Perspectives of women living with type 1 diabetes regarding preconception and antenatal care: A qualitative evidence synthesis

Abstract Introduction Pregnant women with type 1 diabetes may have an increased risk of complications for both the baby and themselves. Educational programmes, preconception planning, strict glycemic control and comprehensive medical care are some of the antenatal interventions that have been proposed to improve the outcomes of pregnant women with type 1 diabetes. While some evidence‐based recommendations about antenatal care are included in clinical practice guidelines (CPGs), the views, and experiences of women with type 1 diabetes about these interventions are not well known. Aim To understand and synthesize the perceptions of women with type 1 diabetes about the interventions before pregnancy. Method A qualitative evidence synthesis (QES) was carried out with a framework analysis guided by the Cochrane Qualitative and Implementation Methods Group approach. Three online databases (Medline, Embase and Web of Science) were searched. We included qualitative articles that were published from 2011 to 2021 and which were available in English or Spanish. Findings Ten references met the inclusion criteria of the study and were included. Three main themes were identified: (a) acceptability of antenatal care, (b) feasibility and implementation consideration and (c) equity and accessibility difficulties. Conclusion Continuity of care, coordination between health professionals and services, and a more holistic approach are the key aspects women say need to be considered for more acceptable, feasible and equitable preconception and antenatal care. Patient or Public Contribution This QES was carried out as part of the CPGs on diabetes mellitus type 1, carried out as part of the Spanish Network of Health Technology Assessment Agencies. In this CPG, the representatives of the patient associations are Francisco Javier Darias Yanes, from the Association for Diabetes of Tenerife, who has participated in all the phases of the CPG; Aureliano Ruiz Salmón and Julián Antonio González Hernández (representatives of the Spanish Diabetes Federation (FEDE) who have participated as collaborator and external reviewer, respectively.

and Julián Antonio González Hernández (representatives of the Spanish Diabetes Federation (FEDE) who have participated as collaborator and external reviewer, respectively.

K E Y W O R D S
preconception care, pregnancy, qualitative evidence synthesis, type 1 diabetes

| INTRODUCTION
Women with type 1 diabetes mellitus (T1DM) can have healthy pregnancies, but they may experience additional challenges in managing their disease.Poor control of diabetes during pregnancy may lead to increased problems for the baby and the mother.2][3] If blood glucose levels are not well controlled, the newborn is at higher risk of hypoglycemia, hypocalcemia, hyperbilirubinemia and polycythemia. 2,3In addition, women with T1DM present a high caesarean delivery rate. 4Therefore, pregnancy usually increases anxiety and stress for women with T1DM. 5 Careful monitoring of blood glucose levels and detailed planning of daily activities are necessary for a healthy pregnancy.[8] Several interventions before the pregnancy have been developed to improve the outcomes of pregnant women with T1DM.Some educational interventions, preconception planning, strict glycemic control and comprehensive medical care can reduce maternal, foetal and pregnancy risks. 9In this regard, there are studies showing that improving preconception care for women with pre-existing diabetes can diminish adverse outcomes. 10,11The American Diabetes Association 12,13 estimates that preconception counselling can reduce the incidence of major congenital malformations from 9% to 2%.A recent systematic review 11 concluded that preconception care for women with pregestational type 1 or type 2 diabetes mellitus is effective in decreasing congenital malformations, and improving the risk of preterm delivery and admission to the neonatal intensive care unit.In addition, this care probably reduces maternal HbA1C in the first trimester of pregnancy, perinatal mortality and the cases of small for gestational age births. 11However, although the impact of preconception counselling on cognitive, psychosocial and behavioural outcomes, as well as its cost-effectiveness have been assessed for policy and implementation decision-making, the perspectives of women with T1DM are considered less in the process.Women with T1DM are experts in their disease and self-care, but at the same time, they are in a situation in need of care in the context of the challenges of pregnancy. 14A better understanding of their perspectives can play a crucial role in relation to the acceptability, feasibility and equitability of antenatal care interventions.This is of particular relevance when developing clinical practice guidelines (CPGs).CPGs are evidence-based, clearly written and easily accessible to clinicians.However, well-developed CPGs and effective CPG implementation methods are needed, as both development and implementation need to be improved to have a better impact in clinical practice.To improve the impact of the recommendations, an important aspect might be to include the patient's views, since patient and public involvement is considered an essential element of trustworthy guideline development. 15Thus, the goal of this article is to address what the women's perceptions are about the interventions before pregnancy that can be recommended for T1DM women.The research was carried out as part of the development of the CPG on diabetes mellitus type 1, funded by the Spanish Ministry of Health.

| METHODS
A qualitative evidence synthesis (QES) was developed with a framework analysis guided by the Cochrane Qualitative and Implementation Methods Group approach. 16Enhancing transparency was used in reporting the synthesis of the qualitative research checklist (ENTREQ) to guide the reporting of this QES. 17which can be found in Supporting Information: File S1.The framework analysis used the categories of acceptability, feasibility and equity from the Evidence to Decision framework from Grading of Recommendations Assessment, Development and Evaluation. 18,19This framework sets out research questions that can guide the comprehension of each category (Table 1).
A scoping search in Pubmed, CINHAL and PSYCINFO was performed to gain an overview of the existing literature.The following two search strategies were used: 'type 1 diabetes mellitus and (preconception care or preconception intervention or prepregnancy care) and (qualitative or interview or focus group)' and 'type 1 diabetes mellitus and (preconception care or preconception intervention or pre-pregnancy care) and (acceptability or feasibility or equity or ethics)'.
The scoping search helped refine and test a systematic search.
Additionally, the scoping phase showed the necessity to include a more general and introductory theme that collected the experiences of T1DM women to contextualize the framework research questions.
The literature in Spanish and English was searched in Medline, Embase and Web of Science.The search was limited to the last search strategies.References of the included studies were screened to find potential additions.
Two researchers independently screened each reference for eligibility, first by title and abstract and then by reading the full text.
References were included if they addressed the objectives of this review, used qualitative techniques and reported qualitative findings separately.Studies in languages other than Spanish and English were excluded.Disagreements were resolved by discussion within the team.The complete inclusion and exclusion criteria can be found in Table 2 and a Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) (Figure 1).
A thematic synthesis adapted from Thomas and Harden 21 was carried out within the framework analysis using the software Nvivo12 ® to support the process.The whole team independently coded a sample of two studies each to develop an initial code book based on the abovementioned framework for this study extended with deductive codes from the included studies.The code book was then discussed within the team and used to code the rest of the studies.The final version of the code book can be seen in Table 3.
Two independent reviewers extracted all relevant qualitative findings in each of the studies and descriptive themes were generated and discussed among the whole team.A data reporting form was prepared according to the selected framework and the cited categories of acceptability, feasibility and equity.
The research team has some experience that influenced the analysis of the results of the QES.The team included mothers and a person with T1DM, certified nurses, anthropologists and a bioethics T A B L E 1 Frameworks research questions.

Acceptability
Is the intervention acceptable to key actors?
• Are there key stakeholders that would not accept the distribution of the benefits, harms and costs?
• Are there key stakeholders that would not accept the costs or undesirable effects in the short term for desirable effects (benefits) in the future?• Are there key stakeholders that would not agree with the values attached to the desirable or undesirable effects (because of how they might be affected personally or because of their perceptions of the relative importance of the effects for others)?• Would the intervention adversely affect people's autonomy?
• Are there key stakeholders that would disapprove of the intervention morally, for reasons other than its effects on people's autonomy (e.g., in relation to ethical principles such as no maleficence, beneficence or justice)?
Feasibility Is the intervention feasible to implement?
• Is the intervention or option sustainable?
• Are there important barriers that are likely to limit the feasibility of implementing the intervention (option) or require consideration when implementing it?

Equity
What would be the impact on equity?
• Are there groups or settings that might be disadvantaged in relation to the problem or options that are considered?• Are there plausible reasons for anticipating differences in the relative effectiveness of the option for disadvantaged groups or settings?• Are there different baseline conditions across groups or settings that affect the absolute effectiveness of the intervention or the importance of the problem for disadvantaged groups or settings?• Are there important considerations that should be made when implementing the intervention to ensure that inequities are reduced, if possible, and that they are not increased?
Note: Adapted from Moberg et al. 19 T A B L E 2 Inclusion and exclusion criteria for the selection of the studies.expert.This expertize helped clarify doubts and the contextualization of the findings.

The characteristics of individual studies were collected in a table
specifically designed for this review.The table includes the first author, year of publication, country, aim of the study, qualitative study design, sample, setting and methodological limitations of the study.The CASPe checklist was used as a critical appraisal tool to assess the methodological quality of the studies. 22

| RESULTS
Eighty-four were selected citations which included 21 duplicates.
Sixty-three articles were screened by title and abstract and 39 references were assessed for eligibility by reading their full texts.Six additional references were found in those full-text articles.Ten references were finally included.Figure 1 shows the PRISMA flow diagram for the selection of the studies.
The characteristics of the included studies are shown in Table 4.
Studies were variable in terms of setting, aim, qualitative design and population.All studies were set in high-income countries.Six studies took place in the United Kingdom, two in Sweden and one in Australia and one in the United States of America.All studies used interviews and/or focus groups as data collection techniques.The population included women with T1DM at childbearing age, nonpregnant, pregnant or those already had given birth.Most studies included none or few women of diverse ethnicity [25][26][27][28][29][30][31][32] or did not collect ethnicity date 23,24 and there was little information about other social determinants of health.The quality assessments of the studies are summarized in Supporting Information: File S3. 20 For more information, visit: http://www.prisma-statement.org/.PRISMA, Preferred Reporting Items for Systematic Reviews and Meta-Analyses.
T A B L E 3 Code book.

Category Codes Description
Theme: Acceptability Support Professional support Relationships and communication with health professionals.

Informal support
Family and social support received during pregnancy planning and development.
T A B L E 4 Characteristics of the studies.Focus group.Women with with T1DM who had also experienced pregnancy, breastfeeding and nurturing of children (n = 3).
A medium-sized hospital.
The design and use of qualitative methodology is partially adequate.
The sample is very small and the data collection technique is not described accurately.Pregnancy with diabetes requires hard work to manage optimum glycemic levels, which includes feelings of pressure, mental effort and anxiety due to the risk to the foetus. 27,30,31But women with T1DM and children also consider pregnancy doable and worth the effort. 29e desire to get pregnant and have children is full of fears and anxiety for many women with T1DM. 28Pregnancy planning is important for women with T1DM who want to become pregnant and this can take them anywhere from a few days to years. 27,28,31ese experiences are expressed in the following quotations:

| Acceptability of preconception and antenatal care
25][26]31 There is an important variability about what women consider adequate information regarding diabetes and pregnancy; some women demand more information, others prefer not to know many details because this can generate anxiety and or feelings of being overwhelmed. 25,27,28,32Timing of information is also important, since women sometimes receive a lot of information at a prenatal appointment, and they consider that it may be more helpful to receive such information further into their pregnancy. 3225][26]31 Women with T1DM receive spontaneous pregnancy advice from an early age.These early tips are welcome in helping plan for a healthy pregnancy. 23However, on many occasions, women with T1DM receive discouraging or fear-mongering messages from both health professionals and other people.[29] The following vignettes allow us to illustrate the above: As a child when I was diagnosed, at that time, I was told that I may never have children and that if I was to have children that perhaps the last 3 months of pregnancy would be spent in Melbourne [major city], in bed and restricted and things like that, so having children was always a scary, scary thing for me.

| Feasibility and implementation considerations for preconception or antenatal care
Lack of awareness about the importance of preconception care are some of the barriers for feasibility.Not all women with T1DM are aware of the risks of pregnancy.Awareness is an incentive to attend antenatal care. 25,28The most common reason for women's attendance was for a referral. 25In addition, women who had lost their babies in previous pregnancies sought preconceptual help for a new pregnancy. 30Another barrier for feasibility might be the lack of support from their peers.Women who had not received support from their peers said they wished they had.Thus, access to antenatal care may be improved through contact with other women in the same situation, and sharing experiences, which seems to be important for women. 23,24,29Health care professionals working with young diabetic patients should contemplate hosting discussion groups for mothers and nulliparous women. 29e third barrier identified is the need for specialized, qualified or trained health care professionals in both diabetes and pregnancy.
Health professionals who are not specialized in diabetes and care for women with T1DM during pregnancy do not always have the necessary skills or training to help these women properly with their pregnancy management. 24,27nally, the continuity of care, coordination between health professionals and services, a more holistic approach in identifying individual needs and recognizing patients as experts in their own condition can alleviate women's frustrations with the medical model of care is a key aspect for women. 24,25,28,31,32The midwife's role is considered fundamental in the provision of normalized care for pregnant women. 31ese are some of the quotations that illustrate these ideas:

| Equity and preconception or antenatal care accessibility
The included studies did not explore the specificities of the experiences of women from ethnically, educational or socioeconomically diverse backgrounds.Access was the main topic related to equity.Accessibility to pre-conception clinics on a regular basis has barriers, such as the adapting working hours, being unable to park or the unpredictably long waits and limited levels of experience with diabetes and pregnancy, particularly in rural areas, were perceived.
Referral seems to improve accessibility. 25,27ese ideas can be followed through these quotations: There was a lot of fear and I guess that's because of a combination of a lack of knowledge, lack of resources, lack of networks and fear of litigation.(King, 2007)   Basically, he's about the only endocrinologist on the Coast, you just don't have any choice here … I've actually only seen him twice … I would like to have someone to be talking to more regularly about the diabetes.(King, 2007)  The findings of the present study show the variety of experiences that women with T1DM have in relation to antenatal care.Thus, the main results confirm that (1) preconception and antenatal care might be acceptable to the majority of women with T1DM, with some exceptions; (2) antenatal care may improve with individualized care, continuity of care, coordination within the health care system, and peer support; and (3) to increase equitability, preconception care needs to be improved in rural areas.Although in a previous review Earle et al. 25 explored views on the provision of, and facilitators of and barriers to the uptake of, preconception care through qualitative research, the present study is the first QES analyzing acceptability, feasibility and equity addressing a research gap.In a recent paper aimed at discussing solutions to improve antenatal care quality, access and delivery, the authors stated that more attention should be paid to a fuller understanding from the user's perspective, that should be inclusive, and that this could help to reduce some of the barriers to quality care. 33periences of care should be taken into account when developing CPGs and making evidence-based recommendations to support a better implementation of these recommendations.In this regard, a QES can provide decision-makers with additional evidence to improve their understanding of the complexity of the interventions, contextual variations and further understanding of values, attitudes and experiences of those who receive the interventions or who implement them. 16In line with person-centred care frameworks, 34,35 the results here point to the importance of a more holistic approach, individualized care, continuity of care, better coordination between health professionals and services and one in which pregnant T1DM women can be recognized as experts in their diabetes, while having more focus on the pregnancy itself.In diabetes care, personalized care planning has a proven small positive effect in measured glycated haemoglobin (HbA1c), when compared to usual care. 36Person-centred care can be implemented by integrating the elicitation of personalized goals, preparing a care plan that includes the care delivery process and the monitoring of the goal attainment. 34To ensure continuity of care, professional and interdisciplinary cooperation at the micro, meso and macro levels needs to be enhanced. 37In conception care for T1DM women, the role of the midwife is central but needs to be coupled with specialized diabetes care.
The feasibility of person-centred antenatal care for women with  38 A study evaluating a regional antenatal care programme may show savings when the excess costs of adverse pregnancy outcomes are taken into account .

39
1][42] Recruitment seems to be an important consideration as, even in studies with a focus on ethnic diversity, participants ended up being mostly white. 25,43Nevertheless, the need to invest in improving accessibility to professional care with experience in diabetes and pregnancy in rural areas is clear.In this sense, a recent systematic review exploring rural health care delivery and maternal and infant outcomes for diabetes in pregnancy. 44shows a gap in published research in the matter as it identified only two studies on such interventions. 45,46Both models proposed a specific model of care adapted to rural areas.Only one, Murfet et al. 45 reported an improvement in neonatal outcomes and did not increase the number of specialist referrals by forming a multidisciplinary team coordinated by a nursing practitioner which included a dietitian, diabetes educator, obstetrician and antenatal nurse.

| LIMITATIONS
This study has some limitations.no grey literature was included, which may have excluded some relevant articles.However, due to their relevance, articles before 2011 were added after checking the references.Third, it was not possible to register the protocol of the review due to a pressing deadline for the CPG, but a version in Spanish can be provided.

| CONCLUSIONS
The findings here show that preconception care may be acceptable to the majority of women with T1DM, although the importance of individualized care and trusting relationships with the professionals to improve acceptability should be mentioned.Continuity of care, coordination between health professionals and services and a more holistic approach are key aspects for women that need to be considered for more feasible antenatal care.Finally, in rural areas, limited levels of experience with diabetes and pregnancy were perceived, which can mean inequitable access.Antenatal care is highly variable and dependent on many factors, such as the geographical area or the professionals' training.More protocols are needed to support women with T1DM in prepregnancy interventions and during pregnancy, taking into account issues of acceptability, feasibility and equity.

First
23  Sweden To analyze the experiences of pregnant women and mothers with T1DM using a prototype of the MODIAB-web by reporting on the communication between the women, their midwives, and the researchers and data programmers as part of this pilot programme.

T1DM requires the promotion
of awareness of the need for such care and the planned interventions, improved pathways for patients and training for professionals.A full assessment of feasibility could be complemented by a review of studies that consider the perspectives of the different health professionals (midwives, diabetes specialists and others) and settings involved (primary/specialist care or antenatal clinics), and the costs of the interventions.Health care professionals may need training and communication skills to provide personcentred T1DM antenatal care.
First, only studies in Spanish and English were included, which may have excluded relevant works in other languages and contexts.Most of the articles included were from anglo-saxon or European countries.Nevertheless, local stakeholders such as patients, patient organizations or health care professionals participating in the development of the CPG discussed and contrasted the findings to adapt the recommendations to the local context.Second, due to the need for rapid GPC recommendations, the search was restricted to the period from 2011 to 2021, and those who work with diabetes and pregnancy know the facts of how diabetes works, but they can never understand the feelings that are involved, how you make it work in your everyday life, they can only provide tips about how others deal with it, it's easier to talk to someone who is actually in the exact same