Evidence map›Paper›PMID 34127476›Full record

ArticleCanadian family physician Medecin de famille canadien2021

Newborn screening for cystic fibrosis: Role of primary care providers in caring for infants with positive screening results.

June C Carroll, Robin Z Hayeems, Fiona A Miller, Carolyn J Barg, Yvonne Bombard, Pranesh Chakraborty, Beth K Potter, Jessica Peace Bytautas, Karen Tam, Louise Taylor and 5 more

Open access · diamondAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Canadian family physician Medecin de famille canadien, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.

0numbers the graph read from it
0cells of the map it votes in
9citing papers in PubMed
1.5field-weighted citation impact, top 17% 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

9 citing papers in PubMed, 11 citations in OpenAlex.

  1. Review
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  3. Article
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  5. Review
  6. Article
  7. Primary care providers' role in newborn screening result notification for cystic fibrosis.Canadian family physician Medecin de famille canadien · 2021
    Article
  8. Actualities in neonatal endocrine and metabolic screening.Acta endocrinologica (Bucharest, Romania : 2005)
    Article
  9. 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

15 authors at 12 institutions in 4 countries.

June C CarrollFamily physician and clinician scientist, Professor, and Sydney G. Frankfort Chair in Family Medicine in the Department of Family and Community Medicine with the Sinai Health System and the University of Toronto in Ontario. june.carroll@sinaihealth.ca.
Robin Z HayeemsScientist in the Child Health Evaluative Sciences Program at the Hospital for Sick Children in Toronto and Associate Professor in the Institute of Health Policy, Management and Evaluation at the University of Toronto.
Fiona A MillerProfessor of Health Policy and holds the Chair in Health Management Strategies at the Institute of Health Policy, Management and Evaluation at the University of Toronto.
Carolyn J BargResearch analyst at Health Quality Ontario in Toronto.
Yvonne BombardScientist at the Li Ka Shing Knowledge Institute at St Michael's Hospital in Toronto and Associate Professor in the Institute of Health Policy, Management and Evaluation at the University of Toronto.
Pranesh ChakrabortyExecutive Director and Chief Medical Officer of Newborn Screening Ontario in Ottawa, a medical biochemist in the Department of Pediatrics at the Children's Hospital of Eastern Ontario in Ottawa and Associate Professor of Pediatrics at the University of Ottawa.
Beth K PotterAssociate Professor in the Department of Epidemiology and Community Medicine at the University of Ottawa and holds the University Research Chair in Health Services for Children with Rare Diseases.
Jessica Peace BytautasDoctoral student in the Dalla Lana School of Public Health and a research assistant in the Institute of Health Policy, Management and Evaluation at the University of Toronto.
Karen TamCertified genetic counselor and screening specialist at Newborn Screening Ontario.
Louise TaylorNurse practitioner with expertise in caring for children with cystic fibrosis at the Hospital for Sick Children.
Elizabeth KerrClinical neuropsychologist and a scientist in the Department of Pediatrics, Division of Neurology, at the Hospital for Sick Children, and Adjunct Faculty in the Department of Pediatrics at the University of Toronto.
Christine DaviesCertified genetic counselor in Ottawa.
Jennifer MilburnOperations Director for Newborn Screening Ontario.
Felix RatjenDivision Chief of Pediatric Respiratory Medicine, Co-lead of the Cystic Fibrosis Centre, Senior Scientist at the Research Institute in the Translational Medicine research program, and Medical Director of the Clinical Research Unit, all at the Hospital for Sick Children, and Professor of Pediatrics at the University of Toronto.
Astrid GuttmannClinician scientist in the Division of Pediatric Medicine at the Hospital for Sick Children, Chief Science Officer and Senior Scientist at ICES, and Professor of Pediatrics with a cross appointment at the Institute of Health Policy, Management and Evaluation and the Epidemiology Division of the Dalla Lana School of Public Health at the University of Toronto.
Hospital for Sick Children · CANewborn Screening Ontario · CAAmerican Institute of Certified Public Accountants · USCenter for Children · USChildren's Hospital of Eastern Ontario · CADepartment of Health Services · NPInstitute for Clinical Evaluative Sciences · CAInstitute of Health Economics · CAInstitute of Health Services and Policy Research · CAQuality Research · USSinai Health System · CASt. Michael's Hospital · CA

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveTo explore primary care providers' (PCPs') preferred roles and confidence in caring for infants receiving a positive cystic fibrosis (CF) newborn screening (NBS) result, as well as management of CF family planning issues, given that expanded NBS has resulted in an increase in positive results.

designMailed questionnaire.

settingOntario.

participantsOntario FPs, pediatricians, and midwives identified by Newborn Screening Ontario as having had an infant with a positive CF NBS result in their practice in the previous 6 months.

main outcome measurePrimary care providers' preferred roles in providing well-baby care for infants with positive CF screening results.

resultsOverall, 321 of 628 (51%) completed surveys (208 FPs, 68 pediatricians, 45 midwives). For well-baby care for infants confirmed to have CF, 77% of PCPs indicated they would not provide total care (ie, 68% would share care with other specialists and 9% would refer to specialists completely); for infants with an inconclusive CF diagnosis, 50% of PCPs would provide total care, 45% would provide shared care, and 5% would refer to a specialist; for CF carriers, 89% of PCPs would provide total care, 9% would provide shared care, and 2% would refer. Half (54%) of PCPs were extremely or very confident in providing reassurance about CF carriers' health. Only 25% knew how to order parents' CF carrier testing; 67% knew how to refer for prenatal diagnosis. Confidence in reassuring parents about the health of CF carrier children was associated with providing total well-baby care for CF carriers (risk ratio of 1.50; 95% CI 1.14 to 1.97) and infants with an inconclusive diagnosis (risk ratio of 3.30; 95% CI 1.34 to 8.16).

conclusionMost PCPs indicated willingness to treat infants with a range of CF NBS results in some capacity. It is concerning that some indicated CF carriers should have specialist involvement and only half were extremely or very confident about reassuring families about carrier status. This raises issues about possible medicalization of those with carrier status, prompting the need for PCP education about genetic disorders and the meaning of genetic test results.

Indexed as

Cystic FibrosisNeonatal ScreeningChildFemaleHealth PersonnelHumansInfantInfant, NewbornOntarioPregnancyPrimary Health Care

Identifiers

PMID34127476
PMCPMC8202744
OpenAlexW3170722326

What OpenQuestion holds

Textmetadata
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.