A support package for parents of excessively crying infants: development and feasibility study

Identification and selection of interventions to show to focus group parents

As set out in the study protocol, the review was based on an update of the 2011 systematic review by Douglas and Hill2 concerning the causes of excessive crying and interventions to manage term infants who cry excessively in the first few months of age. For the purposes of this review, ‘excessive crying’ was taken to mean any crying behaviour that was considered problematic by parents.

Full details of the inclusion criteria and search strategies are provided in Appendix 2. The materials identified through the initial search were reviewed against additional criteria to identify a selection of interventions that could be viewed and rated by focus group parents to inform the development of the intervention for this study. These criteria were:

• intervention materials available in a published and exportable form

• delivery costs that enabled the potential adoption of the intervention within the NHS

• at least provisional evidence of clinical effectiveness

• offering a variety of styles of presentation.

Contracts with the organisations involved in producing these interventions were arranged to allow the website and supplementary materials to be reviewed by the focus group participants.

Identifying evidence for interventions that support parents experiencing postpartum distress

As noted in Aims, the literature review also aimed to review recent evidence for interventions found to be effective in supporting parental coping and mental health, particularly during the postpartum period. This was based on the premise that, as noted in Chapter 1, Background, infant crying can be viewed as a stressor and its impact on parents is, therefore, partly attributable to how parents evaluate and cope with it and, consequently, to parents’ underlying vulnerabilities, circumstances and resources. The strategies described above were used to identify relevant research in this area.

Participants

A purposive sample of 20 parents participated in stage 1. The eligibility criteria for parents were:

• living in the LPT area

• previously had a healthy baby whose excessive crying in the first 6 months of age had caused concern for either parent

• their baby was no longer excessively crying

• English speaking or supported by an English speaker.

Parents whose baby was still crying or was judged to be ill at the time of crying by their HV/SCPHN or another qualified professional were excluded from participating. Parents who lived outside the LPT area were also excluded.

Participants were recruited to the study by two routes:

1. They were referred to the study team via their HV/SCPHN, having previously spoken with them about their baby’s crying.

2. They contacted the study team directly after seeing the call for eligible parents on the NCT website or on flyers distributed to local children’s centres in which HVs/SCPHNs were based. An example recruitment flyer can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018).

All parents received high-street shopping vouchers in acknowledgement of their participation; one was given at the end of the focus group session and another was given when they reviewed the developed package.

Table 2 shows the sociodemographic profile of participants, noting that complete information was not available for two parents. The two male parents attended with partners. Most parents were white British, had an undergraduate or postgraduate degree and were married or living with a partner at the time when their baby cried excessively.

Materials

A short participant demographic questionnaire was devised to obtain participant sociodemographic details prior to the focus groups. The questionnaire can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018). A brief semistructured topic guide was also developed (see Appendix 3), dividing the focus groups into two parts. Part 1 involved explaining the group aims and procedures, and introductions (participants could use pseudonyms if they wished to maintain anonymity). Participants were asked to describe their experience of having a baby who cried excessively: what was most challenging?; what helped?; what sorts of support would they have liked to have received at the time?; and their thoughts on what should be included as part of routine NHS care.

Part 2 involved parents completing rating scales to establish the best way of delivering a package of support (i.e. website, leaflet, direct and/or telephone contact with a professional) and which electronic devices they used to access information and support. Materials from the four example support packages identified by the literature review were evaluated by participants, using five 5-point rating scales, with an opportunity for participants to make written comments on features that they felt were important, they disliked or they felt could be improved. These questionnaires can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018). All four example packages included a website, together, in some cases, with supplementary materials. Sample materials from each package were shown to parents with a changed presentation order for each session to reduce order effects. Appendix 1 provides details of the materials shown and how these were sampled from the example packages.

Safeguarding procedures

Together with LPT’s safeguarding officers, HVs/SCPHNs and the study paediatrician, the research team developed safeguarding protocols to ensure the safety of parents and babies involved in the study (see Appendix 4).

Health visitor involvement

In the UK, HVs/SCPHNs provide universal primary care for parents with young babies and are the obvious choice for delivering the intended service within the NHS. With support from the LPT managers collaborating on the study, HVs/SCPHNs from five health-visiting teams within Leicester, Leicestershire and Rutland attended briefing meetings about the study. A total of 55 HVs/SCPHNs gave written agreement to collaborate in stage 1 by referring eligible parents to the research team. All HVs/SCPHNs were given an information pack containing details about the study and how to approach and refer eligible parents.

All HV/SCPHN teams were revisited when the draft package was developed in order to obtain their overall views regarding its suitability.

Parent recruitment

The HVs/SCPHNs approached parents whom they knew to have previously had a healthy baby whose excessive crying in the first 6 months of life had caused concern for either parent. They provided brief details about the study in order to seek expressions of interest for researchers to contact them. To supplement HV/SCPHN contacts with parents, information about the study was also circulated via the NCT network, through researcher attendance at local parent and baby groups and through flyers that were distributed to local children’s centres, HV/SCPHN bases and local parenting/baby groups. An example recruitment flyer can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018).

On receipt of an expression of interest, the researchers contacted each parent by telephone to fully explain the study and assess eligibility. Eligible parents were sent a formal letter of invitation to participate from the principal investigator (PI) and an information sheet and consent form if their HV/SCPHN had not already provided them. Parents were invited to attend a pre-arranged focus group discussion, bringing their babies and/or older children with them if need be, and were invited to provide feedback on the draft.

Parent involvement

Participants were invited to attend pre-arranged focus groups over a 5-month period (April to August 2015), held at venues and locations convenient for parents. Seven arranged groups were cancelled or rearranged owing to a lack of parent availability. Focus groups continued when only one parent attended, provided the parent was happy for this to happen, and the structure and format remained the same. In effect, these sessions became interviews. Parental written consent was obtained prior to the focus group/interview sessions. In total, 10 focus groups/interview sessions were held.

Two researchers who were experienced in focus groups and interview methods facilitated each session and two others made observational notes or provided informal childcare. When only one parent attended, two researchers shared the duties. All sessions were audio-recorded for later verbatim transcription. Each session lasted approximately 2.5–3 hours. Parents were asked to revisit the example packages following the focus groups and were recontacted when possible by the researchers to establish if they had done so and whether or not this more extensive exposure had changed their initial opinions and ratings. At this point, parents were invited to further contribute to the development of the package by consenting to the inclusion of their stories and experiences in one of three ways:

1. permitting the use of anonymous direct quotations from the audio recording of the sessions

2. providing more detailed information about their experiences in a written format

3. appearing in video recordings talking about their experiences.

Parent qualitative discussions

All sessions were digitally recorded and transcribed verbatim. Data analysis and its interpretation were informed by hermeneutic phenomenology38 and Ricoeur’s theory of interpretation. 39,40 Analysis was undertaken at three levels; an initial reading of transcripts with the identification of themes was progressed to an explanation and naive understanding of meaning facilitated by the clustering of related themes. This then progressed back and forth between explanation and understanding to assist deeper interpretation and understanding, reminiscent of Gadamer’s hermeneutic circle41 or hermeneutic arc. 40 Analysis was supported by the use of NVivo 10 qualitative data analysis software (QSR International, Warrington, UK). All transcripts were read and reread several times and a line-by-line analysis of content was undertaken and grouped under themed headings that emerged from the data. To enhance credibility, these themed headings were shared and agreed with the researchers present at the focus groups. Initially, a large number of themes were identified; these were refined by grouping similar themes together under new overarching themes. This process was repeated until the following conceptual themes emerged: (1) disrupted experiences of parenthood, (2) feeling different and social isolation, (3) reluctance to seek support and (4) validation of experience and seeking help (see Appendix 5). Participant codes were assigned and potential identifiers removed in order to protect the anonymity and confidentiality of participants. One parent accompanied his partner and did not contribute to discussions, so only his partner’s data were included in the qualitative analysis.

Parent-completed questionnaires

Simple univariate descriptive analyses were conducted on data from the parent-completed questionnaires and a content-analysis approach was used to analyse the free-text comments. Some data were specific to the example package materials and these were summarised as a whole in order to maintain confidentiality of the providers of those packages.

Baby characteristics

The 20 study participants had 19 excessively crying infants; one parent had experienced excessive crying with both her first and second child and two couples participated in the study (their baby’s data have been included only once). Table 3 presents the babies’ characteristics.

Noticeably, only 10 of the babies were firstborn. Eight of the 18 families had other children who had not cried excessively. The babies were equally likely to be boys or girls, and they were almost equally divided between breastfeeding, formula feeding and mixed feeding when the crying started. None had a fever and only two seemed unwell. Although 11 of the babies (58%) were reported by their parents as having had problems with feeding, all the babies had been checked by clinicians for weight gain and 17 (89%) for feeding problems.

The median age at which parents reported that the babies started excessively crying was 1 week (range from birth to 9 weeks). However, there was a substantial variation in the age at which the crying stopped, ranging from 8.5 to 104 weeks (median 19 weeks). In line with this, the excessive crying duration ranged from 4 to 100 weeks (i.e. 1 month to just under 2 years), with a median length of crying of 18 weeks (i.e. 4 months).

Sources of information and support

Participants completed questionnaires about the sources of information that they used and would have found useful when their baby was excessively crying and which formats they felt were the best for presenting information about crying babies to parents (Table 4).

Noticeably, 10 parents (53%) had attended extra visits with a HV/SCPHN, doctor or other health professional about their baby’s crying and 14 (78%) reported that they would have found an extra visit with their HV/SCPHN helpful when their baby was excessively crying. Furthermore, 16 participants (89%) felt that a visit with a HV/SCPHN, GP or other health professional would be an effective format for presenting information about crying babies to parents, stating that this would give reassurance, advice and explanation for the infants’ crying.

Half of parents also had a telephone conversation about their baby’s crying with a HV/SCPHN, doctor or other health service professional. Eleven participants (61%) reported that they would have liked extra telephone calls from a HV/SCPHN when their baby was excessively crying and 13 (76%) reported that a telephone call with a HV/SCPHN, doctor or other health professional would be an effective way to present information to parents. The high usage of visits and telephone support from HVs/SCPHNs, doctors or other health professionals, particularly given that such a high proportion of parents rated them as effective, supports the qualitative finding that participants felt a need for more personal support rather than simply having more information available.

All parents stated that they would have found a group helpful when their baby was excessively crying, with 10 (59%) stating that online discussion boards and 11 (65%) stating that Facebook sessions would have been helpful sources of information; this emphasises the importance of contact with other parents.

Websites were used by 16 respondents (80%), although none reported using a crying-specific website. All 18 said that they would have found a website helpful and 16 (89%) said that websites were an effective format for presenting information. These parents felt that websites were effective sources of information owing to their accessibility in various situations and availability 24 hours a day.

Leaflets were identified by 13 parents (72%) as a resource they would have found helpful and as an effective format for presenting information, although only nine (45%) had used leaflets when their baby was excessively crying. Participants felt that leaflets made good reference material that could be perused in their own time; however, they expressed concern about the number of leaflets already given to new parents and felt that it would be easy to lose a leaflet or not have it available when needed.

Support sessions

All participants stated that they would have wanted support sessions with a professional if they had been offered to them at the time when their baby was excessively crying. Only one parent wanted one-to-one sessions only, with most parents (55%) wanting group sessions or a mix of both. Most parents would have preferred between four and five sessions, with a range from two to six sessions.

Parents’ feedback on four example packages

Parents completed rating scales on all four of the example packages, giving detailed, package-specific information on which aspects they did and did not like. All parents reported that it was important that materials such as those shown in the focus groups were included as part of routine NHS care.

In order to protect the identities of the packages used in the focus groups (and because the aim was to identify which features parents judged to be needed in future packages, rather than comparing existing packages), overall information is reported about the features of the packages that parents did and did not like, as summarised in Figure 2. This shows that parents positively rated all of the researcher-identified aspects of the packages, such as the practical suggestions for parents, the reliability of the information for parents, reassurance for parents and that the information was aimed at both parents and easy to access when needed.

FIGURE 2.

Aspects of the four sample packages that parents liked (n = 20). 1, Practical suggestions; 2, reassurance that I wasn’t doing anything wrong/it wasn’t my fault; 3, that I could trust what they said; 4, other parents’ experiences and ideas; 5, expert opinion and advice; 6, that the information is aimed at both parents; 7, videos; 8, workbooks; 9, interactive materials – responsive to your interests and concerns; 10, that the materials are easy to access when you need them.

Table 5 provides the reasons parents gave for their preference for certain materials. These largely echo the researcher-identified features of the packages and also reflect the wider discussion that took place in the focus groups. However, some of the participants’ comments went on to clarify what in particular they had liked, providing useful information for the development of the intervention package. For example, participants’ comments made it clear that they liked information that was ‘clearly explained’, ‘relevant’ and in ‘bite-size chunks’. The information and websites needed to be able to be ‘read and understood within a few minutes’, ‘clear’, ‘easy to navigate and find [the] information you are looking for’. Parents valued the reassurance, saying that the packages ‘seemed to “normalise” the crying more and helps you to feel better in yourself’. Participants were positive about other parents’ stories, with one explaining that it ‘gave a realistic situation which I could identify with’. Information aimed at the couple together was also valued; as one parent explained, ‘if your relationship is strengthened, workload shared and baby-related issues discussed and problem-solving done together then I think crying would be easier to deal with’.

Twelve of the 20 parents were successfully recontacted after the focus groups. Eight had revisited one or more of the example websites. None of the parents wished to amend their responses given within the focus groups.

Disrupted experience of parenthood

Parent participants indicated that their expectations of parenthood were positive and that they had little prior awareness that some babies could cry excessively for prolonged periods:

I think the, the expectation that you have after you have been carrying your unborn child and then you have your baby and then this whole feeling of this is going to be wonderful, and you can’t wait to hold your baby and nurture and look after and everything else – and the screaming just doesn’t stop, it just seemed to go on in a bit of an endless cycle and you think to yourself ‘it’s going to stop, it will just be the first week’ and it, for me it didn’t, it didn’t for a long, long, time.

P1P19

Consequently, they were shocked by the experience of having an excessively crying baby and they gave accounts of the many strategies they adopted to comfort their babies. However, their inability to soothe the babies made parents feel that they were somehow responsible for the crying. This, subsequently, led to feelings of inadequacy, frustration and anxiety, which seriously eroded their confidence in their ability to parent these babies:

I would say it made you feel like you are no good at it, like you are helpless really, because you try everything . . . But when she carried on crying you try and feed them, you change them, there is nothing wrong with them you just feel like useless.

P1P7

The crying often resulted in parents feeling exhausted and sleep deprived. This tiredness reduced their ability to cope, and even daily tasks, such as eating and attending to basic personal needs, were compromised for some:

I wasn’t looking after myself, I wanted to but I just couldn’t because, everything was so consumed with the crying that I just didn’t, I just almost neglected myself.

P1P13

Many mothers perceived that their partners did not fully understand their experience of being at home all day with a baby who cried incessantly. They often felt that their experiences were invalidated and dismissed as an exaggeration, creating tension between couples:

I don’t think he really understood what it was like to be at home all day on your own with a crying baby. And whilst he never came home and said ‘well it’s a mess, where is my dinner?’ you still have to do those things. He’d come in and be, ‘oh I am so tired, I have been at work’, and expect to have a chill out and she would be there screaming the place down. I started to resent him . . . a complete breakdown in communication there.

P1P16

However, there were several examples of a positive shared parental approach:

He would do all the night feeds. I went to bed at 10 [p.m.] and then he would do the night feeds and then I would get up at 6 [a.m.]. With the crying to be fair he never lost his temper once, he didn’t shout. In the middle of the night he would wake up and say ‘oh give her to me’. And he slept downstairs for 3 months with her so that I got sleep.

P1P5

Feeling different and social isolation

Parents perceived that both they and their baby were somehow ‘different’ to families with ‘normal babies’:

The year I had my baby there were five of us that all went to school that all had our babies within a few months. All of their babies were happy, they never particularly cried. So we were very much the odd ones out.

P1P16

Negative comparisons between their situation and that of others increased their sense of isolation and loneliness. Parents recognised that meeting friends or attending groups could be beneficial, but physical exhaustion and the all-consuming nature of trying to soothe their babies made getting out of the house both physically and mentally challenging:

I went to them baby massage classes but I only went for one . . . because he was crying the whole time we were there. I couldn’t do a lot with him because I just thought it’s just gonna be embarrassing. Especially cos you’d have all the mums there and they’d be like ‘oh my baby is like so’, ‘he’s brilliant, sleeps 8 hours, happy to just sit there’, but that don’t make you feel better.

P1P17

Reluctance to seek support

These feelings, together with a wish not to impose on others, appeared to inform parents’ initial reluctance to seek professional support, even from partners or close family members in some cases:

It’s sort of like admitting defeat isn’t it . . . you feel like you have been defeated by this little child and it’s actually admitting that to your family. Even my other half I never told him how bad I actually felt.

P1P5

More worryingly still, reluctance to seek help appeared to be fuelled by fears of being judged by others, particularly health-care professionals, who some parents feared would take their baby away:

But you do think that don’t you? ‘Oh my god they [health-care professionals] are going to come round and take her off me.’

P1P1

A reluctance to seek support from others led parents to look for reasons for the excessive crying and self-help strategies from sources such as the internet. However, they reported difficulty with finding information specific to excessive infant crying and how to cope with it, and several questioned the quality of the advice on offer:

Yeah, [I used the internet] a lot, when I was trying to look on Google [Google Inc., Mountain View, CA, USA], some stuff came up but it wasn’t stuff that gave you proper advice or anything on what to do.

P1P10

Parents indicated that they found online parent forums valuable and were reassured to find that they were not the only parents dealing with an excessively crying baby:

[I was] looking for information. I was just looking for what other people had been through and what they’d said; the whole act of finding and reading what other parents have been through helped me – I felt like I wasn’t on my own.

P1P18

As well as providing reassurance, online forums also provided the opportunity to interact with other parents who were sharing the same experience and, as a consequence, participants felt less isolated.

Validation of experience and seeking help

Parents described trying to find a way of relieving their baby’s crying, often going through a mental checklist of things that may be causing the baby distress, such as their nappy, feeding, temperature, etc. When they had exhausted these options, they became concerned that their baby was unwell and sought advice and reassurance from health-care professionals. As no physical cause was found and the crying continued, many parents spoke of repeated appointments as they were convinced, despite reassurance, that there was something seriously wrong.

It [crying] went on and on for 2 weeks straight it was, he just cried so much. I remember going to the doctors and then the midwife was coming round and I asked, ‘what’s he crying for?’. And I just kept looking at him and thought ‘what’s wrong with him?’, ‘why is he crying?’.

P1P12

However, despite frequent contact with health professionals, some parents felt that their concerns were not taken seriously or that professionals thought that they were exaggerating. This increased their frustration at not being heard and added to their distress:

You are banging your head against a brick wall, you are knocking on everyone’s door and you are not getting anywhere. So not only did I feel undervalued from a mother’s point of view [and] feel that what I was doing was wrong, but as an individual, as an adult I felt like nobody was listening to me, nobody cared, nobody is taking me seriously, it was a nightmare.

P1P11

Other parents found health professionals to be sympathetic and helpful; however, they questioned their level of experience in relation to excessively crying babies and their knowledge and skills in the area:

I found that the health visitor was helpful, she was quite sympathetic. She didn’t have the answers that I felt that I needed, and she didn’t have probably the experience of babies that cry. But it just made it just nicer to talk to somebody who was sympathetic to talk to.

P1P8

Parents valued acknowledgement of their concerns. Moreover, when there was continuity of care, and they were able to build up a relationship with health-care professionals, parents felt more able to share their concerns:

My health visitor was really supportive; even now she’s really supportive. She was really nice, really good, even like now she is really good, and she always like rings me to see how I am. She has given me her number and said if you ever feel down, if you ever want to talk to anyone ring me and, I go to the groups and she is there sometimes and she has a chat and she is really helpful.

P1P9

The importance of continuity and the quality of the relationship with health professionals in supporting parents was evident:

It’s a lot of work, because we have had so many health visitors it’s like starting from scratch all over again. He has three already, he’s only 15 months old and not one of them knows him. So when they ring I just say ‘yes I am fine’. I just don’t bother now.

P1P5

Although parents used a variety of means to access information about crying babies, a home visit from a HV was particularly valued. Parents felt that these visits allowed HVs to see first-hand how the baby was crying, and in doing so supported validation of the parent experience:

I used to think she [HV] should come to my house and see what I am going through. She never came to my house, she’d ring me and she was just trying to tell me things to do over the phone.

P1P7

Implications of these findings

In the opinion of parents who have experienced an excessively crying baby, the supports available for parents in this position can be improved in several ways:

• Parent education prenatally or in the first few postnatal weeks should include information about excessive infant crying, including signposting of parents towards resources and sources of help.

• Training resources in how to support parents should be developed for health-care professionals involved with the care of parents before and after their baby’s birth.

• Care models that provide continuity of care and allow parents to develop relationships that support disclosure of their concerns and anxieties should be promoted.

• There is a need to promote awareness of excessive infant crying more widely to the general public using a number of media, including television, radio and articles in popular magazines.

Parents who have had an excessively crying baby have reported that it is a bewildering and anxiety-provoking experience that undermined their confidence in their skills and aptitude to be proficient parents and shattered their expectations of parenthood. Participants felt that they might have been able to cope better if they had more information prior to the birth of their baby or even in the early days of parenthood. Understanding the parent experience of excessive crying and the perceived stigma associated with this enables the identification of how to better support parents. For the parents of excessively crying infants, this would need to include enhancing the education and training of the health-care professionals who support them. It is important to bear in mind that few fathers and parents from ethnic minority groups were involved in this study, so the findings need to be generalised with care. Nevertheless, the findings highlight an unmet need in the NHS and identify the sorts of support services that parents would like to see developed.

Parent questionnaires

A short participant demographic questionnaire was developed and was completed with all participants at the time of obtaining consent. It can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018).

Participants with an excessively crying baby completed baseline questionnaires to rate their baby’s crying problem severity, feeding and health and well-being, using items from previous studies. 44 The questionnaires can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018). They also completed four validated rating scales to measure parental well-being: the EuroQol-5 Dimensions (EQ-5D),45 the Edinburgh Postnatal Depression Scale (EPDS),46 the Generalised Anxiety Disorder-7 (GAD-7) anxiety scale47 and the Maternal Confidence Questionnaire. 48,49

These measures were repeated at the outcome stage to indicate potential improvements and were supplemented with measures of length of breast-milk feeding and crying knowledge. Parents provided ratings of each package component (see Appendix 7) and their suitability for use in the NHS. They were also asked about their willingness to participate in a RCT to evaluate the package components.

Participant take-up of the study support package and the extent of use of each component was recorded. To permit future cost-effectiveness analyses, measures of costs of each component, together with crying-related NHS costs, were also developed; these are discussed in detail in Chapter 13.

Health visitor questionnaire

A HV/SCPHN questionnaire was developed in order to obtain feedback about their involvement in stage 2, their opinions as to how helpful the materials were for parents and the extent to which they were suitable for inclusion in the NHS and their views on the provision of training in supporting parents with excessively crying babies. This questionnaire can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018).

Cognitive–behavioural therapy practitioner questionnaire

A ‘session form’ to enable the CBT practitioners to record information about the CBT sessions was developed, detailing type, length and location of sessions, child attendance at the sessions and homework completion, and included free-text comments to capture information relating to the extent to which the sessions went as planned and the success of the sessions. This form can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018).

Health visitor involvement

The HVs/SCPHNs from the health visiting teams involved in stage 1 attended briefing meetings about stage 2 of the study. Six further health visiting teams were approached to collaborate in the study after initial participant referrals to the study team were slower than anticipated. All HVs/SCPHNs were given an information pack containing details about stage 2 of the study and how to refer eligible parents.

A total of 124 HVs/SCPHNs from 12 HV/SCPHN teams within Leicester, Leicestershire and Rutland gave written agreement to collaborate with the study team by identifying eligible parents.

Following the close of recruitment and follow-up of parents, HVs/SCPHNs were recontacted and asked to complete the HV/SCPHN questionnaire. Over a 3-week period, a total of 124 questionnaires were returned in one of the following ways:

• hand-delivered to HVs/SCPHNs to complete and immediately return to the researcher

• completed over the telephone with HVs/SCPHNs because of the pressures on their time

• left at HV/SCPHN bases with a pre-paid envelope, for HVs/SCPHNs to complete and return in the post to the study team.

Of the 124 questionnaires delivered to HVs/SCPHNs, 96 (77%) were returned completed.

Parent recruitment and involvement

The Consolidated Standards of Reporting Trials (CONSORT) flow diagram in Appendix 8 shows the flow of participants’ involvement throughout stage 2 of the study. A total of 503 parents were considered for the study, of which 259 (51.5%) declined to participate or were considered to be unsuitable or ineligible to participate by their HVs/SCPHNs. In total, 47 parents were referred to the referred crying group and 197 were referred to the new birth visit group.

The HVs/SCPHNs referred both groups of parents to the study team. As in stage 1, HVs/SCPHNs provided parents with brief written details about the study in order to seek parental expressions of interest to enable researchers to contact them. Information about the study was also circulated via the NCT network and flyers were distributed to local children’s centres, HV/SCPHN bases, GP surgeries and venues where parents of young children might attend (e.g. libraries). Parents were thus able to self-refer to the referred crying group.

On receipt of an expression of interest, the researchers contacted each parent by telephone to fully explain the study, assess eligibility and formally invite parents to participate. In instances when the study team were unable to contact the parent using the telephone number given, attempts were made to contact those parents by e-mail, if an e-mail address was available, and telephone conversations were then had with those parents. Appendix 8 shows the number of parents for both groups who were no longer eligible to participate at this point, declined to participate or could not be contacted.

Parent questionnaires

Summary scores for questionnaires were treated as continuous variables. Confidence intervals (CIs) for the summary scores were calculated from linear regression models, which adjusted for the baseline score of the respective questionnaire. CIs calculated were for the follow-up score and the change from baseline, both overall and by group. In addition, differences in the outcomes between the referred crying group and the new birth visit group were investigated, with the results expressed as 95% CIs.

This was a feasibility study with two intervention groups, defined by two different methods of recruitment, and there was no control group. As this was a feasibility study, it was not powered to detect differences between the two groups. Therefore, the findings must be interpreted with caution and further investigation with a full trial would be necessary to evaluate the clinical effectiveness of the intervention. Although some statistical findings are reported when informative, for the most part the data are considered in relation to their implications for further, large-scale studies in the future.

Parent evaluations

Simple univariate and bivariate descriptive analyses were conducted and free-text comments were summarised using a simple thematic or content-analysis-style approach.

Health visitor and specialist community public health nurse feedback

Descriptive statistics (i.e. counts and percentages) were generated using IBM SPSS Statistics version 22 (IBM Corporation, Armonk, NY, USA) and free-text comments were summarised, highlighting key themes in HV/SCPHN responses.

Cognitive–behavioural therapy practitioner feedback

Simple univariate descriptive analyses were conducted and free-text comments were summarised; NVivo 10 was used to review and identify key themes in the data.

Demographic data

Basic demographic information for the study participants is presented in Table 8. Noticeably, there were few, if any, demographic differences between the groups and cases with and without excessively crying infants. The majority of participants were mothers (80–100% across the two groups) and were married or cohabiting (81.5–95%). They were predominantly white, but small numbers of Asian, black, mixed-ethnicity and other-ethnicity parents took part. Around 40% had university degrees and a further 10% had post-Advanced-level vocational qualifications. Many (50–76%) were on maternity or paternity leave when their baby cried excessively.

Pregnancy complications and general health

Parents gave information about any complications during their pregnancy and most recent childbirth and their physical and mental health in the year prior to taking part in the study. These are shown in Table 9 and suggest that, when comparing those in the new birth visit group who have an excessively crying baby with those who do not, those in the former group were more likely to report complications during their most recent pregnancy (mean difference 1.50, 95% CI 0.82 to 2.73), labour and childbirth (mean difference 1.51, 95% CI 1.01 to 2.25). These data require careful interpretation but indicate a need to include measures of parental health, including medical data if possible, in a future study.

Parental well-being and mental health

Table 10 presents the baseline psychometric measures of parental well-being and mental health for parents with excessively crying babies. As there were no significant differences between the referred crying group and the new birth visit group, the overall findings are reported. Changes in these measures at outcome assessment, which are of particular interest, are examined in Chapter 10, Parental well-being and mental health. In addition, the aim was to evaluate the sensitivity and suitability of the measures for use with these parents, both in this study and for a possible future large-scale trial.

The EQ-5D45 is widely used to provide quality-of-life measures, particularly by health economists, and was included partly for that reason (see Chapter 13 for this study’s health economic data). Total scores on the EQ-5D vary from 0 to 1, such that a score near to 1 indicates an excellent quality of life. The scale is designed for use across a variety of physical and mental health areas, including for people with severe disabilities, so that low scores were not expected here. In practice, as Table 10 shows, the mean parental score of 0.86 [standard deviation (SD) 0.20] indicates a good general quality of life at the baseline stage.

The EPDS scores are screening measures that require careful interpretation. There is some variation in the cut-off score used to identify possible depression symptoms and the need for further assessment. Although a score of ≥ 13 is often used for clinical purposes, an influential review50 recommended a score of ≥ 10 as a sensitive indicator of postpartum depression. Because it is important to include all possible cases at this stage, we have adopted this cut-off score of ≥ 10 here. As Table 10 shows, the average parental score at baseline exceeded this cut-off score (mean 10.2, SD 4.9). Using a simple head count, 30 of 57 parents of excessively crying babies were depressed at baseline in accordance with this criterion.

Scores on the GAD-7 anxiety scale can be interpreted to indicate level of anxiety, with total scores of 5, 10 and 15 indicating mild, moderate and severe anxiety, respectively. 47 As Table 10 shows, the study mean GAD-7 score of 6.3 (SD 4.8) indicates that parents in this study were, on average, at least mildly anxious. More specifically, 16 parents were mildly anxious, 11 were moderately anxious and four were severely anxious at baseline.

The Maternal Confidence Questionnaire scores are presented as a total score (which can range from 14 to 70) and a mean score (which can range from 1 to 5). 49 A higher score on the Maternal Confidence Questionnaire indicates higher perceived confidence; a mean score of 2.0 suggests that parents were relatively low in confidence. However, without a comparison group (without excessive crying) it is unclear whether or not this is typical for all parents with babies of this age.

Sleep adequacy

Table 11 provides baseline figures and ratings for sleep adequacy among parents of excessively crying babies. These are chiefly of interest so far as they change at outcome assessment, and the lack of a comparison group without excessively crying babies hampers this interpretation. That said, it is noteworthy from Figure 4 that about half of the parents in both study groups judged their sleep quality to be fairly or very bad at this stage. The average of 5.9 hours of sleep per 24 hours is less than the recommended daily adult sleep range of 7–9 hours for 26- to 64-year-old adults published by the American National Sleep Foundation. 51 The implication is that future studies should include measures of parental sleep adequacy.

FIGURE 4.

Overall sleep quality in previous 2 weeks, at baseline by group. (a) Referred crying group (n = 30); and (b) new birth visit group (n = 27).

Social support

Parents’ reports of the support they received from partners and friends/family are shown in Figures 5–7. Here, too, inclusion of a comparison group with typically crying babies would enhance interpretation. However, notably, the vast majority of parents (about 70%) judged these supports to be enough and few reported that they often needed support but could not get it. Even so, around one-quarter of the parents considered that partner and family/friend support was inadequate. Although this is a concern for a minority of parents, it will be worth including measures of social support in future studies, particularly if they are designed to improve parental well-being. Seven parents (12.2%) stated that they did not have enough/any support from either their partner or their family/friends. These parents may be a particularly vulnerable group in coping with an excessively crying infant.

FIGURE 5.

Support from partner, at baseline by group. (a) Referred crying group (n = 29); and (b) new birth visit group (n = 27).

FIGURE 6.

Support from family and friends, at baseline by group. (a) Referred crying group (n = 30); and (b) new birth visit group (n = 27).

FIGURE 7.

Parents needing support but unable to get it, at baseline by group. (a) Referred crying group (n = 30); and (b) new birth visit group (n = 27).

Babies’ sex and birth order

As shown in Table 12, baseline information was available for 52 babies (five couples participated in the study). There were similar proportions of male and female babies (47% were male and 53% were female). Noticeably, fewer than half of the babies were firstborns, with 40% being secondborn or later-born infants.

Crying patterns and impact on parents

In keeping with existing evidence, the infant’s excessive crying typically began at 2–3 weeks of age (Table 13). The mean age of new birth visit group babies on entering the study at baseline (6.6 weeks) was significantly lower than that of referred crying group babies (12.8 weeks), indicating that the new birth visit group recruitment method identified excessive crying cases at a younger age (mean difference 6.13 weeks, 95% CI 3.50 to 8.77 weeks). On average, across both groups, the babies were reported to cry for 6.9 hours per day, and up to 17 hours in one case. The daily crying pattern showed the typical afternoon and evening peak found at this age. Most parents (77%) judged the crying to be a moderate or large problem for them, but 17% rated it a severe problem. Similarly, most parents (73%) had been moderately or very frustrated by the crying and 15% had been extremely frustrated.

Babies’ feeding

Table 14 provides descriptive figures for the infants’ feeding at baseline.

As Table 14 shows, most babies (71%) were breast-fed when the excessive crying started, 40% were formula-fed and 11.5% were a mixture of breast- and formula-fed. On study entry, 52% were breast-fed, in keeping with other evidence that excessive crying is associated with stopping breastfeeding. Infant weight gain had been checked by a professional in all but one case and feeding had been checked in most cases (58%). Measures of breastfeeding continuation and precise medical record and diagnostic information (when available) should be included in future studies.

Remedies given for crying, contacts with professionals and babies’ health

Tables 15 and 16 describe the remedies that parents had tried for their excessively crying babies, their contacts with professionals regarding the crying and the infants’ baseline health more generally.

Consistent with previous research, a substantial number of parents had tried various remedies, including prescriptions from their doctor, changes of formula and the use of ‘colic drops’. Most (73%) had received HV/SCPHN visits at home and 50% had visited their GP. Websites other than the study website had been accessed by 79% of parents who provided data. Even allowing for parents who did not answer this question, this finding clearly illustrates the importance of internet resources for these parents.

Vomiting and feeding problems accompanied the crying in 40–50% of cases, parents judged around one-quarter of infants to be unwell and half of the infants had been seen by their GP in the last fortnight for reasons other than their crying (Table 17). These figures all involve parental subjective judgements that are hard to interpret; for instance, vomiting is known to be common in healthy babies at this age. 52 Nonetheless, so far as these measures indicate the source of parents’ concerns, it will be worthwhile to include them in future studies, particularly in studies involving the clinical effectiveness of interventions. The values in Table 17 highlight the cost of excessive infant crying for families and health services, a topic that will receive detailed attention in Chapter 13.

Parental well-being and mental health

Table 18 presents the outcome scores and changes from baseline in psychometric measures of well-being and mental health for parents with excessively crying babies. Standardised change measures and CIs are also given. As noted previously, no formal hypothesis tests were conducted because this feasibility study is not powered for such tests. However, the CIs provide an indication of the findings that are likely to be repeated in an adequately powered study.

The EQ-5D findings showed modest improvements in quality of life between the baseline and outcome assessment points. These small improvements are to be expected, given the broad nature of quality-of-life measurements and the lack of severe physical and mental health disabilities in the parents involved in this study. A key reason for including these measures was their utility for health economic analyses, which are examined in Chapter 13. After adjusting for the baseline EQ-5D score and for the change in crying, the change in the quality-of-life scores from baseline in the referred crying group was not significantly different from the change in the new birth visit group (mean difference 0.0053, 95% CI –0.40 to 0.05).

The observed EPDS summary scores indicated that postnatal depression was lower in both groups at follow-up. The summary scores for the GAD-7 anxiety questionnaire and the Maternal Confidence Questionnaire also showed that both groups had lower anxiety and greater parenting confidence, respectively, at follow-up. The mean scores at outcome assessment [EPDS mean score of 7 (SD 4.6); GAD-7 mean score of 3.3 (SD 3.3)] no longer approximated to the clinical cut-off scores. Expressed as a head count, the number of parents with an EPDS criterion score for depression (≥ 10) halved (from 30 to 15 parents) between the baseline and outcome assessments. Similarly, the number of parents with mild, moderate and severe anxiety reduced from 16, 11 and 4 parents, respectively, at baseline, to 12, 2 and 2 parents, respectively, at outcome assessment. These findings are in keeping with the notion that the impact of infant crying on parents (and vice versa) involves parental mental health and well-being in particular. The associated CIs indicate that similar changes are likely to be found in a future large-scale study with adequate statistical power.

The figures also showed modest increases in parental confidence scores. Although not as substantial as the improvements in anxiety and depression, they are encouraging in so far as the aim is to increase parents’ confidence as part of enhancing parental coping skills and interactions with infants. There is some evidence of an inverse relationship between parental confidence and depression,49 which can be assessed more thoroughly in a future large-scale trial.

In summary, these psychometric findings indicate that measures of parental mental health and well-being should have a central role in future research in this area. They also identify methods that are suitable for such a study. In addition, the findings have implications for theoretical understanding and for health service practice. On the theoretical side, the association between excessive infant crying and maternal depression and anxiety is well known, but there is little consensus about the direction of causation. For example, a recent study27 concluded that maternal anxiety during pregnancy predicted excessive infant crying. The present study was not designed to provide causal information and the absence of a control group, in particular, prevents a conclusion regarding whether the reductions in parental anxiety and depression that were observed between baseline and outcome measurements were attributable to the study’s parental support package, to reductions in infant crying that would be expected with infant development or to the passage of time. What the findings indicate is that levels of parental depression and anxiety, as measured, did reduce in substantial numbers of parents during this relatively short period of postnatal experience. The implication is that, if similar findings emerge from an adequately powered RCT, the findings will increase our knowledge of causation. One plausible theoretical viewpoint is that excessive infant crying provides a stressor that triggers the onset of anxiety and depression, and reduces coping, in vulnerable parents. To examine this theory, a future study would need to track infant crying and parental well-being over time, at least until the crying subsides, in treated (i.e. website, booklet and CBT programme) and control groups. A specified model of change, which sets out how the processes involved are expected to work and includes, for instance, measures of parental coping behaviours, parent–infant interaction and relational measures, such as attachments, could be used to provide evidence to advance scientific knowledge still further.

Equally, the findings from this part of the study have implications for health services. There is reliable evidence that CBT programmes can help to reduce postnatal depression and anxiety in some parents. For the most part, programmes of this type target parental cognitive, emotional and behavioural processes, but do not take environmental factors, such as excessive infant crying, into account. For at least a subgroup of parents, the promise of the findings here is that support programmes that include coping with infant crying will be particularly effective in enhancing parental mental health.

Sleep adequacy

Table 19 provides outcome and change measures of sleep adequacy for parents of excessively crying babies. Both groups of parents reported increased sleep, amounting to an average increase of 1 hour and 6 minutes per 24 hours at the outcome measurement. Both groups also improved their sleep quality, so that the number reporting fairly bad or bad sleep quality halved at the outcome assessment point. These figures reinforce the conclusion that measures of parental sleep amount and quality should be included in future studies.

Social support

Figures for the support that partners and friends gave to parents of excessively crying babies at the outcome assessment, together with changes since the baseline measures, are shown in Table 20. As at baseline, the majority of parents in both groups reported receiving enough support from partners (70–83%) and family and friends (72–74%). The figures indicate small but consistent reductions of around 10% in the number of parents who judged that these supports were not enough and who sometimes needed support but could not get it. However, the smaller overall numbers of parents at the outcome assessment complicate this interpretation. The findings’ main implication is to add to the proposal that social support measures need to be included in future controlled studies with large-enough numbers to allow firm conclusions.

Crying patterns and impact on parents

Table 21 shows the figures for infant crying, its impact on parents at the outcome assessment, as well as changes in these measures since baseline. Fourteen infants were reported to still be excessively crying at the outcome assessment. However, for those that had stopped crying, on average, the crying lasted for 8 weeks and 3 days. Those infants who were still excessively crying at the outcome assessment had an average age of 15 weeks and 6 days, which implies a need for longer follow-up in future studies. The total time that infants spent fussing and crying per 24 hours halved from the baseline assessment to the outcome assessment, from 6.9 to 2.6 hours.

Figure 8 demonstrates that the number of parents reporting crying to be a severe or large problem reduced from 27 parents (47%) to three parents (6.5%) at the outcome assessment. Strikingly, this was matched by a drop in the number of parents feeling extremely or very frustrated by the crying from 30 (58%) at baseline to none at the outcome assessment (Figure 9). If frustration is the trigger for infant abuse, as studies indicate,24 this substantial reduction is a particularly promising and encouraging finding. Broadly, across the groups and findings as a whole, the tendency was for parents to report a reduction in the degree of frustration they felt because of their baby’s crying, rather than a complete elimination. These findings emphasised the importance of longer follow-up. In addition, they indicate the need for a larger-scale study with control groups and sufficient numbers, so that the contribution of the support package, and each of its components, can be distinguished from the reductions that happen with time and age.

FIGURE 8.

Impact of babies’ crying on parents at baseline (n = 57) and outcome assessment (n = 52).

FIGURE 9.

Parents’ frustration at babies’ crying at baseline (n = 57) and outcome assessment (n = 52).

Babies’ feeding

Table 22 shows the figures for babies’ feeding at the outcome assessment, together with changes since baseline. It is worth noting that very little change in breastfeeding and formula-feeding rates was seen between the baseline and outcome assessments. In the 21 cases in which parents reported stopping breastfeeding, the median infant age at this point was 2 weeks. It follows that any effects of infant crying on breastfeeding appeared to happen predominantly at an early age.

Remedies given, contacts with professionals and babies’ health

Measures of infant health and well-being at the outcome assessment, as well as changes, are shown in Tables 23–25. Parents were asked to provide outcome details solely for the period between the baseline and outcome assessments, that is, to omit instances included in the baseline measures. Measures of HV/SCPHN and GP e-mail or internet contact and GP home visits have been omitted from the table because no parent reported these at baseline or outcome. Notably, the length of the period from birth to baseline assessment and from baseline to outcome assessment is different (a mean interval of 9 weeks and 4 days between birth and baseline and 5 weeks and 5 days between baseline and outcome assessment), so changes in the values given in these tables need to be interpreted with caution.

With a few exceptions, the figures indicated improved infant health, reduced contacts with professionals and reduced use of remedies at outcome assessment, compared with baseline (see Tables 23–25). The reductions in the number of HV/SCPHN home visits (from 73% to 11% of parents) and telephone contacts (from 46% to 11% of parents) because of excessive infant crying are substantial, as is the reduced number of visits to GP surgeries (from 50% to 21% of parents) for this reason (see Table 24). Potentially, these figures identify worthwhile benefits and cost savings for families and health services because of the intervention package. However, the figures are difficult to interpret because of differences in the periods of time addressed by the baseline and outcome measures, the reduced numbers providing outcome measures, the dependency on parental reports and the lack of a control group or groups needed to distinguish the effects of the support package from the effects of time and infant age. The implication is to point to the need for a future large-scale controlled trial that enables these factors to be distinguished. Because the figures in both these and the previous tables suggested that excessive infant crying and its impact on parents were continuing, albeit at reduced levels, at the outcome assessment point, an extended follow-up duration until after the crying resolves should also be included in future studies.

Parents’ knowledge of infant crying

To establish whether or not parents’ knowledge of infant crying had been increased by the support package, parents were asked about information included in the package materials, using five questions adapted from the study by Barr et al. 53 Table 26 shows the questions and parents’ answers. Some parents (10–56%) knew some answers before taking part in the study, whereas others (0–14%) reported that this was new information for them, despite its inclusion in the study support materials. The implication that some parents did not access or retain some of the package information is supported by the website access figures shown below. Parents gained some knowledge about infant crying from HVs/SCPHNs. However, 32–64% of parents acquired this knowledge from the support package materials, substantially more than those who acquired it from HVs/SCPHNs (6–14%). The implication is that it will be worthwhile to include these questions, and possibly to assess parents’ knowledge in other ways, in future studies. Whether or not the website and other package elements can be improved to deliver this information more effectively also needs to be considered.

Devices used to access the website

The Google Analytics data showed that parents mostly used mobile phones (n = 31, 57.4%) and tablet computers (n = 17, 31.5%) to access the website, with a small number (n = 6, 11.1%) using a desktop/laptop computer.

Couples and family members

Five couples requested CBT support. Six sessions were planned to be delivered as couple sessions to participants, both of whom consented to take part in the study. One parent did not attend their only booked couple session, but their partner did go on to receive one-to-one sessions. Two couples (four parents) were seen only as a couple, whereas the other two couples had a combination of both couple and one-to-one sessions. Noticeably, males only attended couple sessions; no males requested or received one-to-one sessions. In addition, family members who were not participants in the study were unexpectedly present at five further sessions that were planned as one-to-one sessions (four were participants’ partners, but in one case a participant’s mother and sister also attended).

Most sessions (93%) had at least one child in attendance, with 22% of sessions having two or more children in attendance and, as mentioned above, five sessions included other adults who were not study participants. These figures highlight the complications involved in delivering an intervention in a family home. However, in spite of this, the CBT practitioners delivering the support sessions reported that the sessions largely went as planned.

Although the first two support sessions were planned to cover pre-set topics to establish the core CBT ideas and techniques underpinning the intervention, parents were able to choose which (if any) supplementary topics they would like to be covered. For parents who already had some understanding of the core CBT ideas and techniques, the CBT practitioners were able to cover supplementary topics in the parents’ first session. Table 31 shows which supplementary topics parents received. The majority of parents chose the relaxation and diaphragmatic breathing and more than half of parents chose arranging social supports and asking for help when needed.

Parents who attended more than one support session were given activities to practise the work that had been covered by the CBT practitioner in between sessions. Of the 21 parents who went on to attend more than one session, 18 had completed their home activities, two had tried but been unable to complete their home activities and one parent had not carried out any home activities.

Parental feedback on the Surviving Crying website

The website was considered clear, helpful and relevant by all but two of the 30 participants who provided these judgements and over half rated its attractiveness highly (Table 35).

The website was rated particularly highly for (1) ease of access, (2) giving other parents’ experiences and ideas and (3) making participants feel that they could trust the information supplied, as shown in Table 36. Participants liked the simple design and layout of the website, with one appreciating that they were able to access it on their mobile phone. Almost half of the participants who rated the website highlighted the videos and the sharing of other parents’ experiences as being particularly helpful and reassuring, with some liking that the website also included practical tips and demonstrations. Only one participant reported that they were not interested in hearing about other parents’ experiences. A few participants noted that they particularly liked that the website included a section on research, as they felt that this gave the website credibility and, consequently, they felt that they could trust the information that was given. One participant reported that, as a result, they had looked at the suggested research in more detail.

Around one-third of those who had rated the website gave suggestions for its improvement. Most of those suggested a parent forum section in which parents could share advice and information, as well as offer support to each other. Two participants wanted more information, advice and practical tips from other parents on, for example, alternative soothing techniques and practices, for single parents in particular. One participant thought that the parent videos were too long when they needed immediate reassurance but that they were ‘perfect’ to look at when their baby was asleep.

Parental feedback on the printed information booklet

The printed information booklet was considered to be clear, helpful and relevant by all of the 23 participants who provided these judgements, with most rating its attractiveness highly (Table 37).

Overall, the information booklet was rated particularly highly on being aimed at both parents, the practical suggestions and ease of access, as shown in Table 38. Participants explained that the fact that the information booklet was in a printed format meant that it was easily and readily accessible and available to both parents and that it could easily be read, for example when feeding a baby, taking a bath, or at times when parents needed some instant reassurance. Some participants liked that it provided reassurance by including other parents’ experiences, in the form of quotations, stories, advice and practical tips. Only two participants suggested improvements to the information booklet: one was to produce it in an A5 format, so that it could be portable, and the other suggestion was to include more information, for example advice for older children trying to cope with a crying baby.

Parental feedback on the practitioner-delivered sessions

Information provided as part of the CBT practitioner-delivered session was considered to be clear, helpful and relevant by almost all of the 24 participants who provided judgements (Table 39).

Overall, the sessions were rated particularly highly on convenience of the session location, convenience of session times and trust in the practitioner, as shown in Table 40, and were rated ‘weak’ by only two participants in the areas of practical suggestions given, expert advice, helpfulness for both parents and convenience of session times.

Participants liked that the practitioners provided general practical information and personal advice, that they were non-judgemental, made parents feel supported and gave them time to talk. Some participants liked that the sessions took place in their own homes, where they felt relaxed, and that there was some degree of flexibility around the times of the sessions, although one participant explained that such flexibility resulted in her not giving the sessions priority like she would have done if they were fixed appointments. A couple of participants liked that the sessions were face to face and one to one; in addition, one participant found it helpful that the sessions could involve their partner too. A few participants liked knowing that they were able to contact the practitioners outside the planned sessions if they felt that they were struggling and needed their reassurance.

The breathing and relaxation exercises were highlighted by some participants as being particularly helpful; others highlighted the coping with crying tips and strategies, as well as the written information that could be read and referred to outside the sessions. Some participants felt that the CBT approach was particularly helpful for them in dealing with the crying, for example in changing their thought processes, as well as more broadly, for example in dealing with other people’s unwanted advice and opinions and with their own postnatal depression.

One participant felt that it was particularly important that the CBT practitioner was, as they described, ‘completely independent from health visitors’, but they also stated ‘I would urge the NHS to give this kind of support to all mothers’ (P2C03).

Twenty-three of the 24 participants identified possible improvements to the CBT practitioner-delivered sessions, as shown in Table 41, with a large proportion wanting to see more small-group sessions.

Other suggestions for improvements were more practical advice on helping a crying baby and more evidence-based support, longer sessions (possibly with goal-setting targets), leaflets listing soothing techniques that could be referred to when needed and the timing of the sessions (so that they happened when the crying was at its worst). In keeping with our stage 1 findings, suggestions for improvement included the need for small-group sessions, which proved impractical at this stage because too few parents were available at the same time in nearby locations. Other, possibly online, methods of contact can be considered in the future.

The findings reinforce the need for a large-scale study to evaluate the study support package more rigorously and for the package materials to be revised before and during the next evaluation.

Methods

We collected data on the costs of routine services associated with excessive crying in order to inform a future economic evaluation. We focused on what items of resource use to collect from a NHS and Personal Social Services (PSS) perspective, as this perspective is likely to be relevant for economic evaluations for decision-making bodies such as NICE. 56 Potential cost components using this perspective could include NHS and PSS contacts and medications. Based on the study by Morris et al. ,17 we created a resource questionnaire that was given to participants at the baseline and outcome assessments. This questionnaire can be accessed at www.journalslibrary.nihr.ac.uk/programmes/hta/1215004/#/ (accessed 6 December 2018). Given the results of the study by Morris et al. ,17 the questionnaire specifically itemised NHS contacts, but participants were invited to record any contacts because of a concern about their baby’s crying, which could have included social care. For example, postnatal depression (e.g. related to excessive infant crying) could be related to the increased use of social care services. 57 We also collected data on the use of remedies for infant crying, including those prescribed by a doctor, although based on the findings of Morris et al. ,17 costs associated with NHS medications were expected to be negligible.

At the baseline assessment, the questionnaire asked about contacts since birth. At the outcome assessment, the questionnaire asked about contacts since the baseline assessment.

As noted, at the baseline and outcome assessments parents were asked to record other remedies they used for their baby’s crying. As well as remedies prescribed by a doctor, these included change of formula feed, use of colic drops and herbal remedies, treatment by a chiropractor and other remedies. The costs of these remedies are likely to be borne by families; therefore, we quantified the proportion of families using these remedies but did not include them in the following preliminary cost calculations.

Other non-NHS/PSS costs that might be included if a societal perspective to cost measurement was taken are travel costs for families going to and from NHS visits and days taken off work by parents because of excessive infant crying. We did not include either of these costs in the analysis given the perspective of our analysis. With regard to travel costs for families, we note that most of the NHS contacts are either at home or by telephone, thereby negating the need for travel. We also did not collect data on days off work given the fact that many parents with babies at this age are on maternity/paternity leave, making the measurement of days off work difficult.

For the non-zero cost components identified using the resource use questionnaire, we identified potential unit costs that could be used in a future economic evaluation. These were mainly derived from Curtis and Burns58 and are reported in 2015/16 Great British pounds (GBP). For HV contacts, we based the cost per contact on the cost per hour reported by Curtis and Burns. 58 It was felt that in the case of HV/SCPHN contacts, it was unlikely that a whole contact would be exclusively devoted to discussing infant crying problems; therefore, we quantified the amount of time spent discussing these problems during a visit. To do this, HVs/SCPHNs from the LPT teams participating in the study recorded the time spent discussing infant crying problems in contacts (all types) in which these were discussed. Across 445 contacts, the mean time was 17.3 minutes (range < 1 minute to 120 minutes). As this figure related to all HV/SCPHN contacts in which infant crying problems were discussed, it was used as the mean duration of all these contacts. For all other contacts (e.g. with a GP), we assumed that the whole contact was used to discuss infant crying problems and that the duration of each contact was as stated by Curtis and Burns. 58

The additional contacts included as free text in the questionnaire were classified into six categories based on the responses received:

1. nurse home visit

2. midwife home visit

3. A&E department visit

4. outpatient visit

5. walk-in centre visit

6. health centre visit.

No details were recorded on the type and grade of nurse and the grade of midwife; these were monetised using the same unit costs for HVs, described previously in this section. No additional details were provided for A&E, outpatient, walk-in centre and health centre visits. Therefore, A&E visits were monetised using the national average unit cost for all consultant-led outpatient attendances reported by Curtis and Burns. 58 Outpatient visits were monetised using the national average unit cost for all outpatient attendances reported by Curtis and Burns. 58 Walk-in centre visits were monetised using the national average unit cost for all non-consultant-led outpatient attendances reported by Curtis and Burns. 58 Health centre visits were monetised using the unit cost of a practice nurse visit with a duration of 17.3 minutes reported by Curtis and Burns. 58

We calculated the mean NHS cost per family of excessive crying for three time periods (i.e. birth to baseline measurement, baseline measurement to outcome measurement and birth to outcome measurement) by summing the different cost components. It should be noted that there was a difference in the duration of the first and second time periods. It should also be noted that the cost estimates for the period from baseline measurement to outcome measurement may be affected by any impact of the study interventions on NHS contacts.

Results

Useable resource use data were collected for 98% of respondents at baseline assessment (56/57 respondents) and 91% of respondents (52/57 respondents) at outcome assessment. Across the whole study period, resource use data were available for 89% of respondents (51/57 respondents). The mean age of babies at the baseline assessment was 9.3 weeks (range 3 to 24 weeks) (Table 46); at the outcome assessment, the mean age was 15.0 weeks (range 9 to 30 weeks) (Table 47).

Across the whole period, there were non-zero contacts for excessive crying for the following items collected in the resource use questionnaire (Tables 46–48):

• HV home visit

• HV clinic visit

• HV contact by telephone

• GP surgery visit

• GP contact by telephone.

There were zero contacts for the following items:

• HV contact by e-mail or internet

• GP home visit

• GP contact by e-mail or internet.

The most common type of contact for excessive crying was with the HV/SCPHN. In the period from birth to baseline assessment (mean 9.3 weeks), the most common HV/SCPHN contact was a home visit, with a mean number of visits per family of 2.1 (range 0 to 8 visits) (see Table 46). In the period from baseline to outcome assessment (mean 5.7 weeks), the most common HV/SCPHN contact was by telephone, with a mean number of calls per family of 0.5 (range 0 to 10 calls) (see Table 47). Across the whole period (mean 15.0 weeks), the most common HV/SCPHN contact was a home visit (mean number of visits per family 2.4, range 0 to 8 visits) (see Table 48).

When respondents were asked to record other types of contact they had made, these were categorised into:

• nurse home visit

• midwife home visit

• A&E visit

• outpatient visit

• walk-in centre visit

• health centre visit.

The mean number of contacts of each of these types was small (i.e. ≤ 0.2 contacts per family across the whole period) (see Table 48).

The total mean NHS cost per family for excessive crying was £206 in the period from birth to baseline measurement (mean 9.3 weeks), £75 in the period from baseline measurement to outcome measurement (mean 9.3 weeks to mean 15.0 weeks) and £287 across both periods combined (see Table 49). It should be noted that there is a difference in duration between the first and second time periods. It should also be noted that the figures of £75 in the period from baseline measurement to outcome measurement and £287 across both periods combined include any impact of the study interventions on NHS contacts.

Thirty-four per cent of parents reported using a remedy prescribed by a doctor for infant crying during the period from birth to baseline measurement, compared with 31% in the period from baseline measurement to outcome measurement (see Table 50). Remedies recorded as prescribed by the doctor were:

• allergy milk

• amoxicillin

• Gaviscon^® [Reckitt Benckiser Healthcare (UK) Ltd]

• Simeticone (Infacol^®, Purna Pharmaceuticals)

• ranitidine

• hydrolysed milk formulation (Aptamil Pepti^®, Nutricia Ltd; Nutramigen^®, Mead Johnson Nutrition)

• cream for eczema

• lactulose

• nose drops

• chlorphenamine maleate (Piriton^®, GlaxoSmithKline).

Data on whether the medication was for the infant or the parent were not recorded. The majority of these items are available over the counter. Data on the dosage and duration of the remedy were not recorded, although this could be measured in a future economic evaluation.

The most commonly used remedy in the first period was colic drops (73%) (see Table 50). Other remedies that were recorded were:

• baby massage

• changing bottles

• foot manipulation

• gripe water

• Gaviscon

• lactulose

• an osteopath

• ranitidine

• Rescue Remedy^® (A Nelson & Co Ltd)

• cool water

• laxatives

• a hot water bottle

• nose drops

• teething gel

• paracetamol.

Cost burden

In this section, we use the data described in Results to calculate the potential annual burden of excessive crying problems in infants to the NHS in the UK, associated with ‘usual care provision’. We do this by multiplying (1) the mean NHS cost per excessively crying infant by (2) the number of excessively crying infants each year in the UK. The first element (1) was taken from estimates calculated in the present study. We calculated that from birth to 9.3 weeks of age, the mean NHS cost per excessively crying infant was £206. We also calculated that from birth to 15.0 weeks of age, the mean NHS cost per excessively crying infant was £287. It should be noted that the latter figure is calculated over a longer period and includes the period during which parents in the study received the intervention package. Therefore, the costs during this period may be depressed because of the impact of the intervention. We observe in our data that the costs during the second period were lower than in the first period (£206 vs. £75). This is partly attributable to the difference in the time periods (mean 9.3 weeks vs. 5.7 weeks), but the costs per week during the two periods were also different [£22 for the first period (£206/9.3) and £13 for the second period (£75/5.7)]. This difference may be attributable to a reduction in excessive crying over time, a reduction in the propensity to contact health services about excessive crying over time or the impact of the intervention. The first two of these are legitimate sources of lower costs for the cost burden calculation during the second period but the last is not, because we wish to estimate the cost burden in the absence of the intervention. Hence, we estimate the cost burden using both estimates, recognising that the first covers a shorter time period and the latter may be contaminated by the impact of the intervention.

The second element (2) was based on the total number of live births in the UK and the incidence of excessive crying in infants. In 2015, there were 777,165 live births in the UK. 59 There is considerable uncertainty surrounding the incidence of excessive crying, with estimates varying in accordance with the definition of excessive crying and the age of the baby when these are measured. We used five estimates. The first is the incidence found in the new birth visit group in the present study (21.7%; see Chapter 8). The other measures were the maxima and minima reported in previous reviews of this topic (1.5% and 5%, 11.9% and 32.1%). 60,61

Using this approach and these figures, the total annual UK NHS cost of excessively crying infants is estimated to be in the range of £2.4M to £51.4M during the period from birth to 9.3 weeks and £3.3M to £71.7M during the period from birth to 15 weeks (Table 51).

Introduction

In this section, we calculate the intervention costs associated with the Surviving Crying intervention package (i.e. website, printed booklet and CBT programme). We only include the costs of delivering the intervention package, not of developing it. The development costs comprise the costs of the research undertaken during stage 1 of the research study and are not included here. For the website and booklets, the costs reported in the following sections were based on market prices incurred in the production of these materials. The costs of the CBT programme were based on the resource use for the provision of the programme, which were recorded by the practitioner.

Website

The costs of the website include the one-off costs to set the website up, the costs of maintaining the website and the cost of registering new users to it. The one-off cost to produce the website by the developers was £12,296. The annual cost to maintain the website had two elements. First, there was an annual fee of £525.75 for the cost of the website domain and of hosting the website. Second, there were costs associated with maintaining the content of the website. This was estimated to require three person-days’ input per year at a total cost of £957 per year. Combining both elements, the annual cost to maintain the website was £1482.75. Registering new users to the website required approximately 15 minutes of administration time per family to create the new registration and communicate the log-in details and information about the website. This was estimated to incur an additional cost per new registration of £32.33.

Booklet

The second component of the intervention package was the printed booklet, which was designed to match the layout and content of the website as far as possible. The cost to produce each booklet (the cost per family) was £6.46.

The cognitive–behavioural therapy programme

The third component of the intervention package was the CBT programme. This included the following cost components:

• therapist time spent undertaking the following activities –

  • face-to-face CBT sessions

  • telephone contacts with participating families

  • text, e-mail or internet contacts with participating families

  • preparation for the CBT sessions

  • administration

  • travel to the participants’ homes

• costs to travel to the participants’ homes

• printed information booklet.

The therapist providing the CBT sessions recorded the total time spent on each task across all 21 participating families. These totals were monetised and divided by the number of families participating in the CBT programme to calculate the mean cost per family. The cost of therapist time was valued at £105.82 per hour, based on the total cost per hour of CBT therapist time reported by Curtis and Burns. 58 The mean therapist time input per family was 3.92 hours, giving a total cost of £415 per family (Table 52). The therapist also recorded the distance travelled to each home (in miles) to enable the calculation of travel costs. These were valued at a cost of £0.56 per mile. 62 The mean distance travelled per family was 24.38 miles and, therefore, the mean travel cost per family was £14. All participating families received a printed booklet; the cost to produce each booklet (the cost per family) was £7.83. Note that this is different to the cost of the booklet based on the Surviving Crying website, which was £6.46. Combining each cost component, the mean cost per family participating in the CBT programme was £437.

Summary

The aim of this analysis was to undertake preliminary research to inform a future economic evaluation of the Surviving Crying intervention package from a NHS perspective. We focused on the measurement of costs, explored crying-related NHS costs and the intervention costs associated with the Surviving Crying intervention package. We asked study participants to retrospectively record data on NHS and other contacts related to their baby’s excessive crying and achieved high response rates. We collected data on the frequency of different types of NHS contact, which can be used to identify which contacts ought to be included in future resource use questionnaires and which are likely to be less important. We calculated the cost burden associated with excessive crying and found that this varied considerably, mainly in accordance with variations in the incidence of excessive crying. We calculated the intervention costs associated with the Surviving Crying intervention package (i.e. website, printed booklet and CBT programme). The website incurred a one-off cost to be produced of £12,296 and an annual cost to maintain of £1482.75. In addition, it incurred an additional cost per new registration of £32.33. The cost per family for each printed booklet was £6.46. The mean cost per family participating in the CBT programme was £437.

Lessons learned

The following is a list of lessons learned from the economic analysis, which may be useful in planning future economic evaluations:

• Based on the data collected in this study, especially the high response rates, it is likely to be feasible to undertake an economic evaluation of an intervention to manage infant crying problems alongside a RCT.

• Retrospective resource use questionnaires to collect cost data are feasible in this population group, at least over the period considered in the present study (birth to 15 weeks).

• When collecting resource use data, it is important to specify whether the contacts relate to all contacts or just those relating to crying problems. This is because, especially in the case of HV/SCPHN contacts, it is unlikely that a whole contact will be exclusively devoted to discussing crying problems. It is important to quantify the amount of time spent discussing these problems during a visit.

• Resource use questionnaires should include the following items: (1) HV/SCPHN home visits, (2) HV/SCPHN clinic visits, (3) HV/SCPHN contacts by telephone, (4) GP surgery visits and (5) GP contacts by telephone. The following items had no contacts in the present study and so may not be required: (1) HV/SCPHN contacts by e-mail or internet, (2) GP home visits and (3) GP contacts by e-mail or internet. No PSS contacts were recorded. In addition, it may be worth delineating specific contacts for midwife visits, other nurse visits and contacts with secondary care (e.g. A&E, outpatient and walk-in centre contacts) and health centre visits, although the total number of contacts of these types is likely to be small.

• Resource use questionnaires could also include medications prescribed by the GP and whether these are for the infant or for the parent. If these items are to be costed, then data ought to be collected on the name of the drug and the dosage and duration of treatment.

• If non-NHS/PSS costs are to be included in a cost analysis using a societal perspective, then these could include remedies for excessive crying paid for by families, including colic drops and herbal remedies.

• Future economic evaluations could comprise both cost-effectiveness analyses and cost–utility analyses. The former can include both infant and parent outcomes; the latter is likely to be restricted to parent QALYs only, given the difficulties in measuring QALYs in infants.

• Further research to evaluate the cost of excessive crying would be useful, and such analyses could be based on resource use in a control group of a RCT, provided treatment in this group reflected usual care. The total cost burden depends on the incidence of excessive crying, and there are a wide range of available estimates. A reliable measure of this incidence is needed to produce an accurate cost burden estimate.