Abstract
Objective
Having a child with a cleft lip with or without palate (CL ± P) can greatly impact parents’ lives. Retrospective studies have shown that receiving a CL ± P diagnosis is an emotional event, requiring considerable adjustment. However, there has been limited prospective research on how parents experience pregnancy when faced with a prenatal diagnosis of CL ± P. This study aims to examine the experiences of parents during pregnancy, focusing on their concerns and perspectives regarding cleft care, pregnancy, childbirth, and upbringing.
Design
Participants were interviewed between 24 to 39 weeks of gestational age using semi-structured interviews. Data were analyzed using inductive reflexive thematic analysis.
Setting
Interviews took place through a videocall, a phone call or face-to-face, following participants’ preferences. Interviews lasted an average of 33 min.
Participants
Eleven expectant mothers and 8 expectant fathers were recruited from the cleft team at a university medical center.
Results
Four main themes were identified: “Suspected diagnosis: An emotional rollercoaster,” “After diagnosis: So much information and healthcare professionals,” “No carefree pregnancy,” and “What will the future bring?”. Receiving a prenatal CL ± P diagnosis was experienced as an emotional rollercoaster, accompanied by numerous uncertainties. Parents reported that the cleft care trajectory did not always optimally support them in emotionally processing the diagnosis.
Conclusions
Results highlight a need for specialized cleft care tailored to parental needs and wishes. Specifically, empathetic reactions of healthcare providers are crucial, as well as an additional contact moment for parents before childbirth.
Introduction
In the Netherlands, approximately 1 or 2 in every thousand children is born with a cleft lip with or without cleft palate (CL ± P).1,2 In children with CL ± P, the lip or both the lip and palate do not fuse entirely during the eighth week of gestation.3,4 In the past, CL ± P became apparent only at childbirth. 5 Nowadays, a cleft lip is often detected at ultrasounds conducted before 24 weeks of gestational age,6,7 while a cleft palate remains harder to detect prenatally. CL ± P can occur by itself or as part of a genetic syndrome and almost always requires multidisciplinary healthcare throughout childhood and adolescence, starting with surgeries in the first year of life.4,8
CL ± P influences a child's physical and psychological development in several ways. 9 Physically, children frequently encounter problems with feeding and oral health. They may also require speech therapists and dental health professionals in addition to the multidisciplinary care they already receive.10,11 Psychologically, children may experience lower health-related quality of life and psychological problems later in childhood and adolescence, for which care can be provided by pediatric psychologists.10,12
Importantly, CL ± P impacts not only the child itself, but also their parents. The prenatal diagnosis often is the start of an intense medical trajectory. Several appointments and tests follow the diagnosis, with parents facing many complex and emotional considerations.13,14 An especially emotional consideration is the possibility of termination of pregnancy. The prevalence of CL ± P has decreased since the introduction of the second-trimester anomaly scan in the Netherlands, 2 but there is no direct evidence linking this decrease to termination of pregnancies. Indeed, evidence from the Netherlands and Argentina suggest that most parents do not perceive CL ± P as severe and decide against termination of the pregnancy.15,16
A prenatal diagnosis of a congenital condition can strongly affect parents. Generally, a prenatal diagnosis comes with feelings of loss (eg, of expectations and hopes), a need for clear and trustworthy information, and unease about the pregnancy as well as worries of stigmatization of the child. 17 For CL ± P specifically, retrospective studies have reported grief, feelings of loss, feeling unprepared, guilt, and anxiety.18–20
Previous studies using quantitative data and/or retrospective designs showed conflicting findings on the differences between maternal and paternal responses to the diagnosis.21–23 Prospective cohort studies have indicated that mothers of a child with non-syndromic CL ± P experience higher rates of distress during pregnancy and higher rates of depression and distress after childbirth, compared to mothers of healthy children.24,25 Fathers have not been studied as extensively as mothers, however, preliminary retrospective findings indicate that their experiences seem comparable with those of mothers. 23 Later, during the child's adolescence and young adulthood, parental quality of life may still be impacted by the condition. 26 Parents may experience uncertainty about treatment, distress about possible stigmatization of their child, and may feel as if they have to protect their child from the outside world.19,27 These worries may already start during pregnancy after receiving a prenatal diagnosis.
Importantly, stress and anxiety during pregnancy in general have been related to preterm birth28,29 and maternal stress during pregnancy seems related to children with CL ± P being more prone to developing behavioral problems.30–32 These findings highlight the importance of high-quality perinatal cleft care to support families of children with CL ± P early-on in the medical trajectory, to minimize negative long-term effects on their emotional well-being. To improve the quality of cleft care, a clear understanding is needed of parental experiences on whether cleft care provision is tailored toward their wants and needs.
Therefore, the current study aims to examine how parents receiving a prenatal CL ± P diagnosis experience care provision during pregnancy and to understand their needs and concerns related to the pregnancy, childbirth, and period directly after childbirth. Findings will have important implications for the future care of patients with CL ± P and their parents, and other prenatal congenital conditions. Moreover, our conclusions will be helpful for healthcare professionals that are involved with cleft care.
Methods
Study Design
A qualitative design was employed, with participants taking part in individual semi-structured interviews. This study was performed according to the declaration of Helsinki 33 and written according to the SRQR guidelines. 34 The Medical Research Ethics Committee of the Erasmus MC rated this study as exempt research (MEC-2021-0183).
Participants and Recruitment
Expectant parents were recruited between July 2021 and April 2024 from the Erasmus MC Sophia Children's Hospital in the Netherlands from the specialized cleft team. This team serves as a regional center. Each year, the cleft team conducts an average of 28 prenatal counseling sessions and provides care for 48 newborns. Parents who have not received prenatal counseling often have a child with an (isolated) cleft palate, a condition that is often not detected before birth. 7
Parents were eligible if they (a) received a prenatal diagnosis of CL ± P affecting their child, and (b) were aged at least 18 years old. Parents were excluded if they (a) had insufficient mastery of the Dutch language, (b) the pregnancy was seen as a high-risk pregnancy (eg, due to psychological or physical conditions), or (c) had an unwanted pregnancy. Both mothers and fathers were eligible for participation.
Procedure
A convenience sampling approach was used to recruit participants after referral to the nurse specialist of the cleft team. Parents were informed about the study after prenatal counseling. A few days later, the nurse specialist reached out by telephone to answer questions and assess whether the parents wanted to participate. After agreeing to participate, parents signed an informed consent form. Interviews were scheduled for an approximate duration of 45 min, around 6 weeks before the due date. Interviews were done via Microsoft Teams or face-to-face in the hospital, following parent's preferences. Mothers and fathers were interviewed separately to ensure they felt safe and free to speak about their own feelings and experiences. Parents received a gift card worth €15.00 to thank them for their time and effort.
Interviews
Semi-structured interviews were conducted by the nurse specialist (MvVvdH). A mock interview was carried out by the nurse specialist and (MvD) with a student. Following this mock interview, some questions were changed to allow participants to provide broader, unbiased responses. For example, questions on “Do you have sleeping problems?” or “Do you feel down?” were changed to a more general “How do you feel?”.
Interviews focused on 3 main topics, namely (a) the diagnosis: process in healthcare and prenatal counseling (past), (b) parents’ current situation and emotional well-being (present), and (c) parental expectations or worries about parent-child bonding and expectations about childbirth (future). The interview schedule is shown in Table 1.
Interview Topics and Subtopics.
The participants were already familiar with the nurse specialist who conducted the final interviews, as she had provided their prenatal counseling. During the interviews the nurse specialist encouraged the participants to openly share their thoughts and feelings and emphasized that the care provision would not be influenced by their answers. Participants could withdraw consent at any moment during the interview.
Dutch Care Pathway
There is a significant heterogeneity in healthcare systems worldwide for pregnancy and childbirth. Expectant parents are situated within a specific healthcare context that influences and colors their experiences. A schematic overview of the Dutch care pathway is shown in Figure 1.
Schematic overview of the Dutch healthcare trajectory.
In the Netherlands, a fetal structural anomaly 20-week ultrasound is offered to every pregnant woman and carried out by trained and certified local midwives, sonographers or gynecologists in a (university) hospital. In case of abnormal findings, parents are referred to a fetal medicine center where specialized ultrasound is carried out. Additional genetic tests are offered (via chorionic villus biopsy and/or amniocentesis) to assess the possible presence of a genetic syndrome. In case of CL ± P, an internal referral to the cleft team takes place. Prenatal counseling with the nurse specialist of the cleft team is planned to inform parents about the diagnosis, the clinical pathway, and feeding after childbirth. In this session, parents’ psychosocial circumstances are also discussed.
In the Netherlands, termination of pregnancy is legally allowed until 24 weeks of gestational age. As the additional genetic tests may take weeks to process, parents may find themselves with limited time to make an informed decision about termination. If the pregnancy is continued, no planned contact with the cleft team is scheduled after the prenatal counseling until childbirth. The child's care is continued from childbirth up until 22 years old.
Data Collection and Analysis
Demographic and medical background characteristics (eg, gender of the parent, parity, suspected cleft diagnosis and other relevant pregnancy details) were retrieved from the parents’ electronic health records to provide context to the interviews. In addition, the interviews began by asking about the current situation and if there were other stressors surrounding this pregnancy.
Interviews were audio-recorded, and transcribed verbatim by the nurse specialist, students or a paid transcription service. Inductive reflexive thematic analysis at a semantic level was used to analyze and code the data. 35 This method includes several steps: familiarizing oneself with the data, generating initial codes, searching for themes, reviewing themes, defining and naming themes, and producing the report. The first author completed these steps and made notes of first impressions during the interviews.
Although the first author (MvVvdH) conducted the coding process, she was supervised by the last author (MvD) to ensure a rigorous coding process. For instance, the authors coded the first interview independently and discussed their codes and coding process. The first author then coded all the other interviews, which were discussed by both authors to identify potential themes. The final themes were also assessed for clarity and coherence by a clinical psychologist (JO). NVivo software (release 1.7) was used as supportive software for data analysis. Selected quotes were translated from Dutch to English and edited for readability.
Researcher Reflexivity and Positionality
The interview and analysis process was mainly done by 2 authors. The first author conducted the interviews and analyses, and is the nurse specialist of the cleft team. She has over 16 years of experience providing healthcare to patients with a cleft and their parents. She was supervised by MvD, who is a postdoctoral researcher in pediatric psychology with extensive qualitative and quantitative research experience in supporting children and parents with a visible difference. Other authors have research and clinical expertise in pediatric psychology, gynecology, and craniomaxillofacial surgery.
None of the members of the research team had CL ± P themselves, had a child with a cleft or another visible difference, or had personal experience of receiving a prenatal cleft diagnosis. One of the team members received a different prenatal diagnosis during the write-up of study results.
Results
Participants
Twenty-one parents were assessed for eligibility. A total of 19 expectant parents (11 mothers and 8 fathers), with 11 pregnancies agreed to participate. None of the participants withdrew consent after starting the study. The mean age of the expectant mothers was 33.55 years (SD = 5.12) and fathers were an average of 36.63 years old (SD = 8.93). In 3 cases, expectant mothers had experienced complications in an earlier childbirth. The number of previous pregnancies ranged from 0 to 5 and parents expected their first to fourth child. One pregnancy was a twin pregnancy, in which 1 of the children was expected to be born with a cleft lip. In 2 pregnancies, a bilateral cleft was expected, while for the other pregnancies, a unilateral cleft was expected. More information is shown in Table 2.
Participant Characteristics, with Pseudonymized Names.
Names serve as aliases and do not reflect the actual identities.
Most interviews were conducted between 33 and 39 weeks of gestational age, with one outlier: a parent couple interviewed at 24 weeks as part of an initial attempt to conduct multiple interviews during pregnancy. As this approach proved unfeasible, subsequent interviews were scheduled around 34 weeks of gestational age. Interviews lasted an average of 33 min (range 14-62 min). All participants, except one mother, preferred doing the interview online due to travel distances or other practical considerations. Some interviews were carried out by phone instead of a videocall, due to logistical reasons. Parents were interviewed separately, except for one couple that was interviewed together.
Themes
Four main themes were identified: “Suspected diagnosis: An emotional rollercoaster,” “After diagnosis: So much information and healthcare professionals,” “No carefree pregnancy,” and “What will the future bring?”. See Table 3 for an overview of all themes and subthemes.
Themes and Subthemes as Derived From the Interviews.
Theme 1: Suspected Diagnosis: An Emotional Rollercoaster
This theme reflects parents’ experiences of receiving the diagnosis. Parents often experienced feelings of shock, grief, guilt, and stress. Some parents did not expect a prenatal diagnosis, because non-invasive prenatal testing did not show any abnormalities. In addition, most parents highlighted that waiting for further medical examinations such as advanced ultrasound research in the university hospital, and waiting for genetic results, was hard to cope with or “inhumane.”
Parents reported that, even though the advanced ultrasound research took place a few days after the initial diagnosis, this felt like “waiting forever.” Most parents noted they experienced high levels of stress: “I really was so emotional, I could not think clear, actually I was going crazy, a bit.” (Odilia, mother, 26 years old).
Or felt like they went through a range of emotions: “It was also strange for my other boys. Mommy, why you are staying in your bed? So, you also try to hide things actually. So it really was a rollercoaster of emotions.” (Davina, mother, 35 years old)
Subtheme 1: So Much to Take in
Parents’ experiences with the way they received the suspected cleft diagnosis were mixed. Some parents already noticed the cleft during the ultrasound, before being told by the sonographer. Others were informed of the suspected diagnosis directly by the sonographer.
Healthcare professionals differed in the amount of information they provided when making the referral for a specialized ultrasound. One parent received a “waterfall of medical information” about “extra chromosomes, a chromosomic malformation and the option to do amniocentesis.” Other parents were only informed that something was detected on the lip, without the healthcare professional specifically describing it as a cleft.
In addition to the emotional rollercoaster, parents had a lot of information to process. As one parent highlighted: “We did the advanced ultrasound in the academic hospital, but at that moment there is so much going through your mind. There is so much coming your way, so much information.” (Edward, father, 33 years old)
Subtheme 2: Time and a Personal Approach by Healthcare Professional are Important
Parents experienced the diagnosis at the 20-week ultrasound as the most impactful moment. However, most parents felt somewhat positive looking back at the moment of diagnosis. When asked what was helpful, they noted that not feeling rushed during the procedure and receiving clear messages were important. For instance, it was helpful to hear “something is wrong, there is a cleft and this is not my expertise, so I will refer you for further examination” (Davina, mother, 35 years old).
Parents appreciated a personal approach by healthcare professionals during the medical process; however, the hospital environment may be perceived as somewhat less personal and “more formal.” Leaving parents to feel “less supported.” They also highlighted differences between healthcare providers: “It's a relief because you [the nurse specialist who does the prenatal counselling] brought a certain calmness and a certain kind of clarity. That is what I missed with the professionals before you. I think for the entire process, it's nice that you could take a breather for a bit.” (Geoffrey, father, 37 years old)
Summary Theme 1
In short, this first theme describes how parents struggle with first hearing about the suspected diagnoses. Parents go through an emotional rollercoaster while also dealing with hospital appointments for ultrasounds, genetic testing, and prenatal counseling. During these appointments, parents appreciated a warm and personal approach by the healthcare providers. They also felt that the hospital environment was less welcoming than their previous experiences with their own obstetrician.
Theme 2: After Diagnosis: A lot of Information and Healthcare Professionals
This second theme reflects parents’ experiences after formally receiving the diagnosis, their evolving emotions around the diagnosis and how they coped with the situation. When opting for genetic testing, parents must deal with another period of waiting on test results, thereby extending their feelings of stress and uncertainty. For some parents, it was important to receive more information, which could also help in making (medical) choices: “I just want to know where we stand, because it is quite a thing… And I want to make the right decisions based on the information we have.” (Lex, father, 32 years old)
Dutch regulations state that the termination of pregnancy is possible until 24 weeks of gestational age. Some parents received the results of additional genetic tests just one day before the 24 weeks of gestational age, thereby adding to the already emotional and stressful period. Parents experienced this as especially “nerve-wracking” and “stressful.” One parent described how this made her feel: “It is so much to take in. We already made the choice, if genetic testing gives a negative result, we will terminate this pregnancy. So you are thinking about a funeral instead of a birth announcement.” (Davina, mother, 35 years old)
Subtheme 1: Prenatal Counseling is Reassuring
Most parents experienced the prenatal counseling with the nurse specialist as reassuring and some reported “feeling relieved afterwards.” They appreciated that time was taken to explain the diagnosis, practical information was given, and attention was paid to their thoughts and feelings: “The consult was super nice. We both had a very good feeling: “We have only been there once, but you almost feel like coming home already.” (Lizzy, mother, 34 years old)
One of the parents appreciated that there was one contact person, with whom they could get familiar: “What I thought to be nice…the person who does the prenatal consult, the point of contact then, remains the point of contact. It is always nice to have a familiar face somewhere.” (Livia, mother, 42 years old)
Subtheme 2: Mixed Experiences Toward Additional Ultrasounds
For most parents, the initial ultrasound was followed by further examinations to monitor the baby's development throughout the pregnancy. Several parents experienced these additional ultrasounds as comforting, while others experienced these as intensive and sometimes stressful: “Because you are being monitored so closely, you know so much more which you normally should not have known.” (Hannah, mother, 25 years old)
Subtheme 3: Preparation by Focusing on Practicalities
All parents felt the need to cope with the situation, although they differed with regard to their preferred coping styles. For instance, 1 parent felt the need to retrieve all possible information, describing it as “an obsession”: “I have really dug into it, almost being obsessive. I just want to know what we are facing, as it's not small feat of course. And based on the information there is, I want to make the right decisions, so that's what I tried to do and I hope we will [make the right decisions].” (Lex, father, 32 years old)
Social media helped parents to feel supported and seen in their process, since sharing experiences reduced their feelings of loneliness. Additionally, parents sought support from other healthcare professionals such as a pediatrician, lactation consultant, or pregnancy coach. Other parents focused mainly on arranging the practical aspects and preparing for the childbirth, for instance by already purchasing feeding bottles and exploring options for childcare and work leave after cleft surgery: “I immediately researched things and it turns out I get twenty extra days off of work. And after every surgery there are additional days with care leave. So it's very convenient to have that figured out beforehand.” (Davina, mother, 35 years old)
Overall, parents often desired a feeling of control over the situation.
Summary Theme 2
This second theme describes the overload of information and unfamiliar healthcare professionals. Parent simultaneously have to process all the information, think about doing genetic testing and think about what to do with the results of these tests. Parents appreciated the prenatal counseling, as a first point of contact with the cleft team. Experiences toward additional ultrasounds were mixed, but almost all parents felt the need to regain control as a way to cope with the situation.
Theme 3: No Carefree Pregnancy
This third theme reflects parents’ view on the pregnancy, knowing the child will be born with a cleft. Parents reported many concerns during the pregnancy and felt worried about the period right after childbirth as well.
Subtheme 1: Is my Child Okay?
This subtheme describes the worries parents have with regard to the child's health. Some mothers paid more attention to the child's movements, to see if the child was doing well, as the baby was perceived to be “more vulnerable.” As one parent mentioned: “I regularly kept watching the baby, sometimes I took a seat and then… I didn’t feel the baby move.” (Ruth, mother, 41 years old)
Other parents worried more about the child's health in general: “The midwife told me: it is just the cleft. Overall, it is a healthy child. This is a bit difficult, because you are afraid […], that something will go wrong. Despite the results you have. So really, you are always thinking about this, if you feel something different [in your belly].” (Davina, mother, 35 years old)
Subtheme 2: No Guarantees for the Future
After receiving the diagnosis, parents experienced a highly uncertain time period during which they worried about several topics. They often reported worrying about whether their child would be born with a cleft palate and what the future would look like: “I think you are more worried because you know there is something wrong. […] I just hope that the baby is healthy, while at that moment, if I hadn’t known, I would have assumed this more. […] There are no guarantees, but I think it [not having a prenatal diagnosis] would make you more carefree. (Anne, mother, 32 years old)
Parents also described a feeling of uncertainty toward their future: “When I am sad, it is just a feeling of powerlessness, a bit of guilt, a bit of disappointment and I find it very exciting everything…a baby is quite exciting, but this makes it just somewhat more daunting.” (Lizzy, mother, 34 years old)
Subtheme 3: Unusual Bonding Experiences
Parents highly differed regarding their bonding experiences. Feeling guilty or struggling emotionally was reported by several parents. One mother thought of herself as “mean” because she was so happy to be pregnant but now grieved over the diagnosis. Another mother mentioned delayed bonding with the child. “It took a long time, I was very worried, I didn’t like being pregnant, actually I didn’t bond with the child. But now I feel a bit more positive.” (Odilia, mother, 26 years old)
On the one hand, some parents thought that they subconsciously made less contact with the child, like this father described: “I think I was just too focussed on it. You just don't know what to expect, right? So you look at pictures, which is what I have done, and that really scares you. Of course there are differences, you see a double [bilateral cleft] and a single [unilateral cleft] and different types of clefts. […] And I think subconsciously, you give a little less attention to your girlfriend or the pregnancy.” (Edward, father, 33 years old)
On the other hand, others reported feeling extra protective: “I have an extreme motherly feeling [towards the baby]. I love you even more, because you will get this start. I feel more love and a certain feeling of protection towards the baby.” (Emma, mother, 33 years old)
Others reported being “done with the stress” and wanting to meet the child.
Summary Theme 3
This third theme describes parents’ experiences of the pregnancy as being far from carefree. Despite additional ultrasounds and positive genetic results, expectant mothers were hyperaware of the baby's movements. Both mothers and fathers reported worrying about whether the child would be born with a cleft palate and about the uncertain future. Parents also described an abnormal bonding process, with some parents feeling extra protective and others feeling guilt or shame over their grief for the diagnosis.
Theme 4: What Will the Future Bring?
This fourth theme describes parents’ worries toward the future in terms of their social environment, childbirth, and period after childbirth.
Subtheme 1: How Will Others React?
During the pregnancy, parents thought of how others would react to the child's appearance after birth. Some parents felt supported by their friends or family, while others did not. Parents found emotional support from friends, family, and healthcare professionals helpful, and also valued practical assistance, such as help with childcare for siblings during medical appointments: “Having people watch my children and that kind of thing has really helped me [during the pregnancy]. I feel very supported by others.” (Odilia, mother, 26 years old)
Some parents worried that their child would be bullied and felt they had to protect their child: “That's something that keeps stressing me out and also for the future for her [the child]. With bullying and that sort of thing. Yes, that's something that worries me.” (Edward, father, 33 years old)
Parents also worried about how the cleft would impact their family life, in particular the additional care required and whether they would be able to manage.
Subtheme 2: Will the Baby be Seen as a Child or a Patient?
Most parents did not fear childbirth, except those who had experienced complications during a previous delivery. Some parents worried about their child's breathing directly after birth.
Parents also mentioned that sometimes their midwife or gynecologist questioned the planned location of the delivery (eg, hospital birth in a regional or university hospital, or home birth), which caused feelings of uncertainty in parents. Some mothers chose to take pain relief during childbirth to relieve worries: “During childbirth I probably have enough to deal with, with all the other thoughts in my head.” (Hannah, mother, 25 years old)
Several parents wished to keep their child close after childbirth and hoped to avoid unnecessary medical intervention: “I hope they will lay the baby down on my belly for the first hour and that they won’t do anything that is not necessary at that time.” (Emma, mother, 33 years old)
Subtheme 3: How Will we Feed the Child?
In the last weeks of pregnancy, most parents were curious about their child and the severity of the cleft. Moreover, they worried about potential feeding problems: “I do not worry about childbirth, but the moment after.. the feeding process, which is not self-evident. But then the situation.. it may be choking, or not, I just don’t know yet.” (Lex, father, 32 years old)
Some parents felt grief and guilt because they knew that breastfeeding would probably be impossible in case of a cleft palate: “For me this is the worst…as a mother you really want to give this to your baby.” (Lizzy, mother, 34 years old).
Sometimes, parents compared this to a sibling child for whom breastfeeding was possible and felt like their children were treated differently: “It doesn’t feel fair, because I breastfed my firstborn for a year.” (Emma, mother, 33 years old). On the other hand, a father shared that he would now also be able to feed his child and could therefore bond with his child.
Subtheme 4: A Second Beginning After lip Closure
Some parents looked forward to meeting their child and starting the cleft care trajectory. Others felt like the birth was not the real start of their child's life. They reported that the first surgery would feel more like a new beginning: “I love her, my baby, so much. I shouldn’t bond too much, because she will be different [her appearance].” (Emma, mother, 33 years old)
Summary Theme 4
This last theme describes the worries parents have over an uncertain future. In the near future, parents worry about whether the child will be seen as a patient and about how others will react to the child. They also worry about how they will feed the child, as breastfeeding may not always be a possibility. For the slightly longer term, parents worry about the lip closure and how this may change the child's appearance and their feelings of affection toward the child. Parents also mention worrying about bullying when the child grows up.
Discussion
The current study aimed to examine how parents with a child with a prenatal CL ± P diagnosis experience the cleft care provision during pregnancy. Moreover, we aimed to study parental needs and concerns related to the pregnancy, birth, and period after childbirth. Four main themes were identified following semi-structured interviews with 11 mothers and 8 fathers.
Overall, parents reported receiving an abundance of information during a highly emotional time. They also met many different healthcare providers and appreciated a personal and empathetic approach by these healthcare providers. All parents reported needs and concerns regarding the future, both related to their social environment (ie, how others might react) and to medical issues (ie, feeding the child and managing surgical interventions).
Two major findings are important to highlight. First, parents experienced several worries after a prenatal diagnosis of CL ± P and described the period surrounding the diagnosis as an emotional rollercoaster. This aligns with other studies describing an intense emotional trajectory for parents after a prenatal diagnosis.15–18 In addition, our findings show that parents also worry about the child's further development both during the pregnancy and after childbirth. For instance, parents have practical concerns surrounding feeding, as well as concerns around reactions from others and a potential fresh start after lip surgery. It is imperative for healthcare providers to address these concerns in expectant parents.
Second, results indicated that reactions of healthcare providers are crucial for how parents perceive the cleft care pathway. Clear information, empathy, and enough time for communication about the diagnosis and further trajectory are vital, both when a cleft is suspected in the screening setting and when the diagnosis is confirmed in the hospital setting. Previous studies on information provision surrounding congenital diagnoses have also shown this need for an empathetic response. 5 Providing adequate information, using clear communication, and acknowledging all feelings and emotions help parents cope with the diagnosis.36,37 For healthcare providers, midwives or sonographers who might detect/suspect a cleft in the screening setting, it is important to keep the message short and avoid a “waterfall” of information. To improve the way this message is delivered, training of healthcare professionals can be considered. 38
During interviews, parents often highlighted practical concerns and a need for additional care. The prenatal diagnosis starts an emotional trajectory, with parents receiving extensive information about the condition and care pathway simultaneously. Processing and retaining all this information while experiencing intense emotions can be difficult. 39 Parents would appreciate extra information or repetition of information at a later point during the pregnancy, when they are more emotionally stable. This could be through a personal check-in, but could also be realized by providing information in leaflets or videos that can be accessed at a later time, rather than directly after the diagnosis. 40
While the focus of this study was on prenatal diagnoses, it is unclear how the impact of a prenatal diagnosis compares to the impact of a postnatal diagnosis. Up to a third of congenital conditions are diagnosed postnatally. 41 These parents experience a different care pathway, without the potential stress of additional medical check-ups, but also without the time to prepare for the birth of a child with a congenital condition. Literature directly comparing prenatal diagnoses to postnatal diagnoses is scarce, and results are mixed.25,42 But results seem to indicate no significant differences in the longer term. 43 It therefore remains unclear how the results of the current study compare to those of parents who receive a diagnosis postnatally.
Results can also be seen in the light of literature on bad news conversations in healthcare. In line with our findings, these studies show that building a trusting relationship with the healthcare provider relates to how bad news is received by parents. 44 Bad news should be communicated while taking into account the correct and comprehensive terminology as well as showing empathy and compassion. 38 Studies also show that trust and empathy between healthcare providers and parents develop over time. In the current care pathway, parents typically meet each healthcare provider just once, so an additional moment of contact could improve perinatal care.
This study should be interpreted considering some strengths and limitations. Strengths of our study include a prospective design and including both mothers and fathers, eliciting a variety of views and eliminating hindsight bias. Including fathers is especially important as they may have different needs 45 and are generally underrepresented in scientific research on parents. 23 The first author being a familiar healthcare professional could be considered both a strength and a limitation. Parents may have been more open because of the existing trust bond with the researcher. Simultaneously, it may have been more difficult to fully and honestly express feedback on the care directly to the person that provides it. However, throughout the information letters, consent forms, and interviews it was emphasized that all personal input would be treated confidentially and would only be considered in light of the current study. Additionally, parents were reassured that partaking in the interview would not have any consequences for their care provision. A safe and open space was created during the interview while emphasizing that there would be no right or wrong answers, parents could skip any questions if they wanted to and the interview could be halted at any moment. Moreover, the researcher maintained a calm and listening attitude using open communication and questioning techniques.
A limitation of our study was that no triangulation of the data was done to minimize burden of the parents in the end stage of pregnancy. Also, we did not focus on distinguishing between the experiences of mothers and fathers since this was outside the scope of this study. However, including both mothers and fathers in our study already is a step forward since many studies up until now have focused on mothers, though more general literature indicates that fathers may struggle with their emotional well-being perinatally as well. 46 A last limitation is the generalizability of the study results. As this study was conducted in the Netherlands, characterized by an open culture and specific healthcare context, it is unclear how the results generalize to other countries or cultures.
Future studies should focus on the different experiences of expectant mothers and fathers, so that the cleft care pathway can be adjusted accordingly if needed. Furthermore, future studies should dive deeper into the parental experiences of CL ± P during the first months after childbirth. This would provide further input on how to finetune the cleft care pathway so that parents feel optimally supported throughout the start of their child's life. Lastly, future research can build upon our study by examining the differences in experiences of mothers and fathers, so that perinatal care can be improved accordingly.
Regarding clinical implications, we noticed that almost all parents asked for additional information or clarification after the interview was conducted, around 6 weeks before the due date. Parents reported that, as childbirth approached, they required additional information or had forgotten information they received during the prenatal counseling. Given our findings of parental information overload and previous literature showing that trust develops over time, we recommend an additional moment of contact between diagnosis and childbirth. Moreover, our results highlight many different needs and concerns among parents with a child with a prenatal diagnosis, that cannot be solved by a one-size-fits-all solution. This highlights the need for a close collaboration with psychological healthcare providers, so that parents can be referred for additional mental healthcare if needed. This fits with previous suggestions on optimal multidisciplinary prenatal 45 and cleft care. 12
Conclusion
Receiving a prenatal diagnosis of CL ± P comes with a rollercoaster of emotions, decisions, and worries for expectant parents. Using semi-structured interviews, we identified 4 main themes within the parental experience of cleft care. First, the suspected diagnosis was experienced as a rollercoaster, where communication with healthcare providers was described as crucial. Second, parents struggled with processing all information and practical aspects after receiving the diagnosis. Third, a CL ± P diagnosis interfered with a carefree pregnancy and instead added several worries and uncertainties during pregnancy. Finally, parents often worried about what the future would bring, with regard to both practical and (psycho)social consequences of the condition. To minimize and prevent any negative emotional consequences for parents as well as children, specialized cleft care should be improved by emphasizing clear and empathic responses of healthcare providers and including an additional moment of care at 6 weeks before birth.
Footnotes
Ethical Considerations
This study was rated as exempt from medical ethical approval by the Medical Ethics Review Committee (MERC) Rotterdam MEC-2021-0183.
Consent to Participate
All participants provided written informed consent for participating in this study.
Consent for Publication
Not applicable.
Funding
The authors disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by Vaillant Fonds (no grant number). The funder did not have any role in the data collection, analysis, interpretation of the data or the decision to submit the manuscript.
Declaration of Conflicting Interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Data Availability
The participants of this study did not provide written consent for their data to be shared publicly, so due to the sensitive nature of the study supporting data is not available.