Article Text
PDF
Abstract
Objective To understand parental perspectives regarding universal newborn screening (UNS) for congenital cytomegalovirus (cCMV) in Canada.
Design A qualitative, patient-led study using the Patient and Community Engagement Research approach consisting of online focus groups and in-depth individual interviews to understand parental preferences regarding UNS for cCMV. Data were analysed iteratively using inductive thematic analysis and narrative story analysis.
Setting Canada-wide study conducted via video conference from October to December 2023.
Patients 12 participants from five Canadian provinces who self-identified as 18 years of age or older and as having parental lived experience with cytomegalovirus (CMV) or cCMV participated in the study.
Results We identified three themes: (1) attitudes about UNS for cCMV, including participants’ unanimous support for UNS and confirmation that parental anxiety is not a deterrent for screening, (2) cCMV diagnosis, including the importance of coupling cCMV diagnosis with access to treatment and medical support and (3) awareness of cCMV, where participants shared their frustration about the lack of public and pregnant people’s awareness of cCMV.
Conclusions Parental anxiety is not a deterrent for UNS for cCMV. Children with cCMV and their families deserve every opportunity to attain their best possible outcomes. UNS offers children with cCMV access to early intervention if they need it, and also helps to raise awareness and education to prevent future CMV infections.
- child health
- caregivers
- qualitative research
- virology
Data availability statement
No data are available. Our ethics approval does not allow us to share raw study data.
This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/.
Statistics from Altmetric.com
WHAT IS ALREADY KNOWN ON THIS TOPIC
Congenital cytomegalovirus (cCMV) is the most common non-genetic and preventable cause of birth defects and hearing loss in infants.
The majority of Canadian provinces do not include cCMV as part of their universal newborn screening (UNS) programmes.
WHAT THIS STUDY ADDS
Immense parental support for UNS programmes for cCMV to be implemented nationally as parental anxiety is not a deterrent to newborn screening, parents want the information to be able to make decisions for their family.
Caregivers value early diagnosis as it leads to quick access to medical follow-up, supports and resources for the best possible outcomes for children.
UNS for cCMV may also contribute to raising awareness for cCMV among the general public and pregnant individuals which can lead to prevention of cCMV.
HOW THIS STUDY MIGHT AFFECT RESEARCH, PRACTICE OR POLICY
UNS for cCMV should be implemented nationally in Canada.
Raising awareness about cytomegalovirus (CMV)/cCMV should be a Canada-wide priority as every pregnant person should know what CMV/cCMV is and how to avoid contracting the virus during pregnancy.
Introduction
Cytomegalovirus (CMV) is a common virus that rarely causes concern in healthy individuals.1 However, when a baby contracts CMV in utero, known as congenital CMV (cCMV), mild to severe life-long health and developmental consequences may result for the infant, such as hearing loss, visual impairments, seizures, developmental or motor delay, spina bifida and death.2 cCMV is the most common non-genetic and preventable cause of birth defects and hearing loss in infants.2 3 It is estimated that 1 in 200 unborn children are infected with CMV during pregnancy, and 20% of those infants may go on to develop a permanent disability.2 3 Transmission of CMV occurs through sharing of body fluids.2 Many expectant mothers who are CMV positive, will not pass the virus to their unborn child, and even when they do, there is an approximate 75%–80% chance that there will be no consequences for the child.3 Given this reality, many healthcare providers are hesitant to discuss cCMV with expectant mothers or recommend behavioural changes, such as not kissing their toddler on the lips, as these measures may negatively impact the mother/child bonding and cause undue stress and anxiety.4 Early diagnosis of cCMV is necessary to obtain antiviral medications, detect health consequences and establish a variety of early supports.3
Universal newborn screening (UNS) allows all infants to be screened for various conditions that may not be evident at birth,5 the conditions that are screened for vary by province in Canada.6 The diagnostic tests typically used for UNS for cCMV are saliva, urine or dried blood spots which are tested using PCR and results are usually available within days of screening.7 We found no published studies on Canadian parental perspectives of UNS for cCMV. Studies from the USA,8–10 Italy11 and Finland12 highlight concerns associated with UNS for cCMV,13 including families experiencing distressing feelings after a cCMV diagnosis, doctors not having enough information about cCMV to provide in-depth information to families, adverse effects of antiviral treatments administered unnecessarily to babies, and additional burden on the healthcare system and families from unnecessary doctor visits for newborns who test positive but may not develop health concerns. Despite these findings, parents supported that cCMV newborn screening is beneficial for early detection, and subsequently, the protection of children’s health and development.8 11 13–16 The aim of this study is to explore parental perspectives regarding UNS for cCMV in Canada.
Methods
We used the Patient and Community Engagement Research (PaCER) approach17 to obtain rich descriptions of participants’ thoughts, perspectives and experiences. The PaCER approach is patient-led and consists of three distinct stages, SET, COLLECT and REFLECT.17 SET engages other patients as partners in discussion groups to define the scope and research question. COLLECT includes various data collection activities, such as interviews and focus groups. REFLECT is held with participants from COLLECT to review study findings and ensure accuracy.
Phase 1: SET
Five patient partners with cCMV lived experience attended a 2-hour SET discussion group in May 2023 to define the study scope and research question. Key findings from SET included support for the study direction and research question, the expressed need for increased general awareness about CMV, and the position that UNS is important to diagnose and treat cCMV as early as possible. All research team members attended the SET discussion group; RC and BT cofacilitated.
Phase 2: COLLECT
Recruitment
We strove to recruit a diverse group of parents and expectant parents from across Canada, with varying backgrounds and cCMV experiences. Inclusion criteria included individuals who were 18 years of age or older, self-identified as having parental lived experience with CMV or cCMV, and lived in Canada. Purposive sampling included advertising via social media, CMV Canada, Alberta Health Services Patient and Family Advisory Network, Alberta Strategy for Patient Oriented Research (SPOR) SUPPORT Unit and other SPOR SUPPORT Unit patient networks.
Data collection
We conducted 90 min online focus groups and 30–60 min interviews in October 2023. Our questions were tested with patient partners prior to data collection and related to participants’ lived experience with cCMV, including their cCMV diagnosis journey, reflections on UNS and their insights on how these could be improved for future families. The focus groups and interviews were digitally recorded and transcribed. All participants were given a participant key to ensure anonymity. Consent forms were signed by each participant, and they were reminded of the voluntary nature of this study and their right to withdraw at any time. One team member, II, was responsible for all communication with participants.
We held two focus groups, SP and LB facilitated the first focus group and II and BT the second one. We offered individual interviews for those who were unable to attend focus groups, or who preferred an individual setting. Two team members were present for each interview; one facilitated, while the other took notes.
Data analysis
Focus group data were analysed thematically to identify and analyse patterns of meaning from the data and shed light on ideas and concepts that arose.18 For this study, we defined a theme as a specific pattern of explicit and/or implicit meaning evident in the raw data. The team reviewed the transcripts and individually created codes. We then met to review and consolidate the codes into a codebook. Team members used the codebook to code independently before meeting to establish consensus. Once coding was complete, themes and subthemes were identified and defined.
We used a narrative analysis to analyse accounts shared by participants under the assumption that people organise their daily experiences and interpretations of reality in the form of narratives that give shape to their lives and identities.19 Narrative data collected during interviews were examined for setting, timeline, characters, plot and positioning.19 As a team, we reviewed notes, transcripts and recordings from the interviews and captured two or three main narratives from each interview using quotes from the transcript to substantiate the themes. Narrative analysis added additional context to the thematic analysis.
Phase 3: REFLECT
We invited COLLECT participants to attend REFLECT sessions in December 2023 to review the study findings and provide feedback on our reflections and recommendations. Two participants attended a 90 min focus group, and we held five individual interviews.
Patient and public involvement
This study was conducted by patient research partners; two had lived experience with cCMV. During our SET stage, we worked with five additional parents with cCMV experience to codesign the study. Results were analysed by patient research partners and presented back to participants during the last phase, providing participants an opportunity to confirm themes and provide additional insights prior to finalising results.
Results
12 participants with parental lived experience with CMV/cCMV shared their unique and diverse experiences from five Canadian provinces (see table 1).
Self-reported characteristics of study participants (n=12)
We identified three themes from the data: (1) attitudes about UNS for cCMV, (2) cCMV diagnosis and (3) awareness of cCMV. These themes and their subthemes are presented in the following sections.
Attitudes about UNS for cCMV
This theme captured participants’ thoughts and opinions regarding UNS for cCMV. It included subthemes of ‘support for universal cCMV newborn screening’ and ‘anxiety is not a deterrent’ that validate the immense support for the implementation of UNS for cCMV (see table 2). Our participants shared their concerns that not offering screening for cCMV could be considered unethical in that it unnecessarily denies families and caregivers important medical information about their baby and the opportunity for early intervention and treatment. Further, participants shared their experiences that protocols are in place for healthcare providers to follow when a cCMV diagnosis is confirmed to prevent overtreatment.
Subthemes for ‘attitudes about universal newborn screening for cCMV’
cCMV diagnosis
This theme describes participants’ experiences with their child’s diagnosis of cCMV. Participants reflected on pregnancy experiences and their pathways to diagnosis including the impact of screening, medical processes, communications with medical professionals, variances in health and development impacts, emotional impacts of the diagnosis, and supports and resources needed postdiagnosis. Participants described a broad range of experiences that resulted in three subthemes: ‘quick access to medical follow-up postdiagnosis’, ‘supports and resources’ and ‘medical professional communication’ (see table 3). One participant expressed that her infant began antiviral treatment at 3 months and does not believe it is a coincidence that her development is consistently 3 months delayed. Another participant shared that her daughter was diagnosed with a mild hearing loss, however, by the time they began the antiviral treatment 1 month later her daughter’s hearing loss was profound; both comments relate to the need for early diagnosis and quick access to medical treatment. Participants also related their experiences with varying levels of support (eg, psychosocial) and resources (eg, education, information) that were available and/or provided during their diagnosis journey. Some participants shared that they felt overwhelmed and alone without sufficient support and resources, whereas others expressed gratitude for the comprehensive and coordinated support and resources they received.
Subthemes for ‘cCMV diagnosis’
Awareness of cCMV
This theme encompassed the participants’ thoughts regarding the overall lack of education and awareness around CMV/cCMV and was consistently reinforced by all participants. Three subthemes captured participant experiences: ‘general lack of awareness about CMV’, ‘lack of awareness about CMV infection during pregnancy’ and ‘UNS for cCMV improves awareness’ (see table 4). Participants expressed frustration and/or anger about not being informed of CMV/cCMV during pregnancy. Participants shared that they followed healthy pregnancy guidelines, but their providers did not inform them about cCMV, so they did not know about preventive measures that could have profoundly changed their children’s future health and development. Some participants shared feeling guilty about transmitting CMV to their fetus, and that guilt was longstanding, even while acknowledging they had not known how to protect them.
Subthemes for ‘awareness of cCMV’
Recommendations
Participants identified three key recommendations during COLLECT, which were confirmed and refined with participants during REFLECT (see box 1). Participants expressed deep passion about the third recommendation and suggested specific campaigns for targeted audiences, including: (1) provide pamphlets to centres where expectant families visit (ie, doctor’s offices; public health units; daycares, etc), (2) present at medical/nursing schools, public health or related conferences, (3) develop digital content and infographics for medical, childcare and/or provincial healthcare websites, (4) engage traditional media, (5) post on social media (ie, public health, ‘mommy and me’ sites, etc) and (6) inform public health officials and government ministries.
Recommendations
Universal newborn screening for cCMV should be implemented nationally in Canada.
Medical professionals should provide coordinated and comprehensive resources and support at the time of the CMV/cCMV diagnosis, with established protocols and processes to promote ease of access and support positive diagnosis.
Raising awareness about CMV/cCMV should be a Canada-wide priority. Every pregnant person should know what CMV/cCMV is and how to avoid contracting the virus during pregnancy.
CMV, cytomegalovirus; cCMV, congenital cytomegalovirus.
Discussion
To the best of our knowledge, this is the first patient-led, Canadian, qualitative research study to explore parental preference regarding UNS for cCMV. Participants experienced various pathways to their CMV/cCMV diagnoses, however, regardless of how participants received their CMV/cCMV diagnosis, and whether their children were experiencing cCMV-related health and developmental impacts, they unanimously supported UNS.
The support for UNS for cCMV aligns with other studies, in which most participants supported UNS and that the initial worry caused by UNS is not a reason to not screen or discuss it with parents.8–10 20–22 Our participants strongly expressed that anxiety should not be a deterrent for UNS. Our participants agreed and recognised that receiving a cCMV diagnosis could cause undue stress, worry or anxiety for some families whose children may not develop cCMV related health or developmental impacts; however, the benefit of avoiding worry for some parents does not outweigh the cost of lost opportunities for other children’s early intervention and treatment. Our participants expressed that knowledge is power, and that knowing about a cCMV diagnosis gives families the best opportunity to support their children to become the best version of themselves.
Additionally, participants stated that quick access to treatment and early intervention is essential to prevent or lessen the development of cCMV symptoms. They also noted that unnecessarily withholding this information and associated interventions would be unethical. Although participants were unanimous in their support for UNS, some participants’ support was at least partly contingent on health systems adequately supporting families throughout the cCMV diagnosis journey and beyond. It will be important to ensure that support and resources are provided and/or available, given the increased demand that UNS for cCMV will create.
Our findings call for medical professionals to be supportive, informative and strongly consider how they deliver a cCMV diagnosis to families, as the news impacts them deeply and are consistent with previous findings.10 13 Some participants experienced excellent medical care and support, other participants struggled with the way their health professionals communicated their cCMV diagnosis. UNS for cCMV requires increased professional education and training, as more health professionals will be supporting families through the cCMV diagnosis and follow-up journey.
Although the Society of Obstetricians and Gynaecologists of Canada’s practice guidelines recommend educating pregnant people on CMV infection and hygienic measures to prevent acquisition during pregnancy,23 our participants had significant concerns regarding the lack of cCMV awareness during pregnancy. Whether diagnosed years ago or recently, participants asserted that the ongoing lack of awareness is unacceptable and expressed frustration and/or anger about not being informed of CMV/cCMV. It was difficult to reconcile the effects of cCMV in their children and realising transmission could have been prevented. As cCMV becomes part of newborn screening programmes, media and health ministries may pay more attention and there may be increased research interest, therefore, UNS could be a catalyst for widespread awareness.
Our recommendations align with our three themes and could be implemented using the integrated care model.24 According to the WHO, integrated health services allow people to receive a continuum of health promotion, disease prevention, diagnosis, treatment, management, rehabilitation and palliative care, coordinated throughout their lives.25 In relation to the integrated care model,24 UNS for cCMV would potentially prevent or lessen the severity of health and developmental impacts on children with cCMV by facilitating a quick diagnosis, access to treatment and disease management, and provide for coordinated short-term and long-term supports for symptomatic and asymptomatic children.
We ensured the dependability of the study by consulting patient partners at the outset, developing a detailed protocol, documenting the process and changes, coding data individually and then collaborating to establish data consensus and member-checking the post-data analysis to validate results. Confirmability was achieved through individual reflection as well as team discussions about bias. This study included several male/paternal perspectives which is an area that not many other studies included. Our study had a small number of participants and would have benefitted from more diverse perspective additional perspectives of caregivers whose children had received a cCMV diagnosis, but who did not currently exhibit symptoms. However, qualitative research aim is to not reach generalised findings, but to gather rich descriptions of their thoughts and perspectives to further understand their lived experiences.26
Children with cCMV deserve every opportunity to realise their best possible outcomes. UNS offers children with cCMV an effective and low impact opportunity for early intervention, as well as short- and/or long-term follow-up. Screening can promote comprehensive and coordinated support and resources from well-informed medical professionals, and lead to increased public awareness and education to prevent future transmission, as well as equity to better outcomes.
Data availability statement
No data are available. Our ethics approval does not allow us to share raw study data.
Ethics statements
Patient consent for publication
Ethics approval
This study involves human participants. On receiving approval from University of Calgary Conjoint Health Research Ethics Board (Ethics ID number REB23-0878), the remaining two stages of the PaCER research process commenced. Participants gave informed consent to participate in the study before taking part.
Acknowledgments
The authors are incredibly grateful to all the participants for their interest and involvement, and to the patient partners with CMV lived experience who helped us codesign this study. We would also like to thank Alberta Health Services, Maternal Newborn Child & Youth Strategic Clinical Network for sponsoring our project and CMV Canada for their support in recruitment of patient research partners and participants.
Footnotes
X @pembanetwork
Contributors All authors have substantially contributed to this article and have been involved in writing and revising it. LB prepared the manuscript with input from all authors. The study was conceptualised by EC and MB. LB, BT, RC, SP, II, CL and MB designed the study, collected and analysed the data and prepared the findings. IN, MB, MJS and EC provided methodology and study support. All authors have read and agreed to the published version of the manuscript. EC is the guarantor of this work.
Funding This work was funded by the Strategic Clinical Networks of Alberta Health Services.
Competing interests EC is on the scientific committee for CMV Canada, however, this link had no bearing on data analysis or publication. There are no relevant financial disclosure to be made for any of the authors in this manuscript.
Patient and public involvement Patients and/or the public were involved in the design, or conduct, or reporting, or dissemination plans of this research. Refer to the Methods section for further details.
Provenance and peer review Not commissioned; externally peer reviewed.