the National Institutes of Health. The first component of the study is to assess the ethical, legal, and social implications of population-based pilot studies with a focus on public engagement and parental decision-making for the proposed opt-out approach in this research. We conducted focus groups with members of the general public to ascertain attitudes about the pilot study and acceptability of an opt-out approach in two states, Colorado and Utah, where the pilot screening is being proposed (N = 70). We developed an informational video for the project and showed it to the groups prior to the discussion in order to inform participants about population-based research, newborn screening (NBS), permission/consent models, and SMA. Results indicated support for the conduct of pilot studies that is consistent with the current standard of practice for similar population-based programs. There was support for an opt-out approach for parental decision-making; however there was limited parental knowledge about population-based research, NBS and SMA. In general, our participants considered this pilot study to be low risk and of potential benefit to infants and families. The majority of participants were supportive of an opt-out approach with information delivered through various avenues
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What OpenQuestion holds
Registered trials
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.