A Systematic Review of the Effects of Disclosing Carrier Results Generated Through Newborn Screening

Hayeems, Robin Z.; Bytautas, Jessica P; Miller, Fiona A.

doi:10.1007/s10897-008-9180-1

Cited by 38 publications

(41 citation statements)

References 42 publications

(71 reference statements)

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“…In their systematic review published in 2004, confined to experimental evidence, Oliver et al 8 found no controlled trials of interventions to disclose newborn carrier status to parents, and this remains the case. Reviews 1,8,20,[55][56][57] have focused on the cost and effectiveness of antenatal and newborn screening for SC and thalassaemia 20,21 or newborn screening for CF 1 and its benefits and risks 7 rather than on methods and effects of communicating carrier results. Other reviews note evidence on parents' experiences of newborn screening has been limited, 55 in particular concerning their experiences of receiving carrier results, with what is available being less explored for SC carrier communication.…”

Section: Summary Of Current Evidencementioning

confidence: 99%

“…Other reviews note evidence on parents' experiences of newborn screening has been limited, 55 in particular concerning their experiences of receiving carrier results, with what is available being less explored for SC carrier communication. 57 Research with parents following newborn screening supports disclosure of newborn carrier status. 13,15,[57][58][59][60] However, there is commonly inconsistency in whether specific communication protocols are in place for SC or CF carrier results, 37,[61][62][63] and further research is required into parents' experiences or understanding of such communication 13 and the most effective and acceptable communication models.…”

Section: Summary Of Current Evidencementioning

confidence: 99%

See 1 more Smart Citation

Communication of carrier status information following universal newborn screening for sickle cell disorders and cystic fibrosis: qualitative study of experience and practice

Kai

Ulph²,

Cullinan³

et al. 2009

Health Technol Assess

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How to obtain copies of this and other HTA programme reports An electronic version of this publication, in Adobe Acrobat format, is available for downloading free of charge for personal use from the HTA website (www.hta.ac.uk). A fully searchable CD-ROM is also available (see below).Printed copies of HTA monographs cost £20 each (post and packing free in the UK) to both public and private sector purchasers from our Despatch Agents.Non-UK purchasers will have to pay a small fee for post and packing. For European countries the cost is £2 per monograph and for the rest of the world £3 per monograph.You can order HTA monographs from our Despatch Agents:-fax (with credit card or official purchase order) -post (with credit card or official purchase order or cheque) -phone during office hours (credit card only).Additionally the HTA website allows you either to pay securely by credit card or to print out your order and then post or fax it. Contact details are as follows: Payment methods Paying by chequeIf you pay by cheque, the cheque must be in pounds sterling, made payable to Direct Mail Works Ltd and drawn on a bank with a UK address. Paying by credit cardThe following cards are accepted by phone, fax, post or via the website ordering pages: Delta, Eurocard, Mastercard, Solo, Switch and Visa. We advise against sending credit card details in a plain email. Paying by official purchase orderYou can post or fax these, but they must be from public bodies (i.e. NHS or universities) within the UK. We cannot at present accept purchase orders from commercial companies or from outside the UK. How do I get a copy of HTA on CD?Please use the form on the HTA website (www.hta.ac.uk/htacd.htm). Or contact Direct Mail Works (see contact details above) by email, post, fax or phone. HTA on CD is currently free of charge worldwide.The website also provides information about the HTA programme and lists the membership of the various committees. HTA NIHR Health Technology Assessment programmeT he Health Technology Assessment (HTA) programme, part of the National Institute for Health Research (NIHR), was set up in 1993. It produces high-quality research information on the effectiveness, costs and broader impact of health technologies for those who use, manage and provide care in the NHS. 'Health technologies' are broadly defined as all interventions used to promote health, prevent and treat disease, and improve rehabilitation and long-term care. The research findings from the HTA programme directly influence decision-making bodies such as the National Institute for Health and Clinical Excellence (NICE) and the National Screening Committee (NSC). HTA findings also help to improve the quality of clinical practice in the NHS indirectly in that they form a key component of the 'National Knowledge Service'. The HTA programme is needs led in that it fills gaps in the evidence needed by the NHS. There are three routes to the start of projects. First is the commissioned route. Suggestions for research are actively sought from people working in t...

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Section: Summary Of Current Evidencementioning

confidence: 99%

Section: Summary Of Current Evidencementioning

confidence: 99%

Communication of carrier status information following universal newborn screening for sickle cell disorders and cystic fibrosis: qualitative study of experience and practice

Kai

Ulph²,

Cullinan³

et al. 2009

Health Technol Assess

View full text Add to dashboard Cite

show abstract

“…For example, such sociological analysis can be valuable in developing strategies for interventions for carriers identified through newborn screening programs, as this is not a well-defined or well-understood community (Hayeems et al 2008). When it is difficult to categorize the people on the front end of screening, it is also difficult to customize a program for them.…”

Section: Methodology and Implementationmentioning

confidence: 99%

Commentary: a sociologist's view on community genetics

Raz

2010

J Community Genet

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This commentary illustrates and discusses potential research directions for sociologists and anthropologists interested in the field of community genetics and its emerging networks of individuals genetically at risk. Community genetics-the application of medical genetics in community settings for the benefit of individuals-also involves social issues of lay-professional misunderstandings (and more recently also the different perspectives of various expert communities), stigmatization, discrimination, and medicalization. Focusing on a socio-anthropological perspective regarding the views and disagreements surrounding the definition and scope of community genetics, I overview several epistemological, methodological, and practical contributions that such perspective can offer to the study of community genetics.

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“…This finding may be due to evidence that other factors such as lower income, minority status, and lack of social support are also strongly associated with adverse mental health outcomes. 32 Although much has been written about public attitudes toward the return of genomic research findings, most data come from hypothetical studies. 33 We recently completed a multisite fragile X newborn screening pilot study on the basis of the assumption that research studying the experiences of offering testing and communicating results is needed to fully understand benefits and harms.…”

Section: What This Study Addsmentioning

confidence: 99%

Maternal Consequences of the Detection of Fragile X Carriers in Newborn Screening

et al. 2015

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The possibility of newborn screening for fragile X syndrome is complicated by the potential for identifying premutation carriers. Although knowing the child's carrier status has potential benefits, the possibility of late-onset disorders in carrier children and their parents raises concerns about whether such information would be distressing to parents and potentially more harmful than helpful. This study sought to answer this question by offering voluntary fragile X screening to new parents and returning results for both the full mutation and premutation FMR1 gene expansions. We tested the assumption that such information could lead to adverse mental health outcomes or decision regret. We also wanted to know if child age and spousal support were associated with the outcomes of interest.METHODS: Eighteen mothers of screen-positive infants with the premutation and 15 comparison mothers completed a battery of assessments of maternal anxiety, postpartum depression, stress, family quality of life, decision regret, and spousal support. The study was longitudinal, with an average of 3 assessments per mother. RESULTS:The premutation group was not statistically different from the comparison group on measures of anxiety, depression, stress, or quality of life. A subset of mothers experienced clinically significant anxiety and decision regret, but factors associated with these outcomes could not be identified. Greater spousal support was generally associated with more positive outcomes.CONCLUSIONS: Although we did not find evidence of significant adverse events, disclosure of newborn carrier status remains an important consideration in newborn screening policy.WHAT'S KNOWN ON THIS SUBJECT: Parents generally adapt well to newborn screening results, but reactions to carrier status for X-linked conditions are unknown. WHAT THIS STUDY ADDS:Results suggest that detection and disclosure of FMR1 newborn carrier status may not result in significant adverse events for mothers.

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A Systematic Review of the Effects of Disclosing Carrier Results Generated Through Newborn Screening

Cited by 38 publications

References 42 publications

Communication of carrier status information following universal newborn screening for sickle cell disorders and cystic fibrosis: qualitative study of experience and practice

Communication of carrier status information following universal newborn screening for sickle cell disorders and cystic fibrosis: qualitative study of experience and practice

Commentary: a sociologist's view on community genetics

Maternal Consequences of the Detection of Fragile X Carriers in Newborn Screening

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