Publications by authors named "Fiona Ulph"

36 Publications

Pharmacogenetics to Avoid Loss of Hearing (PALOH) trial: a protocol for a prospective observational implementation trial.

BMJ Open 2021 06 16;11(6):e044457. Epub 2021 Jun 16.

Manchester Centre for Genomic Medicine, Manchester University NHS Foundation Trust, Manchester, UK.

Introduction: In conjunction with a beta-lactam, aminoglycosides are the first-choice antibiotic for empirical treatment of sepsis in the neonatal period. The m.1555A>G variant predisposes to ototoxicity after aminoglycoside administration and has a prevalence of 1 in 500. Current genetic testing can take over 24 hours, an unacceptable delay in the acute setting. This prospective-observational trial will implement a rapid point of care test (POCT), facilitating tailored antibiotic prescribing to avoid hearing loss.

Methods And Analysis: The genedrive POCT can detect the m.1555A>G variant in 26 min from buccal swab. This system will be integrated into the clinical pathways at two large UK neonatal centres over a minimum 6-month period. The primary outcome is the number of neonates successfully tested for the variant out of all babies prescribed antibiotics. As a secondary outcome, clinical timings will be compared with data collected prior to implementation, measuring the impact on routine practice.

Ethics And Dissemination: Approval for the trial was granted by the Research Ethics Committee (REC) and Human Research Authority in August 2019. Results will be published in full on completion of the study.

Trial Registration Number: ISRCTN13704894.

Protocol Version: V 1.3.
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http://dx.doi.org/10.1136/bmjopen-2020-044457DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8211036PMC
June 2021

Processing of positive newborn screening results: a qualitative exploration of current practice in England.

BMJ Open 2020 12 12;10(12):e044755. Epub 2020 Dec 12.

Florence Nightingale Faculty of Nursing, Midwifery & Palliative Care, King's College London, London, UK.

Objective: To explore current communication practices for positive newborn screening results from the newborn bloodspot screening (NBS) laboratory to clinicians to highlight differences, understand how the pathways are implemented in practice, identify barriers and facilitators and make recommendations for future practice and research.

Design: A qualitative exploratory design was employed using semi-structured interviews.

Setting: Thirteen NBS laboratories in England.

Participants: Seventy-one clinicians; 22 NBS laboratory staff across 13 laboratories and 49 members of relevant clinical teams were interviewed.

Results: Assurance of quality and consistency was a priority for all NBS laboratories. Findings indicated variation in approaches to communicating positive NBS results from laboratories to clinical teams. This was particularly evident for congenital hypothyroidism and was largely influenced by local arrangements, resources and the fact individual laboratories had detailed standard operating procedures for how they work. Obtaining feedback from clinical teams to the laboratory after the child had been seen could be challenging and time-consuming for those involved. Pathways for communicating carrier results for cystic fibrosis and sickle cell disease could be ambiguous and inconsistent which in turn could hamper the laboratories efforts to obtain timely feedback regarding whether or not the result had been communicated to the family. Communication pathways for positive NBS results between laboratories and clinical teams could therefore be time-consuming and resource-intensive.

Conclusion: The importance placed on ensuring positive NBS results were communicated effectively and in a timely fashion from the laboratory to the clinical team was evident from all participants. However, variation existed in terms of the processes used to report positive NBS results to clinical teams and the people involved. Variant practice identified may reflect local needs, but more often reflected local resources and a more consistent 'best practice' approach is required, not just in the UK but perhaps globally.

Trial Registration Number: ISRCTN15330120.
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http://dx.doi.org/10.1136/bmjopen-2020-044755DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7735110PMC
December 2020

Constructing a Bioethical Framework to Evaluate and Optimise Newborn Bloodspot Screening for Cystic Fibrosis.

Int J Neonatal Screen 2020 06 26;6(2):40. Epub 2020 May 26.

Department of Women's and Children's Health, University of Liverpool, Liverpool L12 2AP, UK;

Newborn bloodspot screening for cystic fibrosis is a valid public health strategy for populations with a high incidence of this inherited condition. There are a wide variety of approaches to screening and in this paper, we propose that a bioethical framework is required to determine the most appropriate screening protocol for a population. This framework depends on the detailed evaluation of the ethical consequences of all screening outcomes and placing these in the context of the genetic profile of the population screened, the geography of the region and the healthcare resources available.
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http://dx.doi.org/10.3390/ijns6020040DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7422997PMC
June 2020

Qualitative exploration of health professionals' experiences of communicating positive newborn bloodspot screening results for nine conditions in England.

BMJ Open 2020 10 1;10(10):e037081. Epub 2020 Oct 1.

Paediatrics, Institute of Child Health, Merseyside, UK.

Objective: To explore health professionals' experiences of communicating positive newborn bloodspot screening (NBS) results, highlight differences, share good practice and make recommendations for future research.

Design: Qualitative exploratory design was employed using semi-structured interviews SETTING: Three National Health Service provider organisations in England PARTICIPANTS: Seventeen health professionals involved in communicating positive newborn bloodspot screening results to parents for all nine conditions currently included in the newborn bloodspot screening programme in England.

Results: Findings indicated variation in approaches to communicating positive newborn bloodspot screening results to parents, largely influenced by resources available and the lack of clear guidance. Health professionals emphasised the importance of communicating results to families in a way that is sensitive to their needs. However, many challenges hindered communication including logistical considerations; difficulty contacting the family and other health professionals; language barriers; parental reactions; resource considerations; lack of training; and insufficient time.

Conclusion: Health professionals invest a lot of time and energy trying to ensure communication of positive newborn bloodspot screening results to families is done well. However, there continues to be great variation in the way these results are communicated to parents and this is largely influenced by resources available but also the lack of concrete guidance. How best to support health professionals undertaking this challenging and emotive task requires further exploration. We recommend evaluation of a more cohesive approach that meets the needs of parents and staff while being sensitive to the subtleties of each condition.

Trial Registration Number: ISRCTN15330120.
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http://dx.doi.org/10.1136/bmjopen-2020-037081DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7534703PMC
October 2020

What are the benefits and harms of risk stratified screening as part of the NHS breast screening Programme? Study protocol for a multi-site non-randomised comparison of BC-predict versus usual screening (NCT04359420).

BMC Cancer 2020 Jun 18;20(1):570. Epub 2020 Jun 18.

NIHR Manchester Biomedical Research Centre, Manchester Academic Health Science Centre, Central Manchester University Hospitals NHS Foundation Trust, Manchester, England.

Background: In principle, risk-stratification as a routine part of the NHS Breast Screening Programme (NHSBSP) should produce a better balance of benefits and harms. The main benefit is the offer of NICE-approved more frequent screening and/ or chemoprevention for women who are at increased risk, but are unaware of this. We have developed BC-Predict, to be offered to women when invited to NHSBSP which collects information on risk factors (self-reported information on family history and hormone-related factors via questionnaire; mammographic density; and in a sub-sample, Single Nucleotide Polymorphisms). BC-Predict produces risk feedback letters, inviting women at high risk (≥8% 10-year) or moderate risk (≥5 to < 8% 10-year) to have discussion of prevention and early detection options at Family History, Risk and Prevention Clinics. Despite the promise of systems such as BC-Predict, there are still too many uncertainties for a fully-powered definitive trial to be appropriate or ethical. The present research aims to identify these key uncertainties regarding the feasibility of integrating BC-Predict into the NHSBSP. Key objectives of the present research are to quantify important potential benefits and harms, and identify key drivers of the relative cost-effectiveness of embedding BC-Predict into NHSBSP.

Methods: A non-randomised fully counterbalanced study design will be used, to include approximately equal numbers of women offered NHSBSP (n = 18,700) and BC-Predict (n = 18,700) from selected screening sites (n = 7). In the initial 8-month time period, women eligible for NHSBSP will be offered BC-Predict in four screening sites. Three screening sites will offer women usual NHSBSP. In the following 8-months the study sites offering usual NHSBSP switch to BC-Predict and vice versa. Key potential benefits including uptake of risk consultations, chemoprevention and additional screening will be obtained for both groups. Key potential harms such as increased anxiety will be obtained via self-report questionnaires, with embedded qualitative process analysis. A decision-analytic model-based cost-effectiveness analysis will identify the key uncertainties underpinning the relative cost-effectiveness of embedding BC-Predict into NHSBSP.

Discussion: We will assess the feasibility of integrating BC-Predict into the NHSBSP, and identify the main uncertainties for a definitive evaluation of the clinical and cost-effectiveness of BC-Predict.

Trial Registration: Retrospectively registered with clinicaltrials.gov (NCT04359420).
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http://dx.doi.org/10.1186/s12885-020-07054-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7302349PMC
June 2020

The introduction of risk stratified screening into the NHS breast screening Programme: views from British-Pakistani women.

BMC Cancer 2020 May 20;20(1):452. Epub 2020 May 20.

Nightingale & Prevent Breast Cancer Research Unit, Manchester University NHS Foundation Trust (MFT), Southmoor Road, Wythenshawe, Manchester, M23 9LT, UK.

Background: UK national guidelines suggest women at high-risk of breast cancer should be offered more frequent screening or preventative medications. Currently, only 1 in 6 high-risk women are identified. One route to identify more high-risk women is via multifactorial risk assessment as part of the UK's NHS Breast Screening Programme (NHSBSP). As lower socioeconomic and minority ethnic populations continue to experience barriers to screening, it is important that any new service does not exacerbate issues further. To inform service development, this study explored views of women from underserved backgrounds regarding the introduction of risk stratification into the NHSBSP.

Methods: Nineteen semi-structured interviews were conducted with British-Pakistani women from low socioeconomic backgrounds from East Lancashire, UK. Fourteen interviews were conducted via an interpreter.

Results: Thematic analysis produced three themes. Attitudes toward risk awareness concerns the positive views women have toward the idea of receiving personalised breast cancer risk information. Anticipated barriers to accessibility emphasises the difficulties associated with women's limited English skills for accessing information, and their I.T proficiency for completing an online risk assessment questionnaire. Acceptability of risk communication strategy highlights the diversity of opinion regarding the suitability of receiving risk results via letter, with the option for support from a healthcare professional deemed essential.

Conclusions: The idea of risk stratification was favourable amongst this underserved community. To avoid exacerbating inequities, this new service should provide information in multiple languages and modalities and offer women the opportunity to speak to a healthcare professional about risk. This service should also enable completion of personal risk information via paper questionnaires, as well as online.
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http://dx.doi.org/10.1186/s12885-020-06959-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7240981PMC
May 2020

Engagement barriers and service inequities in the NHS Breast Screening Programme: Views from British-Pakistani women.

J Med Screen 2020 09 2;27(3):130-137. Epub 2019 Dec 2.

Nightingale & Prevent Breast Cancer Research Unit, Manchester University NHS Foundation Trust (MFT), Manchester, UK.

Objectives: Previous research has largely attempted to explore breast screening experiences of South Asian women by combining opinions from Pakistani, Bangladeshi, and Indian women. This research often fails to reach the most underserved sub-groups of this population, with socioeconomic status not routinely reported, and English fluency being a participation requirement. With uptake low amongst British-Pakistani women, this study explores the experiences these women encounter when accessing the NHS Breast Screening Programme.

Methods: 19 one-to-one semi-structured interviews were carried out with British-Pakistani women from East Lancashire, UK. 14 interviews were conducted via an interpreter.

Results: Data were analysed using thematic analysis. Three themes were identified: describes how currently the screening service does not facilitate confidentiality or independence. Access requires third-party intervention, with language barriers preventing self-expression. makes distinctions between community and NHS communication. Whereas community communication was invaluable, NHS materials were deemed inaccessible due to translation incongruences and incomprehensible terminology. explores the subjective issues associated with disengagement, including, the cultural misalignment of the service, and perceiving screening as a symptomatic service.

Conclusions: British-Pakistani women face some unique challenges when accessing breast screening. To promote uptake, the service needs to address the translation of screening materials and optimize upon community networks to disseminate knowledge, including knowledge of the screening environment within the context of culture to promote informed choice about attendance.
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http://dx.doi.org/10.1177/0969141319887405DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7645618PMC
September 2020

Diagnosing and Preventing Hearing Loss in the Genomic Age.

Trends Hear 2019 Jan-Dec;23:2331216519878983

Manchester Centre for Genomic Medicine, St Mary's Hospital, Manchester University NHS Foundation Trust, UK.

Over the past two decades, significant technological advances have facilitated the identification of hundreds of genes associated with hearing loss. Variants in many of these genes result in severe congenital hearing loss with profound implications for the affected individual and their family. This review collates these advances, summarizing the current state of genomic knowledge in childhood hearing loss. We consider how current and emerging genetic technologies have the potential to alter our approach to the management and diagnosis of hearing loss. We review approaches being taken to ensure that these discoveries are used in clinical practice to detect genetic hearing loss as soon as possible to reduce unnecessary investigations, provide information about reproductive risks, and facilitate regular follow-up and early treatment. We also highlight how rapid sequencing technology has the potential to identify children susceptible to antibiotic-induced hearing loss and how this adverse reaction can be avoided.
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http://dx.doi.org/10.1177/2331216519878983DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6798159PMC
March 2020

Rethinking Strategies for Positive Newborn Screening Result (NBS+) Delivery (ReSPoND): a process evaluation of co-designing interventions to minimise impact on parental emotional well-being and stress.

Pilot Feasibility Stud 2019 4;5:108. Epub 2019 Sep 4.

9Women's and Children's Health, University of Liverpool, Liverpool, L69 3BX UK.

Background: Newborn blood spot (NBS) screening seeks to prevent ill health, disability and death through early diagnosis and effective intervention. Each year, around 10,000 parents of babies born in England are given a positive NBS result indicating their child may be affected or carriers of one of the nine conditions currently screened for. Despite guidance, these results are inconsistently delivered to parents across geographical regions. There is evidence that many parents are dissatisfied with how NBS results are communicated to them and that poor communication practices can lead to various negative sequelae. The purpose of this study is to co-design, implement and undertake a process evaluation of new, co-designed interventions to improve delivery of initial positive NBS results to parents.

Methods: This mixed-methods study will use four phases with defined outputs. Family Systems Theory will form the theoretical basis for the study. The principles and methods of experience-based co-design will underpin intervention development. Normalisation Process Theory will underpin the process evaluation of the interventions co-designed to improve the delivery of positive NBS results to parents. An economic analysis will determine resource use and costs of current practice and of implementing the new co-designed interventions. The nominal group technique will be used to inform the selection of suitable outcome measures for a future evaluation study.

Discussion: The main output of the proposed study will be co-designed interventions for initial communication of positive NBS results to parents ready to be evaluated in a definitive evaluation study.The interventions, co-designed with parents, will help to minimise potential negative sequelae associated with poor communication practices by considering parental and staff experiences as well as healthcare challenges such as finite resources. In addition, information about indicative costs associated with different communication strategies will be determined.It is anticipated it may also be possible to extrapolate principles of good communication practices from the present study for the delivery of bad news to parents for children newly diagnosed with other conditions including cancer and other chronic conditions such as diabetes or epilepsy.

Trial Registration: ISRCTN 15330120 date of registration 17/01/2018.
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http://dx.doi.org/10.1186/s40814-019-0487-5DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6724281PMC
September 2019

Psychological Impact on Parents of an Inconclusive Diagnosis Following Newborn Bloodspot Screening for Cystic Fibrosis: A Qualitative Study.

Int J Neonatal Screen 2019 Jun 11;5(2):23. Epub 2019 Jun 11.

Division of Psychology & Mental Health, School of Health Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester Academic Health Science Centre, Oxford Road, Manchester M13 9PL, UK.

Genetic results of uncertain clinical significance are being returned to parents following newborn screening, representing a paradigm change in how society considers health and illness. 'Cystic Fibrosis screen positive, inconclusive diagnosis' (CFSPID) is a designation given to newborns with a positive screening result for, but not a definitive diagnosis of, cystic fibrosis. We explored the psychological impact of receiving a CFSPID result on parents. Five semi-structured interviews were conducted with eight parents whose children have CFSPID. Interpretative phenomenological analysis identified these themes: "The way we were told": 'diagnosis as a traumatic event' focused on how parents were distressed and dissatisfied by the initial screening result communication, 'Facing and challenging traditional ideas about health and illness' explored the emerging problem of how CFSPID does not fit the commonly accepted medical model, and 'Making certainty out of uncertainty' explored the varying strategies parents developed to adapt to the uncertainty regarding their child's prognosis. Findings suggest that CFSPID results caused parents' distress, initiated with the first communication of the result and persisting thereafter. Our data suggests approaches to the delivery of CFSPID results that may reduce the impact. Work is needed to close the gap between healthcare advances and societies commonly held medical model.
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http://dx.doi.org/10.3390/ijns5020023DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7510205PMC
June 2019

Facilitators and Barriers to Participating in a Randomized Controlled Trial of a Psychological Therapy for Substance Use.

J Nerv Ment Dis 2019 Jun;207(6):487-496

Division of Psychology and Mental Health, School of Health Sciences, University of Manchester, Manchester, United Kingdom.

Research involving substance users with psychosis has high refusal and attrition rates. Understanding participants' experiences of research participation is therefore important. Twenty-two cannabis users with schizophrenia spectrum disorders were recruited. They were involved in a randomized controlled trial exploring the effectiveness of motivational interviewing plus cognitive behavioral therapy. Semistructured interviews were conducted, and their responses were analyzed using thematic analysis. Thematic analysis identified facilitators such as therapeutic benefit, knowledge about medical conditions, awareness of own drug habits, helping others, confidentiality, rapport with researchers, desiring social contact, and views of significant others. Barriers identified were cognitive challenge, amount of assessments, duration, and discussions about the past. Based on these insights, we recommend informing participants about clinical equipoise, confidentiality, and demands of the study; minimizing demands on participants; acknowledging participants' efforts; involving potential participants' significant others during recruitment; advertising research as an opportunity to help; and training researchers in building rapport.
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http://dx.doi.org/10.1097/NMD.0000000000001000DOI Listing
June 2019

Preferences for aspects of antenatal and newborn screening: a systematic review.

BMC Pregnancy Childbirth 2019 Apr 16;19(1):131. Epub 2019 Apr 16.

Manchester Centre for Health Economics, The University of Manchester, Oxford Road, Manchester, M13 9PL, UK.

Background: Many countries offer screening programmes to unborn and newborn babies (antenatal and newborn screening) to identify those at risk of certain conditions to aid earlier diagnosis and treatment. Technological advances have stimulated the development of screening programmes to include more conditions, subsequently changing the information required and potential benefit-risk trade-offs driving participation. Quantifying preferences for screening programmes can provide programme commissioners with data to understand potential demand, the drivers of this demand, information provision required to support the programmes and the extent to which preferences differ in a population. This study aimed to identify published studies eliciting preferences for antenatal and newborn screening programmes and provide an overview of key methods and findings.

Methods: A systematic search of electronic databases for key terms identified eligible studies (discrete choice experiments (DCEs) or best-worst scaling (BWS) studies related to antenatal/newborn testing/screening published between 1990 and October 2018). Data were systematically extracted, tabulated and summarised in a narrative review.

Results: A total of 19 studies using a DCE or BWS to elicit preferences for antenatal (n = 15; 79%) and newborn screening (n = 4; 21%) programmes were identified. Most of the studies were conducted in Europe (n = 12; 63%) but there were some examples from North America (n = 2; 11%) and Australia (n = 2; 11%). Attributes most commonly included were accuracy of screening (n = 15; 79%) and when screening occurred (n = 13; 68%). Other commonly occurring attributes included information content (n = 11; 58%) and risk of miscarriage (n = 10; 53%). Pregnant women (n = 11; 58%) and healthcare professionals (n = 11; 58%) were the most common study samples. Ten studies (53%) compared preferences across different respondents. Two studies (11%) made comparisons between countries. The most popular analytical model was a standard conditional logit model (n = 11; 58%) and one study investigated preference heterogeneity with latent class analysis.

Conclusion: There is an existing literature identifying stated preferences for antenatal and newborn screening but the incorporation of more sophisticated design and analytical methods to investigate preference heterogeneity could extend the relevance of the findings to inform commissioning of new screening programmes.
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http://dx.doi.org/10.1186/s12884-019-2278-7DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6469127PMC
April 2019

Understanding Midwives' Preferences for Providing Information About Newborn Bloodspot Screening.

MDM Policy Pract 2018 Jan-Jun;3(1):2381468317746170. Epub 2018 Jan 18.

Manchester Centre for Health Economics, Division of Population Health, Health Services Research & Primary Care (SJW, KP).

Understanding preferences for information provision in the context of health care service provision is challenging because of the number of potential attributes that may influence preferences. This study aimed to identify midwives' preferences for the process and outcomes of information provision in an expanded national newborn bloodspot screening program. A sample of practicing midwives completed a hybrid-stated preference survey including a conjoint analysis (CA) and discrete choice experiment to quantify preferences for the types of, and way in which, information should be provided in a newborn bloodspot screening program. Six conjoint analysis questions captured the impact of different types of information on parents' ability to make a decision, and 10 discrete choice experiment questions identified preferences for four process attributes (including parents' ability to make a decision). Midwives employed by the UK National Health Service (n = 134) completed the survey. All types of information content were perceived to improve parents' ability to make a decision except for the possibility of false-positive results. Late pregnancy was seen to be the best time to provide information, followed by day 3 postbirth. Information before 20 weeks of pregnancy was viewed as reducing parents' ability to make a decision. Midwives preferred information to be provided by an individual discussion and did not think parents should receive information on the Internet. A hybrid stated preference survey design identified that a wide variety of information should be provided to maximize parents' ability to make a decision ideally provided late in pregnancy or on day 3 postbirth.
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http://dx.doi.org/10.1177/2381468317746170DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6125045PMC
January 2018

Communication with children about sickle cell disease: A qualitative study of parent experience.

Br J Health Psychol 2018 09 9;23(3):685-700. Epub 2018 Jun 9.

Division of Psychology and Mental Health, The University of Manchester, UK.

Objectives: This study aimed to explore how parents communicate with children affected by sickle cell disease, a condition associated with social and cultural complexities that pose risks to open parent-child communication.

Design: A contextualist approach informed the qualitative exploration of parent experience using an individual interview design.

Methods: Twelve semi-structured interviews were conducted with parents whose child had a diagnosis of sickle cell disease. Interviews were audio-recorded, transcribed verbatim, and analysed using inductive thematic analysis.

Results: Parental communication was aimed at educating and protecting children from the physical and emotional effects of SCD in an age appropriate way. Parents commonly described avoidant communication and a small number described using this as a more predominant approach. This appeared to relate to fears about SCD, stigma, and uncertainty about how to best to respond.

Conclusions: As a known mediator of child outcomes, communication offers a promising target for intervention. The findings of this study suggest that parents of children with SCD are likely to benefit from formal support to adapt their communication to their child's needs. Parenting interventions should, in future, include the SCD parent population as a specific group to benefit child outcomes. Statement of contribution What is already known on this subject? Talking openly with children who have genetic conditions is known to promote acceptance and adjustment to illness. Sickle cell disease (SCD) is a genetic condition that is surrounded by various sociocultural issues that may act as barriers to parent-child communication. For example, it is condition that affects only Black and Minority Ethnic (BME) groups and is associated with longstanding stigma within BME communities due to the hereditary nature of the disease. What does this study add? It adds a unique focus on communication in the SCD parent population (previous work has tended to study this group alongside parents of various other genetic conditions). The findings provide a rich insight into parent experience of communication with children about SCD. It reveals that many parents find it difficult to talk openly with children and often avoid sensitive issues such as inheritance, physical limitations, and risk of death which has important implications for child coping and adjustment. It is therefore recommended that health care services are designed to support parents with communication about SCD with their child.
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http://dx.doi.org/10.1111/bjhp.12311DOI Listing
September 2018

The prioritization of symptom beliefs over illness beliefs: The development and validation of the Pain Perception Questionnaire for Young People.

Br J Health Psychol 2018 02 9;23(1):68-87. Epub 2017 Oct 9.

Division of Musculoskeletal and Dermatological Research, School of Biological Sciences, Faculty of Biology, Medicine and Health, Manchester Academic Health Science Centre, University of Manchester, UK.

Objectives: To investigate the suitability of the revised Illness Perception Questionnaire (IPQ-R) for use with adolescents with a long-term pain condition and to validate a new questionnaire for use with this age group.

Design: A three-phase mixed-methods study.

Methods: Phase 1 comprised in-depth qualitative analyses of audio-recorded cognitive interviews with 20 adolescents with juvenile idiopathic arthritis who were answering IPQ-R items. Transcripts were coded using framework analysis. A content analysis of their intended responses to individual items was also conducted. In Phase 2, a new questionnaire was developed and its linguistic and face validity were assessed with 18 adolescents without long-term conditions. In Phase 3, the construct validity of the new questionnaire was assessed with 240 adolescents with juvenile idiopathic arthritis. A subset of 43 adolescents completed the questionnaire a second time to assess test-retest reliability. All participants were aged 11-16 years.

Results: Participants described both conceptual and response format difficulties when answering IPQ-R items. In response, the Pain Perception Questionnaire for Young People (PPQ-YP) was designed which incorporated significant modifications to both wording and response formats when compared with the IPQ-R. A principal component analysis of the PPQ-YP identified ten constructs in the new questionnaire. Emotional representations were separated into two constructs, responsive and anticipatory emotions. The PPQ-YP showed high test-retest reliability.

Conclusions: Symptom beliefs appear to be more salient to adolescents with a long-term pain condition than beliefs about the illness as a whole. A new questionnaire to assess pain beliefs of adolescents was designed. Further validation work may be needed to assess its suitability for use with other pain conditions. Statement of contribution What is already known on this subject? Versions of the adult Revised Illness Perception Questionnaire (IPQ-R) have been adapted for adolescents and children by changing item wording; however, research to assess the degree to which the underlying IPQ-R constructs are relevant to adolescents with a long-term condition had not been performed. What the present study adds? In adolescents, beliefs about symptoms of their condition are more salient than beliefs about the illness as a whole. Question response formats for children and young people need to take account of age-specific abilities. A new questionnaire has been designed for adolescents with pain. It is theoretically congruent with the CS-SRM.
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http://dx.doi.org/10.1111/bjhp.12275DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5765490PMC
February 2018

Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project.

Health Technol Assess 2017 10;21(55):1-240

Division of Nursing, Midwifery and Social Work, School of Health Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK.

Background: Participation in the UK Newborn Bloodspot Screening Programme (NBSP) requires parental consent but concerns exist about whether or not this happens in practice and the best methods and timing to obtain consent at reasonable cost.

Objectives: To collate all possible modes of prescreening communication and consent for newborn (neonatal) screening (NBS); examine midwives', screening professionals' and users' views about the feasibility, efficiency and impact on understanding of each; measure midwives' and parents' preferences for information provision; and identify key drivers of cost-effectiveness for alternative modes of information provision.

Design: Six study designs were used: (1) realist review - to generate alternative communication and consent models; (2) qualitative interviews with parents and health professionals - to examine the implications of current practice for understanding and views on alternative models; (3) survey and observation of midwives - to establish current costs; (4) stated preference surveys with midwives, parents and potential future parents - to establish preferences for information provision; (5) economic analysis - to identify cost-effectiveness drivers of alternative models; and (6) stakeholder validation focus groups and interviews - to examine the acceptability, views and broader impact of alternative communication and consent models.

Setting: Providers and users of NBS in England.

Participants: Study 2: 45 parents and 37 health professionals; study 3: 22 midwives and eight observations; study 4: 705 adults aged 18-45 years and 134 midwives; and study 6: 12 health-care professionals and five parents.

Results: The realist review identified low parental knowledge and evidence of coercive consent practices. Interview, focus group and stated preference data suggested a preference for full information, with some valuing this more than choice. Health professionals preferred informed choice models but parents and health professionals queried whether or not current consent was fully informed. Barriers to using leaflets effectively were highlighted. All studies indicated that a 'personalised' approach to NBS communication, allowing parents to select the mode and level of information suited to their learning needs, could have added value. A personalised approach should rely on midwife communication and should occur in the third trimester. Overall awareness was identified as requiring improvement. Starting NBS communication by alerting parents that they have a choice to make and telling them that samples could be stored are both likely to enhance engagement. The methods of information provision and maternal anxiety causing additional visits to health-care professionals were the drivers of relative cost-effectiveness. Lack of data to populate an economic analysis, confirmed by value of information analysis, indicated a need for further research.

Limitations: There are some limitations with regard to the range of participants used in studies 2 and 3 and so caution should be exercised when interpreting some of the results.

Conclusions: This project highlighted the importance of focusing on information receipt and identified key communication barriers. Health professionals strongly preferred informed consent, which parents endorsed if they were made aware of sample storage. Uniform models of information provision were perceived as ineffective. A choice of information provision was supported by health professionals and parents, which both enhances cost-effectiveness and improves engagement, understanding and the validity of consent. Remaining uncertainties suggest that more research is needed before new communication modes are introduced into practice. Future research should measure the impact of the suggested practice changes (informing in third trimester, information toolkits, changed role of midwife).

Trial Registration: Current Controlled Trials ISRCTN70227207.

Funding: This project was funded by the NIHR Health Technology Assessment programme and will be published in full in ; Vol. 21, No. 55. See the NIHR Journals Library website for further project information.
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http://dx.doi.org/10.3310/hta21550DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5641821PMC
October 2017

Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project.

Health Technol Assess 2017 10;21(55):1-240

Division of Nursing, Midwifery and Social Work, School of Health Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK.

Background: Participation in the UK Newborn Bloodspot Screening Programme (NBSP) requires parental consent but concerns exist about whether or not this happens in practice and the best methods and timing to obtain consent at reasonable cost.

Objectives: To collate all possible modes of prescreening communication and consent for newborn (neonatal) screening (NBS); examine midwives', screening professionals' and users' views about the feasibility, efficiency and impact on understanding of each; measure midwives' and parents' preferences for information provision; and identify key drivers of cost-effectiveness for alternative modes of information provision.

Design: Six study designs were used: (1) realist review - to generate alternative communication and consent models; (2) qualitative interviews with parents and health professionals - to examine the implications of current practice for understanding and views on alternative models; (3) survey and observation of midwives - to establish current costs; (4) stated preference surveys with midwives, parents and potential future parents - to establish preferences for information provision; (5) economic analysis - to identify cost-effectiveness drivers of alternative models; and (6) stakeholder validation focus groups and interviews - to examine the acceptability, views and broader impact of alternative communication and consent models.

Setting: Providers and users of NBS in England.

Participants: Study 2: 45 parents and 37 health professionals; study 3: 22 midwives and eight observations; study 4: 705 adults aged 18-45 years and 134 midwives; and study 6: 12 health-care professionals and five parents.

Results: The realist review identified low parental knowledge and evidence of coercive consent practices. Interview, focus group and stated preference data suggested a preference for full information, with some valuing this more than choice. Health professionals preferred informed choice models but parents and health professionals queried whether or not current consent was fully informed. Barriers to using leaflets effectively were highlighted. All studies indicated that a 'personalised' approach to NBS communication, allowing parents to select the mode and level of information suited to their learning needs, could have added value. A personalised approach should rely on midwife communication and should occur in the third trimester. Overall awareness was identified as requiring improvement. Starting NBS communication by alerting parents that they have a choice to make and telling them that samples could be stored are both likely to enhance engagement. The methods of information provision and maternal anxiety causing additional visits to health-care professionals were the drivers of relative cost-effectiveness. Lack of data to populate an economic analysis, confirmed by value of information analysis, indicated a need for further research.

Limitations: There are some limitations with regard to the range of participants used in studies 2 and 3 and so caution should be exercised when interpreting some of the results.

Conclusions: This project highlighted the importance of focusing on information receipt and identified key communication barriers. Health professionals strongly preferred informed consent, which parents endorsed if they were made aware of sample storage. Uniform models of information provision were perceived as ineffective. A choice of information provision was supported by health professionals and parents, which both enhances cost-effectiveness and improves engagement, understanding and the validity of consent. Remaining uncertainties suggest that more research is needed before new communication modes are introduced into practice. Future research should measure the impact of the suggested practice changes (informing in third trimester, information toolkits, changed role of midwife).

Trial Registration: Current Controlled Trials ISRCTN70227207.

Funding: This project was funded by the NIHR Health Technology Assessment programme and will be published in full in ; Vol. 21, No. 55. See the NIHR Journals Library website for further project information.
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http://dx.doi.org/10.3310/hta21550DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5641821PMC
October 2017

Eliciting Preferences for Information Provision in Newborn Bloodspot Screening Programs.

Value Health 2017 04 3;20(4):651-661. Epub 2017 Jan 3.

Manchester Centre for Health Economics, Division of Population Health, Health Services Research and Primary Care, the University of Manchester, Manchester, UK. Electronic address:

Background: The national newborn bloodspot screening programs (NBSPs) are continually expanding to screen for more conditions.

Objectives: To quantify parents' preferences for information and the way in which this is provided in example NBSPs.

Methods: A hybrid choice experiment, combining a conjoint analysis and a discrete choice experiment, was designed. A sample of current and future parents between the ages of 18 and 45 years was identified via an Internet panel. Respondents completed one of two survey versions (9 conditions and 20 conditions) comprising a validated measure of attitudes toward involvement in decision making, 6 CA questions (11 information attributes), 10 DCE questions (4 attributes: 3 process and the ability to make an informed decision), and demographic questions.

Results: Of the 702 respondents who completed the survey, 58% were women, 48% were between 25 and 34 years old, and 48% were current parents. All types of information were identified to statistically significantly improve parents' ability to make a decision. Participants preferred taking an "active" role in decision making. Respondents to the 9-condition survey preferred information before 20 weeks (willingness to pay [WTP] £11.88; CI £5.56 to £19.53) and the 20-condition group after 20 weeks (WTP £15.91; CI £10.64 to £21.63). All respondents disliked receiving information 3 days after birth, with the 20-condition group also being averse to receiving it on day 5 (WTP -£11.20; CI -£18.40 to 5.72). Respondents in both groups preferred to receive their information in an individual discussion.

Conclusions: This study suggests that parents' preferences for receiving NBS information differ from how this information is given in current UK practice.
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http://dx.doi.org/10.1016/j.jval.2016.11.007DOI Listing
April 2017

Preventing interpersonal violence in Panama: is a parenting intervention developed in Australia culturally appropriate?

Int J Public Health 2016 Nov 30;61(8):915-922. Epub 2016 Aug 30.

Division of Psychology and Mental Health, School of Health Sciences, Zochonis Building, Brunswick Street, The University of Manchester, Manchester, M13 9PL, UK.

Objectives: To explore cultural appropriateness of a transported parenting intervention in Panama.

Methods: Panamanian parents (n = 25) were interviewed after participation in an Australian parenting intervention. A thematic analysis was conducted to interpret qualitative data.

Results: Three themes emerged; cultural context, appropriateness of the intervention, and development of support networks. In terms of cultural context, parents described economic difficulties, living in a dangerous world, struggling to balance parenting and work, and using aggressive communication patterns. In terms of appropriateness of the intervention, they rated materials as appropriate, although suggested modifications to its delivery by including children and teachers in the training. Finally, parents commented that the intervention prompted the development of social networks within their communities.

Conclusions: Overall, parents considered a transported parenting intervention as appropriate to their local needs. This study might be useful to local governments and international funders in charge of deciding whether transporting parenting interventions North to South as a strategy for violence prevention would be respectful of local needs. Our findings cannot be generalized beyond Panama, but the methodology can be replicated to answer this question in other settings.
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http://dx.doi.org/10.1007/s00038-016-0874-0DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5075342PMC
November 2016

Training Genetic Counsellors to Deliver an Innovative Therapeutic Intervention: their Views and Experience of Facilitating Multi-Family Discussion Groups.

J Genet Couns 2017 Apr 10;26(2):199-214. Epub 2016 Oct 10.

University of Manchester, Manchester, UK.

Innovations in clinical genetics have increased diagnosis, treatment and prognosis of inherited genetic conditions (IGCs). This has led to an increased number of families seeking genetic testing and / or genetic counselling and increased the clinical load for genetic counsellors (GCs). Keeping pace with biomedical discoveries, interventions are required to support families to understand, communicate and cope with their Inherited Genetic Condition. The Socio-Psychological Research in Genomics (SPRinG) collaborative have developed a new intervention, based on multi-family discussion groups (MFDGs), to support families affected by IGCs and train GCs in its delivery. A potential challenge to implementing the intervention was whether GCs were willing and able to undergo the training to deliver the MFDG. In analysing three multi-perspective interviews with GCs, this paper evaluates the training received. Findings suggests that MFDGs are a potential valuable resource in supporting families to communicate genetic risk information and can enhance family function and emotional well-being. Furthermore, we demonstrate that it is feasible to train GCs in the delivery of the intervention and that it has the potential to be integrated into clinical practice. Its longer term implementation into routine clinical practice however relies on changes in both organisation of clinical genetics services and genetic counsellors' professional development.
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http://dx.doi.org/10.1007/s10897-016-0008-0DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5382180PMC
April 2017

Young children's experiences of living with a parent with bipolar disorder: Understanding the child's perspective.

Psychol Psychother 2017 06 19;90(2):212-228. Epub 2016 Jul 19.

The University of Manchester, UK.

Objectives: To explore the experiences of young children of living with a parent with bipolar disorder (BD) and how this impacts on their emotional well-being.

Design: Qualitative study using a computer-assisted semi-structured interview, 'In My Shoes' (IMS).

Methods: Ten children aged between 4 and 10 years with a parent with BD identified via self-help groups were interviewed about their experience of family life. Thematic analysis was used following transcription.

Results: Four main themes emerging from thematic analysis were as follows: perception of parents; knowledge and awareness of BD; managing family life with a 'bipolar' parent; and living in a family with BD. Four-year-old children could participate in the IMS interviews and discuss their parent's mood, behaviour, and mental health. Children had candid and insightful discussions about their parent's BD including symptoms and parenting, and could reflect on how having a parent with BD affected them emotionally and practically. Older children were better able to articulate their parent's illness and its impact.

Conclusions: This exploratory study represents an important step in examining directly experiences of young children whose parents have BD. Using IMS, it was possible to gather insightful information from children to generate hypotheses and influence service development. Children of all ages had some knowledge and understanding of their parent's illness, describing both positive and negative experiences in the family. Further research to build understanding of children's perspectives and the support they feel they and their family would benefit from would enhance the development of appropriate services and interventions.

Practitioner Points: Using age-appropriate tools, it is possible to elicit the views of young children about their parent's mental health and parenting. Young children have insight into the impact of bipolar disorder in the family on themselves and family members.
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http://dx.doi.org/10.1111/papt.12099DOI Listing
June 2017

Exploration of Mechanisms behind Changes after Participation in a Parenting Intervention: A Qualitative Study in a Low-Resource Setting.

Am J Community Psychol 2016 03 30;57(1-2):181-9. Epub 2016 Mar 30.

The University of Manchester, Manchester, UK.

Parenting interventions are effective for preventing psychological difficulties in children. However, their active ingredients have not been comprehensively explored. How do they work? What are the mechanisms operating behind changes? In 2012, a randomized controlled trial of a parenting intervention was conducted in low-resource communities of Panama. Effects on child behavioral difficulties, parental stress, and parenting practices were large in the short and long term. This was an ideal opportunity to explore potential mechanisms operating behind effects found in this low-resource setting. Twenty-five parents were interviewed. Data were analyzed through an inductive semantic thematic analysis. Three themes emerged from the data: (a) psychological mechanisms behind changes, (b) behavioral changes in parent, and (c) changes in the children. Parents described that the intervention triggered changes in emotion regulation, self-efficacy, and problem solving. Parents also reported behavioral changes such as praising their children more often, who in turn seemed more responsible and better at following instructions. The study offers participant-driven insight into potential pathways of change after participation in this parenting intervention, pathways that are often overlooked in quantitative studies. Future studies should further explore these pathways, through mediator and moderator analyses, and determine how much is shared across interventions and across different cultural settings.
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http://dx.doi.org/10.1002/ajcp.12020DOI Listing
March 2016

Developing an intervention to facilitate family communication about inherited genetic conditions, and training genetic counsellors in its delivery.

Eur J Hum Genet 2016 06 7;24(6):794-802. Epub 2015 Oct 7.

University of Manchester, Manchester, UK.

Many families experience difficulty in talking about an inherited genetic condition that affects one or more of them. There have now been a number of studies identifying the issues in detail, however few have developed interventions to assist families. The SPRinG collaborative have used the UK Medical Research Council's guidance on Developing and Evaluating Complex Interventions, to work with families and genetic counsellors (GCs) to co-design a psycho-educational intervention to facilitate family communication and promote better coping and adaptation to living with an inherited genetic condition for parents and their children (<18 years). The intervention is modelled on multi-family discussion groups (MFDGs) used in psychiatric settings. The MFDG was developed and tested over three phases. First focus groups with parents, young people, children and health professionals discussed whether MFDG was acceptable and proposed a suitable design. Using evidence and focus group data, the intervention and a training manual were developed and three GCs were trained in its delivery. Finally, a prototype MFDG was led by a family therapist and co-facilitated by the three GCs. Data analysis showed that families attending the focus groups and intervention thought MFDG highly beneficial, and the pilot sessions had a significant impact on their family' functioning. We also demonstrated that it is possible to train GCs to deliver the MFDG intervention. Further studies are now required to test the feasibility of undertaking a definitive randomised controlled trial to evaluate its effectiveness in improving family outcomes before implementing into genetic counselling practice.
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http://dx.doi.org/10.1038/ejhg.2015.215DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4820094PMC
June 2016

Developing an intervention to facilitate family communication about inherited genetic conditions, and training genetic counsellors in its delivery.

Eur J Hum Genet 2016 06 7;24(6):794-802. Epub 2015 Oct 7.

University of Manchester, Manchester, UK.

Many families experience difficulty in talking about an inherited genetic condition that affects one or more of them. There have now been a number of studies identifying the issues in detail, however few have developed interventions to assist families. The SPRinG collaborative have used the UK Medical Research Council's guidance on Developing and Evaluating Complex Interventions, to work with families and genetic counsellors (GCs) to co-design a psycho-educational intervention to facilitate family communication and promote better coping and adaptation to living with an inherited genetic condition for parents and their children (<18 years). The intervention is modelled on multi-family discussion groups (MFDGs) used in psychiatric settings. The MFDG was developed and tested over three phases. First focus groups with parents, young people, children and health professionals discussed whether MFDG was acceptable and proposed a suitable design. Using evidence and focus group data, the intervention and a training manual were developed and three GCs were trained in its delivery. Finally, a prototype MFDG was led by a family therapist and co-facilitated by the three GCs. Data analysis showed that families attending the focus groups and intervention thought MFDG highly beneficial, and the pilot sessions had a significant impact on their family' functioning. We also demonstrated that it is possible to train GCs to deliver the MFDG intervention. Further studies are now required to test the feasibility of undertaking a definitive randomised controlled trial to evaluate its effectiveness in improving family outcomes before implementing into genetic counselling practice.
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http://dx.doi.org/10.1038/ejhg.2015.215DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4820094PMC
June 2016

A qualitative study to explore how professionals in the United Kingdom make decisions to test children for a sickle cell carrier status.

Eur J Hum Genet 2016 Feb 27;24(2):164-70. Epub 2015 May 27.

School of Psychological Sciences, University of Manchester, Manchester, UK.

European guidelines recommend that, unless there are clear benefits of autosomal recessive carrier testing in childhood, it should be deferred to protect children's autonomous decision making. Although it is believed that children receive testing in the United Kingdom, it is unclear how or why professionals make decisions to provide tests. Semi-structured interviews were conducted with 25 professionals in the United Kingdom who advise about, and undertake, childhood sickle cell trait testing. Data were analysed using thematic analysis. Few professionals were aware of, or used, guidelines to inform testing decisions and instead, considered the reproductive and clinical relevance of testing, and autonomous rights of parents. Many professionals believed testing was important and readily offered it to parents. Professionals who discouraged testing were met with parental resistance and often provided testing when conflict was difficult to manage. Children were rarely considered to be capable of making decisions and few were engaged in discussions. When consulted, older children demonstrated interest, but younger children usually declined testing. Wide variation in testing advice emerged because of opposing beliefs about children's best interests and potential benefits or harms of testing. An explanation of how children's best interests should be determined in light of conflicting evidence regarding the psychosocial and clinical implications of carrier status is needed. Improved awareness of guidelines might encourage professionals to support the role of children in testing decisions. Strategies are also required to help professionals determine children's cognitive capacity and to protect children's future autonomy during discussions with persistent parents.
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http://dx.doi.org/10.1038/ejhg.2015.104DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4717195PMC
February 2016

The Role of Information Provision in Economic Evaluations of Newborn Bloodspot Screening: A Systematic Review.

Appl Health Econ Health Policy 2015 Dec;13(6):615-26

Manchester Centre for Health Psychology, School of Psychological Sciences, The University of Manchester, Manchester, UK.

Background: The extent to which economic evaluations have included the healthcare resource and outcome-related implications of information provision in national newborn bloodspot screening programmes (NBSPs) is not currently known.

Objectives: To identify if, and how, information provision has been incorporated into published economic evaluations of NBSPs.

Methods: A systematic review of economic evaluations of NBSPs (up to November 2014) was conducted. Three electronic databases were searched (Ovid: Medline, Embase, CINAHL) using an electronic search strategy combining a published economic search filter with terms related to national NBSPs and screening-related technologies. These electronic searches were supplemented by searching the NHS Economic Evaluations Database (NHS EED) and hand-searching identified study reference lists. The results were tabulated and summarised as part of a narrative synthesis.

Results: A total of 27 economic evaluations [screening-related technologies (n = 11) and NBSPs (n = 16)] were identified. The majority of economic evaluations did not quantify the impact of information provision in terms of healthcare costs or outcomes. Five studies did include an estimate of the time cost associated with information provision. Four studies included a value to reflect the disutility associated with parental anxiety caused by false-positive results, which was used as a proxy for the impact of imperfect information.

Conclusion: A limited evidence base currently quantifies the impact of information provision on the healthcare costs and impact on the users of NBSPs; the parents of newborns. We suggest that economic evaluations of expanded NBSPs need to take account of information provision otherwise the impact on healthcare costs and the outcomes for newborns and their parents may be underestimated.
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http://dx.doi.org/10.1007/s40258-015-0177-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4751163PMC
December 2015

An exploration of parents' perceptions and beliefs about changes following participation in a family skill training program: a qualitative study in a developing country.

Prev Sci 2015 Jul;16(5):674-84

School of Psychological Sciences, The University of Manchester, Zochonis Building, Brunswick Street, Manchester, M13 9PL, UK.

Family skill training programs have been recognized as effective strategies for preventing substance use. However, they have been evaluated mainly in high-income countries. Families in developing countries also face difficulties; therefore, it is important to explore the fit of existing programs in this context. The present study explores parents' perceptions and beliefs about changes following participation in the Strengthening Families Program 10-14, which was implemented in Panama by the United Nations Office on Drugs and Crime. Thirty parents who had taken part in the program between 2010 and 2011 were interviewed. Thematic analysis was conducted taking a participant-driven inductive stand. An exploration of parents' narratives suggested that, after the program, they observed changes in themselves as parents, in their children, in the interaction between the two of them, and in their functioning as a couple. Perceived changes centered on communication, limits, obedience, relationship roles, emotional regulation, and social development. For example, parents reported being able to control their emotions in a healthier manner, reducing the use of shouting and setting limits in a more effective way. All these factors have been recognized in previous research as strategies for preventing substance use. It is important to assess participants' perceptions of programs brought from elsewhere before dissemination efforts can take place. Parents interviewed for this study appeared to hold positive views about this program. This methodology is discussed as a means of evaluating evidence-based interventions in different cultural settings.
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http://dx.doi.org/10.1007/s11121-014-0530-yDOI Listing
July 2015

Disparities in current and future childhood and newborn carrier identification.

J Genet Couns 2014 Oct 11;23(5):701-7. Epub 2014 Jul 11.

School of Psychological Sciences, University of Manchester, Oxford Road, Manchester, M13 9PL, UK,

International carrier testing guidelines discourage testing in childhood to preserve autonomous decision making and prevent detrimental psychosocial consequences. Despite the discouragement of autosomal recessive carrier testing during childhood, some sickle cell disease (SCD) or cystic fibrosis (CF) carriers are incidentally identified through UK and international newborn screening (NBS). This creates a scenario where parents may have knowledge of their newborn's, but not older child's carrier status. In addition, there is wide variation in the identification of CF and SCD carriers due to the screening technologies implemented by different NBS programs. The current and future availability of childhood testing are determined to some extent by the impact of testing on children and parents (whether this is beneficial or detrimental to wellbeing). However empirical research informing carrier guidance and practice is conflicting. Echoing previous calls, this discussion highlights the need for further qualitative and longitudinal research with children to consider the psychosocial impact of carrier testing on children and role of disclosure from parents on adaptation to results. It is recommended that professionals aim to minimize harms resulting from carrier identification by providing support for parents and children following NBS. Support for non-genetics specialists from genetic counselors to enable discussion of carrier results with children is suggested.
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http://dx.doi.org/10.1007/s10897-014-9740-5DOI Listing
October 2014

Parents' responses to receiving sickle cell or cystic fibrosis carrier results for their child following newborn screening.

Eur J Hum Genet 2015 Apr 9;23(4):459-65. Epub 2014 Jul 9.

University of Nottingham, Nottingham, UK.

Universal newborn screening for sickle cell disorders and cystic fibrosis aims to enable the early identification and treatment of affected babies. Screening can also identify infants who are healthy carriers, with carrier results being the commonest outcome for parents and professionals to discuss in practice. However it is unclear what the effect will be on parents on being informed of their baby's carrier result. Semi-structured face-to-face interviews were conducted with a purposeful sample of 67 family members (49 mothers, 16 fathers, 2 grandparents) of 51 infants identified by universal newborn screening as carriers of cystic fibrosis (n=27) and sickle cell (n=24), across all health regions in England. Data were analysed by thematic analysis with subsequent respondent validation. Untoward anxiety or distress among parents appeared influenced by how results were conveyed, rather than the carrier result per se. Parents who had more prior awareness of carrier status or the possibility of a carrier result assimilated the information more readily. Being left in an information vacuum while awaiting results, or before seeing a professional, led some parents to fear that their child had a serious health condition. Parental distress and anxiety appeared mostly transient, subsiding with understanding of carrier status and communication with a professional. Parents regarded carrier results as valuable information and sought to share this with their families and to inform their children in the future. However parents needed greater support after communication of results in considering and accessing cascade testing, and negotiating further communication within their families.
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http://dx.doi.org/10.1038/ejhg.2014.126DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4666569PMC
April 2015

Informing children of their newborn screening carrier result for sickle cell or cystic fibrosis: qualitative study of parents' intentions, views and support needs.

J Genet Couns 2014 Jun 4;23(3):409-20. Epub 2013 Dec 4.

School of Psychological Sciences, University of Manchester, Oxford Road, Manchester, M13 9PL, UK,

Newborn screening for cystic fibrosis and sickle cell disease enables the early identification and treatment of affected children, prolonging and enhancing their quality of life. Screening, however, also identifies carriers. There are minimal or no health concerns for carriers. There are, however, potential implications when carriers reach reproductive age, and thus research attention has been given to how best to convey information about these implications in a meaningful, balanced way which does not raise undue anxieties. Most research focuses on the communication from health professional to parent, yet ultimately this information is of greatest significance to the child. This study examines parents' intentions to inform their child of newborn screening carrier results. Semi-structured interviews with 67 family members explored their intentions to inform the child, and related views and support needs. Parents almost unanimously indicated they planned to inform the child themselves. Health professionals were expected, however, to provide guidance on this process either to parents through advice and provision of written materials, or directly to the child. Although parents initially stated that they would convey the result once their child had developed the ability to understand the information, many appeared to focus on discrete life events linked to informed reproductive decision making. The results highlight ways in which health care providers may assist parents, including providing written material suitable for intergenerational communication and ensuring that cascade screening is accessible for those seeking it. Priorities for further research are identified in light of the results.
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http://dx.doi.org/10.1007/s10897-013-9675-2DOI Listing
June 2014
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