- Email: [email protected]
Qualitative questionnaire on the psychosocial wellbeing of mothers of children with BEEC
Accepted Manuscript Qualitative questionnaire on the psychosocial wellbeing of mothers of children with BEEC Massimo Di Grazia, Sandra Pellizzoni, Luca Giacomo Tonegatti, Waifro Rigamonti PII:
S1477-5131(16)30247-9
DOI:
10.1016/j.jpurol.2016.07.015
Reference:
JPUROL 2318
To appear in:
Journal of Pediatric Urology
Received Date: 7 March 2016 Accepted Date: 1 July 2016
Please cite this article as: Di Grazia M, Pellizzoni S, Tonegatti LG, Rigamonti W, Qualitative questionnaire on the psychosocial wellbeing of mothers of children with BEEC, Journal of Pediatric Urology (2016), doi: 10.1016/j.jpurol.2016.07.015. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Qualitative questionnaire on the psychosocial wellbeing of mothers of children with BEEC Massimo Di Grazia a, Sandra Pellizzoni b,*, Luca Giacomo Tonegatti b, Waifro Rigamonti a,c a
Department of Life Science, University of Trieste, Trieste, Italy c
Institute for Maternal and Child Health – IRCCS “Burlo Garofolo”, Trieste, Italy
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b
Department of Medical, Surgical and Health Science, University of Trieste, Trieste, Italy
* Corresponding author. Department of Life Science, Via Weiss 21, Building W, University of Trieste, 34128 Trieste, Italy. Tel.: +39 339 342 3876; fax: +39 040 675 8539
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E-mail address: [email protected] (S. Pellizzoni).
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Summary Introduction
The bladder exstrophy–epispadias complex (BEEC) represents a spectrum of malformations that affect the anatomical and functional structure of the urogenital system. The parents of patients
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affected by this condition are subject to particularly stressful situations, such as worrying about their child’s health, long hospital stays, concerns about the health and constant need for personal care for their children, that can profoundly compromise the quality of family life.
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Objective
The objective of this explorative qualitative study is to evaluate the social situation and the
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psychological strategies implemented by the mothers of children between 6 and 10 years of age who are affected by BEEC. Study design
Fourteen mothers of children aged 6–10 years and affected by BEEC (9 boys and 5 girls) were interviewed. Data on the mothers’ experiences were collected through semi-structured interviews (Table) Results
1
ACCEPTED MANUSCRIPT The qualitative analysis of the interviews showed that participants described experiences that were characterised by emotions such as fear and anger. Each mother had implemented a different and, sometimes, dysfunctional strategy in order to cope with the complex situation of the son/daughter. The aspects that most clearly emerged from mothers’ descriptions were (1) the traumatic situation
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at the birth of the baby, (2) the sense of embarrassment concerning the pathological condition as the child was growing and the consequent sense of isolation of the mother, and (3) the fluctuation of feelings towards the multidisciplinary staff, which was sometimes seen as an important source of
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help and some other times as too destabilising and not helpful at all. Discussion
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The study provided some insight into the psychological and social conditions experienced by mothers of children with BEEC, which could serve as a basis for developing multidisciplinary teams with greater awareness about families living with this condition and better timing in addressing their needs.
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Conclusions
Mothers of children with BEEC show emotional and social difficulties. This is a crucial aspect to consider when planning a multidisciplinary approach to the treatment/therapy, especially
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considering that children examined in this study are approaching adolescence.
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Table Semi-structured interview topic. Number of questions
1 Impact of the diagnosis
4
2 Relationship with the medical team
2
3 Relationship with the partner
4
4 Social life: relationship with colleagues at work,
4
relatives, and friends
2
ACCEPTED MANUSCRIPT KEYWORDS. Bladder exstrophy–epispadias complex; mothers’ experience; genital malformations
Introduction The bladder exstrophy–epispadias complex (BEEC) represents a spectrum of urogenital
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malformations from epispadias (E) and classic bladder exstrophy (CBE) to cloacal exstrophy (EC). The incidence across the entire spectrum of deformities varies from 1/30,000 for CBE to 1/200,000 for EC [1]. The M/F ratio varies from 1.1:1 to 6.0:1; this ratio is reversed for cloacal exstrophy [2].
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The more innovative pathogenic theories are based on the hypothesis of a polygenic gene–gene interaction and multifactorial gene–environment interactions [3]. Prenatal diagnosis may be carried
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out between the 15th and 32nd week of pregnancy, depending on the severity of the defect and the experience of the operator who is performing the ultrasound [4].
Although a considerable amount of research has been conducted on BEEC psychological repercussions on children affected by it [5], only few scientific studies have investigated the
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emotional and social experiences related to giving birth to a child with BEEC so far [6,7]. Mothers and fathers of children suffering from exstrophy face various stressful factors, such as worrying about their child’s health, long hospital stays, and aspects concerning the child’s
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personal care. They may employ non-adaptive behaviours, such as escape and avoidance, when dealing with these situations [6]. Parents are often devastated when the pain becomes overwhelming,
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and may therefore tend to develop anxiety. This can have a negative effect on the future behaviour of their children, and exacerbate behaviours that these children already exhibit. These factors are further aggravated by the fact the parents do not usually receive counselling with regard to the birth of a child with this very debilitating condition [5]. Previous research on parents of children with BEEC focused mainly on the quality of life of the entire family, simultaneously evaluating adolescents’ conditions as well as parental opinion. Data show that quality of life as described by adolescents does not differ from that experienced by the reference population, whereas parents show emotional distress and family conflicts as a result of 3
ACCEPTED MANUSCRIPT their child’s illness [7,8]. On the basis of such results, we believe that parental emotional experience is a crucial aspect to be considered by analysts, in order to identify successful strategies used to cope with the child’s condition, gaining greater insight into the mothers’ experiences. The reasoning behind the selection of the analysed sample, namely mothers of children between 6 and 10 years of
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age, is twofold: our aim was (1) monitoring a period of time when surgery for continence is planned, and (2) evaluating the psychological wellbeing of mothers before the critical period of adolescence. The explorative qualitative study was carried out using an open questionnaire, as we believe that
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such testing experience is closer to the real experience of having a child with BEEC.
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Materials and methods Participants
Fourteen interviews were conducted with mothers of children with BEEC (nine boys and five girls). The following criteria were applied when selecting the participants in the study: (1) mother of a
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child born with exstrophy and aged between 6 and 10 years at the time of the study; (2) mother of a child being treated at the Urology Surgery ward of the Institute for Maternal and Child Health – IRCCS “Burlo Garofolo” Trieste, Italy. Out of the 14 participants, F1 and M6 had children who had
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an enterocystoplasty and emptied their bladders with intermittent catheterization using the Mitrofanoff procedure, and through the urethra. (Patients that participated in the study are identified
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as F when female and M when male, followed by a progressive number.) All the mothers we interviewed were unaware of their child’s disease until giving birth. We asked nine fathers to participate in the study, but only one of them accepted. Five fathers declared that they could not participate for lack of time, and the remaining four were not comfortable speaking about their child’s surgery. We therefore decided to interview mothers exclusively. None of the participants took part in an exstrophy support group Twenty-two mothers were contacted to participate in the research (Table 1). Those who rejected our invitation stated that they found it difficult to talk about their personal experiences with 4
ACCEPTED MANUSCRIPT their child. They also mentioned surgery, saying that it had been and still was too stressful to discuss. The research protocol was approved by the Ethics Committee of the Institute for Maternal
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and Child Health – IRCCS “Burlo Garofolo” Trieste, Italy.
Procedure
A surgeon from the multidisciplinary team contacted the mothers by phone to invite them to
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participate in the research. Interviews were conducted during one of the follow-up visits for their child. Interviews were organized by a psychologist specialising in urological malformations. They
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took place in a dedicated room of the Urology Surgery Paediatric ward, thus ensuring the highest possible level of privacy; they had an average duration of 60 minutes and were recorded in MP3 format. Open questions were asked on specific topics. Interviewees were given a general idea of the topics that would arise during the interview and they were free of answering no question they found
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overly invasive in terms of privacy. All participants signed an informed consent for privacy. Interviews enabled the analysts to collect stories of individuals who had directly or indirectly lived with BEEC and had gone through a long-term clinical experience [11]. Questions focused on four
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aspects that are considered relevant for this type of disease: (1) impact of the diagnosis, (2) relationship with the medical team, (3) relationship with the partner, (4) social life: at work, with
Results
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relatives and with friends (Table 2)
The four aspects that were discussed in each interview and the relevant answers were then summarised to facilitate subsequent analysis and evaluation.
Impact of the diagnosis
5
ACCEPTED MANUSCRIPT All the mothers told very vivid and articulated stories. They reported experiencing very powerful emotions, that are effectively described in the following answer provided by one of the interviewees: “many of my memories have faded but the fear, anxiety and terror that I felt during that time will stay with me forever”. They all said that they had been shocked at the time of the
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birth. “Not being able to know whether it was a boy or a girl”, “not being able to know what was wrong with my child”, “the looks on the faces of the doctors and the obstetrician when they saw my child”: these memories persist, even years after the birth. The birth of a child with BEEC was
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defined as “being betrayed by life”. A strong sense of guilt was also mentioned, because “I
followed all the rules to be a good mother but I failed”. These emotions provide evidence of
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mothers feeling crashed by their sense of responsibility and guilt for giving birth to an unhealthy child with a very complex condition affecting its genital organs. The mothers described being told about the diagnosis as traumatising, especially due to uncertainty regarding the child’s health conditions, the need to refer to more specialised centres, and doubts on the gender of their unborn
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baby. In the days immediately after the birth the new-borns’ lives were at risk. Two women had to be sedated for several days due to the severity of their psychological reaction to the health conditions of their babies. These types of experiences result in stories concerning the birth of the
EP
child that are highly distressing and laden with fear. Mothers reported feeling devastated every time their babies were undergoing complex
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surgery, as those moments reminded them of the first few days after the birth of their babies, when they bore such a huge emotional load. Relationship with the medical team The results show that, on the one hand, all mothers at some point in their children’s life felt they wanted to know more about their condition, and yet, on the other hand, they refused to discuss the situation and accept advice from the multidisciplinary team in charge of their children. Mothers described their continuous need for reassurance and news about their child’s condition so they could “understand the situation and know what to do”. However, they also declared not wanting to 6
ACCEPTED MANUSCRIPT discuss the situation and rejecting every aspect related to exstrophy: “I didn’t go to appointments. I was tired of hearing about plans for further surgery”; “Life already seemed unbearable and I didn’t want to know more about what might happen”; “I’m frightened by the uncertainty that lies ahead. I’m scared because my child feels fragile and so every little health issue feels like going back in
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time”.
Relationship with the partner
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The interviewer asked questions regarding life with the partner, including the couples’ emotional and sexual life. However, only two women elaborated on the topic, describing their relationship and
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sexual life with the partner; the others provided only generic and dismissive answers, such as “it’s fine”. The interviews, therefore, did not lead to any in-depth analysis of the topic, nor did they shed light on possible points of strength or weakness in the relationships. When the mothers were asked these questions, they often brought the focus back to the first few days after their child was born.
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Twelve mothers agreed to discuss the topic of emotional and sexual relationships, but repeatedly shifted the focus of the conversation to other areas of their lives and labelled this aspect as
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“complex” or “not something their child was ready to face yet”.
Social life (at work, with relatives, and with friends)
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The interviews showed that giving birth to a child with exstrophy led to significant changes in the mother’s professional life. One mother was an accountant before the birth of her child, but started to work as a nurse afterwards. Seven mothers who worked full time before the pregnancy began to work part time and in less demanding positions after their babies were born. Their answers indicate that these changes originated from their desire to spend more time with their children, who needed constant help. Six mothers left their job and stopped working altogether. Four mothers pointed out that they felt extremely frustrated by the changes occurred in their working situation. Seven reported that such changes were frustrating but necessary for their child’s 7
ACCEPTED MANUSCRIPT sake. Three described their sense of guilt being at work when there was a child with such a severe condition waiting at home, which is why they finally felt that giving up their job was the only viable solution. Five mothers out of 14 reported feeling a lack of support from their families, whereas the remaining seven felt they could entirely rely on their own families and relatives.
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Ten mothers out of 14 reported a decrease in their social activities, especially those
involving their friends, as they feared their children might have elicited a sense of pity and/or curiosity because of their genital organs. Four mothers opted to speak openly about their child’s
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health issues with closer friends and relatives. They also discussed how they would allow their child to have a certain amount of independence, including managing their urinary continence using a
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catheter. Seven interviewees remembered trying not to talk to anyone about their child’s issues to avoid being labelled as “the mother of that poor little child”. They explained that they limited their social life to what was strictly necessary “to protect my child”.
Three mothers out of 14 spoke of their lack of acceptance, years after their child was
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diagnosed: “Sometimes, I feel as though I couldn’t accept the disease. It feels as if life had dealt me a hefty blow”. The whole situation was described as “unbelievable”, like something unreal, a dream, especially in one case, where the interviewee reported feeling as if she found herself in a whirlwind
EP
that she could not stop or control, or as if she were suffocating. One of the mothers used the generic term “that thing” to refer to BEEC. Such choice of words suggests the need to distance oneself from
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the issue, and remain as unaware as possible of reality and the actions that must be taken. The main data from the interview were summarized in Table 3.
Discussion The birth of a child with a malformation, who needs invasive surgery after birth and then periodically throughout his/her life, generates a significant amount of stress [12,13]. Our research on the experience of mothers of children with BEEC has shown that emotions such as underlying anger, disappointment and fear are always present in the lives of the parents. The emotional states 8
ACCEPTED MANUSCRIPT that interviewees described corroborate results of previous scientific studies [6,8]. We decided to let these women voice their feelings, listening to them and suggesting specific topics for discussion, such as their personal lives and stories, which are particularly difficult to assess using standardized instruments. Our explorative qualitative interviews allowed access to specific aspects of these
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women’s stories in a way that indicated what content was of most interest for them. The results thus obtained confirm data previously found in the literature, and provide a clearer definition of the psychological dynamics and strategies implemented by mothers facing the birth of a child with
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BEEC. Our observations will be made available to the multidisciplinary medical team, with the aim of providing useful tools that may help improve timing in the administration of care, raising
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awareness about these conditions, which are corollary to the disease, and possible ways of coping with them.
When facing difficult situations, individuals often go through a series of emotions that come in specific stages: (1) denial; (2) anger; (3) bargaining; (4) depression; and finally (5) acceptance
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[14]. The analysis of the topics discussed during the semi-structured interviews showed that, even
after 6 or 10 years, some of the participants had yet to reach the final stage of acceptance. All mothers declared that every follow-up surgical operation their child underwent reminded them of
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distressing moments in the past, when their child was born. This allowed us to appreciate the difficulty of accepting a disease that continuously requires finding new coping mechanisms while
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the child grows. All of the mothers described feeling shocked at the time of the birth because they had not been able to deliver healthy children [15] and because of the malformation characteristics, and the uncertainty surrounding their child’s life and future [16,17]. Therefore, planning a support programme for women facing this kind of situation is of utmost importance. The medical team in charge of the child is seen in two opposite ways, namely both as a source of information used to gain greater awareness about the disease, and as an entity that must be avoided to receive no more bad news. Mothers indicated not seeking information and not having direct contact with the issue as a temporary coping mechanism. Greater emotional awareness of 9
ACCEPTED MANUSCRIPT both caregivers and parents, as regards the latter’s reaction to the malformation and the consequent surgery, could result in significant improvements in the family’s life and relation with the caregiving team. Multidisciplinary teams should therefore always include a psychologist monitoring
provide space and time for dealing with the child’s BEEC.
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emotional reactions to and interaction with healthcare operators, to facilitate communication and
Aspects regarding life with the partner were mentioned only in passing and almost
immediately dismissed by the interviewees, who preferred to bring back the focus on the birth of
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their child. Participants seemed eager to avoid discussing aspects linked to their children’s sexual life, which would certainly be influenced by BEEC once they became adolescents.
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The reactions that parents had regarding the world outside their family and the ways in which they established relationships and communicated with others are particularly significant. During the interviews, mothers described having conflicting wishes: on the one hand, they wanted to experience more freedom in their social life, while, on the other hand, they kept shutting
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themselves off, avoiding certain topics, and enjoying a certain degree of anonymity so as not to be labelled as parents of a child with a disability or with peculiar problems. The inadequate amount of information and the uncertainty of living day by day without clear answers on how the disease
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would evolve feed the desire to hide the hard truth of a disease requiring very invasive interventions, often involving unnatural procedures, such as the use of a catheterization. We believe that when a
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mother of a 6–10-year-old is still living her child’s birth as traumatic, unable to talk about her child’s condition and living a life of isolation because of it, this could represent a potential risk when the time of separation at adolescence comes. Indeed, psychological effects related to the trauma experienced by the mother, when added to the difficulties strictly related to the disease itself, may further deteriorate the emotional state of the child reaching adolescence. Admittedly, the study has various limits, including (1) the limited number of participants, (2) the absence of descriptions given by the fathers on how they dealt with the mothers’ experiences, and (3) the fact that the interviews took place in the hospital, which constituted a non-neutral 10
ACCEPTED MANUSCRIPT environment, having a significant emotional impact on the mothers. Unfortunately, the uneven geographical distribution of the participants made it impossible to conduct the interviews in more neutral locations.
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Conclusions We believe that giving birth to a child with BEEC triggers very complex experiences for the mother, and a need to change family expectations and dynamics. The results suggest that planning a support
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programme may have a significant impact on the way mothers deal with experiences of fear and anger influencing their relationship with the child, from his/her birth onwards. Using adaptive
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procedures to face the disease (1) would allow the mother, but also the child and the whole family, to lead a more serene life, with greater awareness of the disease, and (2) may help improving the surgery planning process with the multidisciplinary team.
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Acknowledgements
We would like to express our gratitude to all the women and mothers who took part in this study. We are very grateful for their willingness to open up and share their personal feelings, and we
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admire their strength and courage in facing everyday life. We would also like acknowledge the work of the three reviewers, whose insightful observations prompted us to re-examine and improve
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our research
Conflict of interest None.
Funding None.
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ACCEPTED MANUSCRIPT References
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survey of 214 families with bladder exstrophy-epispadias complex. J Urol 2008;179:1539–43.
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J Rare Dis 2009;4:23.
Boyadjiev SA, Dodson JL, Radford CL, Ashrafi GH, Beaty TH, Mathews RI, et al. Clinical
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[3]
and molecular characterization of the bladder exstrophy-epispadias complex: Analysis of 232
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Diseth TH, Emblem R, Schultzá A. Mental health, psychosocial functioning, and quality of
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life in patients with bladder exstrophy and epispadias an overview. World J of Urol 1999;17:239–
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Dodson JL, Furth SL, Yenokyan G, Alcorn K, Diener-West M, Wu AW, et al. Parent 12
ACCEPTED MANUSCRIPT perspectives of health related quality of life for adolescents with bladder exstrophy-epispadias as measured by the Child Health Questionnaire-Parent Form 50™. J Urol 2010;184:1656–61. [10]
Hurrell RA, Fullwood C, Keys J, Dickson AP, Fishwick J, Whitnall B, et al. Psychosocial
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Di Grazia M, Rigamonti W. Le malformazioni urogenitali complesse: la presa in carico
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chirurgica e psicologica del bambino – adolescente e della famiglia. Medico e Bambino 2014;
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17(8). http://www.medicoebambino.com/?id=IPS1408_10.html.
Morrison DS, Robertson A, Chalmers J, Youngson G, Ahmed SF. The psychological impact
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Kübler-Ross E. On grief and grieving: finding the meaning of grief through the five stages of loss. New York
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City: Simon & Schuster Ltd, 2005.
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[16]
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natal modifications in parental mental representations in three cases of fetal gastroschisis diagnosed during pregnancy. Infant Ment Health J 2015;36:613–22. Giuliani R, Tripani A, Pellizzoni S, Clarici A, Lonciari I, D’Ottavio G, et al. Pregnancy and
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Di Grazia M, Pellizzoni S, Tonegatti GL, Giurici N, Rigamonti W. Psychosexual development’s management
of bladder exstrophy epispadias in complex patients. J Peditr Urol (in press).
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ACCEPTED MANUSCRIPT Table 1 Distribution of background variables in the sample. N
14 (5 female and 9 male)
40.14
SD
3.97
Range
35-45
Years of schooling 7.85
SD
1.34
Range
6-10
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Mean
Mothers’ place of origin North Italy
5
Central Italy
5
1
In couple Employment status
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Employee
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Single
4
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South Italy Marital state
Unemployed
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Mean
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Age (years)
13 8 6
ACCEPTED MANUSCRIPT Table 2 Specific questions asked during the semi-structured interview. 1
Impact of the diagnosis
Tell me what you went through when your child was born
How did exstrophy affect you?
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How were you told about the disease?
What difficulties did you experience in the days following the birth of your child? 2
Relationship with the medical team
3
Relationship with the partner
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Did you find it difficult to communicate with the doctors?
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Were the doctors you met understanding and accommodating?
Has the relationship between you and your partner changed after the birth of your child? Has your intimate/sexual behaviour towards one another changed since your child with exstrophy was born?
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Have your life choices as a couple and as individuals been influenced by the birth of your child? Given that bladder exstrophy affects the genital organs, have you or your partner ever discussed emotional life and sexuality with your child?
Social life: relationships at work, with relatives and with friends
EP
4
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Have you been able to conciliate your work with your family since your child was born? Has your relationship with your network of friends changed since your child was born? Has your relationship with other family members changed since your child was born? Did you or your partner ever talk to friends and relatives about your child being born with exstrophy?
ACCEPTED MANUSCRIPT Table 3 Interview topics. N Impact of the diagnosis
Relationship with the medical team Ambivalence of feelings towards the medical team
12
Relationship with the partner In-depth description of the relationship and 2
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sexual life Social life
The mother starts working in a less demanding position
7
5
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EP
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Lack of family support Decrease in social activities
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14
SC
Traumatic experience
10
S1477-5131(16)30247-9
DOI:
10.1016/j.jpurol.2016.07.015
Reference:
JPUROL 2318
To appear in:
Journal of Pediatric Urology
Received Date: 7 March 2016 Accepted Date: 1 July 2016
Please cite this article as: Di Grazia M, Pellizzoni S, Tonegatti LG, Rigamonti W, Qualitative questionnaire on the psychosocial wellbeing of mothers of children with BEEC, Journal of Pediatric Urology (2016), doi: 10.1016/j.jpurol.2016.07.015. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Qualitative questionnaire on the psychosocial wellbeing of mothers of children with BEEC Massimo Di Grazia a, Sandra Pellizzoni b,*, Luca Giacomo Tonegatti b, Waifro Rigamonti a,c a
Department of Life Science, University of Trieste, Trieste, Italy c
Institute for Maternal and Child Health – IRCCS “Burlo Garofolo”, Trieste, Italy
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b
Department of Medical, Surgical and Health Science, University of Trieste, Trieste, Italy
* Corresponding author. Department of Life Science, Via Weiss 21, Building W, University of Trieste, 34128 Trieste, Italy. Tel.: +39 339 342 3876; fax: +39 040 675 8539
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E-mail address: [email protected] (S. Pellizzoni).
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Summary Introduction
The bladder exstrophy–epispadias complex (BEEC) represents a spectrum of malformations that affect the anatomical and functional structure of the urogenital system. The parents of patients
TE D
affected by this condition are subject to particularly stressful situations, such as worrying about their child’s health, long hospital stays, concerns about the health and constant need for personal care for their children, that can profoundly compromise the quality of family life.
EP
Objective
The objective of this explorative qualitative study is to evaluate the social situation and the
AC C
psychological strategies implemented by the mothers of children between 6 and 10 years of age who are affected by BEEC. Study design
Fourteen mothers of children aged 6–10 years and affected by BEEC (9 boys and 5 girls) were interviewed. Data on the mothers’ experiences were collected through semi-structured interviews (Table) Results
1
ACCEPTED MANUSCRIPT The qualitative analysis of the interviews showed that participants described experiences that were characterised by emotions such as fear and anger. Each mother had implemented a different and, sometimes, dysfunctional strategy in order to cope with the complex situation of the son/daughter. The aspects that most clearly emerged from mothers’ descriptions were (1) the traumatic situation
RI PT
at the birth of the baby, (2) the sense of embarrassment concerning the pathological condition as the child was growing and the consequent sense of isolation of the mother, and (3) the fluctuation of feelings towards the multidisciplinary staff, which was sometimes seen as an important source of
SC
help and some other times as too destabilising and not helpful at all. Discussion
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The study provided some insight into the psychological and social conditions experienced by mothers of children with BEEC, which could serve as a basis for developing multidisciplinary teams with greater awareness about families living with this condition and better timing in addressing their needs.
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Conclusions
Mothers of children with BEEC show emotional and social difficulties. This is a crucial aspect to consider when planning a multidisciplinary approach to the treatment/therapy, especially
EP
considering that children examined in this study are approaching adolescence.
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Table Semi-structured interview topic. Number of questions
1 Impact of the diagnosis
4
2 Relationship with the medical team
2
3 Relationship with the partner
4
4 Social life: relationship with colleagues at work,
4
relatives, and friends
2
ACCEPTED MANUSCRIPT KEYWORDS. Bladder exstrophy–epispadias complex; mothers’ experience; genital malformations
Introduction The bladder exstrophy–epispadias complex (BEEC) represents a spectrum of urogenital
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malformations from epispadias (E) and classic bladder exstrophy (CBE) to cloacal exstrophy (EC). The incidence across the entire spectrum of deformities varies from 1/30,000 for CBE to 1/200,000 for EC [1]. The M/F ratio varies from 1.1:1 to 6.0:1; this ratio is reversed for cloacal exstrophy [2].
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The more innovative pathogenic theories are based on the hypothesis of a polygenic gene–gene interaction and multifactorial gene–environment interactions [3]. Prenatal diagnosis may be carried
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out between the 15th and 32nd week of pregnancy, depending on the severity of the defect and the experience of the operator who is performing the ultrasound [4].
Although a considerable amount of research has been conducted on BEEC psychological repercussions on children affected by it [5], only few scientific studies have investigated the
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emotional and social experiences related to giving birth to a child with BEEC so far [6,7]. Mothers and fathers of children suffering from exstrophy face various stressful factors, such as worrying about their child’s health, long hospital stays, and aspects concerning the child’s
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personal care. They may employ non-adaptive behaviours, such as escape and avoidance, when dealing with these situations [6]. Parents are often devastated when the pain becomes overwhelming,
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and may therefore tend to develop anxiety. This can have a negative effect on the future behaviour of their children, and exacerbate behaviours that these children already exhibit. These factors are further aggravated by the fact the parents do not usually receive counselling with regard to the birth of a child with this very debilitating condition [5]. Previous research on parents of children with BEEC focused mainly on the quality of life of the entire family, simultaneously evaluating adolescents’ conditions as well as parental opinion. Data show that quality of life as described by adolescents does not differ from that experienced by the reference population, whereas parents show emotional distress and family conflicts as a result of 3
ACCEPTED MANUSCRIPT their child’s illness [7,8]. On the basis of such results, we believe that parental emotional experience is a crucial aspect to be considered by analysts, in order to identify successful strategies used to cope with the child’s condition, gaining greater insight into the mothers’ experiences. The reasoning behind the selection of the analysed sample, namely mothers of children between 6 and 10 years of
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age, is twofold: our aim was (1) monitoring a period of time when surgery for continence is planned, and (2) evaluating the psychological wellbeing of mothers before the critical period of adolescence. The explorative qualitative study was carried out using an open questionnaire, as we believe that
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such testing experience is closer to the real experience of having a child with BEEC.
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Materials and methods Participants
Fourteen interviews were conducted with mothers of children with BEEC (nine boys and five girls). The following criteria were applied when selecting the participants in the study: (1) mother of a
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child born with exstrophy and aged between 6 and 10 years at the time of the study; (2) mother of a child being treated at the Urology Surgery ward of the Institute for Maternal and Child Health – IRCCS “Burlo Garofolo” Trieste, Italy. Out of the 14 participants, F1 and M6 had children who had
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an enterocystoplasty and emptied their bladders with intermittent catheterization using the Mitrofanoff procedure, and through the urethra. (Patients that participated in the study are identified
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as F when female and M when male, followed by a progressive number.) All the mothers we interviewed were unaware of their child’s disease until giving birth. We asked nine fathers to participate in the study, but only one of them accepted. Five fathers declared that they could not participate for lack of time, and the remaining four were not comfortable speaking about their child’s surgery. We therefore decided to interview mothers exclusively. None of the participants took part in an exstrophy support group Twenty-two mothers were contacted to participate in the research (Table 1). Those who rejected our invitation stated that they found it difficult to talk about their personal experiences with 4
ACCEPTED MANUSCRIPT their child. They also mentioned surgery, saying that it had been and still was too stressful to discuss. The research protocol was approved by the Ethics Committee of the Institute for Maternal
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and Child Health – IRCCS “Burlo Garofolo” Trieste, Italy.
Procedure
A surgeon from the multidisciplinary team contacted the mothers by phone to invite them to
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participate in the research. Interviews were conducted during one of the follow-up visits for their child. Interviews were organized by a psychologist specialising in urological malformations. They
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took place in a dedicated room of the Urology Surgery Paediatric ward, thus ensuring the highest possible level of privacy; they had an average duration of 60 minutes and were recorded in MP3 format. Open questions were asked on specific topics. Interviewees were given a general idea of the topics that would arise during the interview and they were free of answering no question they found
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overly invasive in terms of privacy. All participants signed an informed consent for privacy. Interviews enabled the analysts to collect stories of individuals who had directly or indirectly lived with BEEC and had gone through a long-term clinical experience [11]. Questions focused on four
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aspects that are considered relevant for this type of disease: (1) impact of the diagnosis, (2) relationship with the medical team, (3) relationship with the partner, (4) social life: at work, with
Results
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relatives and with friends (Table 2)
The four aspects that were discussed in each interview and the relevant answers were then summarised to facilitate subsequent analysis and evaluation.
Impact of the diagnosis
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ACCEPTED MANUSCRIPT All the mothers told very vivid and articulated stories. They reported experiencing very powerful emotions, that are effectively described in the following answer provided by one of the interviewees: “many of my memories have faded but the fear, anxiety and terror that I felt during that time will stay with me forever”. They all said that they had been shocked at the time of the
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birth. “Not being able to know whether it was a boy or a girl”, “not being able to know what was wrong with my child”, “the looks on the faces of the doctors and the obstetrician when they saw my child”: these memories persist, even years after the birth. The birth of a child with BEEC was
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defined as “being betrayed by life”. A strong sense of guilt was also mentioned, because “I
followed all the rules to be a good mother but I failed”. These emotions provide evidence of
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mothers feeling crashed by their sense of responsibility and guilt for giving birth to an unhealthy child with a very complex condition affecting its genital organs. The mothers described being told about the diagnosis as traumatising, especially due to uncertainty regarding the child’s health conditions, the need to refer to more specialised centres, and doubts on the gender of their unborn
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baby. In the days immediately after the birth the new-borns’ lives were at risk. Two women had to be sedated for several days due to the severity of their psychological reaction to the health conditions of their babies. These types of experiences result in stories concerning the birth of the
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child that are highly distressing and laden with fear. Mothers reported feeling devastated every time their babies were undergoing complex
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surgery, as those moments reminded them of the first few days after the birth of their babies, when they bore such a huge emotional load. Relationship with the medical team The results show that, on the one hand, all mothers at some point in their children’s life felt they wanted to know more about their condition, and yet, on the other hand, they refused to discuss the situation and accept advice from the multidisciplinary team in charge of their children. Mothers described their continuous need for reassurance and news about their child’s condition so they could “understand the situation and know what to do”. However, they also declared not wanting to 6
ACCEPTED MANUSCRIPT discuss the situation and rejecting every aspect related to exstrophy: “I didn’t go to appointments. I was tired of hearing about plans for further surgery”; “Life already seemed unbearable and I didn’t want to know more about what might happen”; “I’m frightened by the uncertainty that lies ahead. I’m scared because my child feels fragile and so every little health issue feels like going back in
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time”.
Relationship with the partner
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The interviewer asked questions regarding life with the partner, including the couples’ emotional and sexual life. However, only two women elaborated on the topic, describing their relationship and
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sexual life with the partner; the others provided only generic and dismissive answers, such as “it’s fine”. The interviews, therefore, did not lead to any in-depth analysis of the topic, nor did they shed light on possible points of strength or weakness in the relationships. When the mothers were asked these questions, they often brought the focus back to the first few days after their child was born.
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Twelve mothers agreed to discuss the topic of emotional and sexual relationships, but repeatedly shifted the focus of the conversation to other areas of their lives and labelled this aspect as
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“complex” or “not something their child was ready to face yet”.
Social life (at work, with relatives, and with friends)
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The interviews showed that giving birth to a child with exstrophy led to significant changes in the mother’s professional life. One mother was an accountant before the birth of her child, but started to work as a nurse afterwards. Seven mothers who worked full time before the pregnancy began to work part time and in less demanding positions after their babies were born. Their answers indicate that these changes originated from their desire to spend more time with their children, who needed constant help. Six mothers left their job and stopped working altogether. Four mothers pointed out that they felt extremely frustrated by the changes occurred in their working situation. Seven reported that such changes were frustrating but necessary for their child’s 7
ACCEPTED MANUSCRIPT sake. Three described their sense of guilt being at work when there was a child with such a severe condition waiting at home, which is why they finally felt that giving up their job was the only viable solution. Five mothers out of 14 reported feeling a lack of support from their families, whereas the remaining seven felt they could entirely rely on their own families and relatives.
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Ten mothers out of 14 reported a decrease in their social activities, especially those
involving their friends, as they feared their children might have elicited a sense of pity and/or curiosity because of their genital organs. Four mothers opted to speak openly about their child’s
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health issues with closer friends and relatives. They also discussed how they would allow their child to have a certain amount of independence, including managing their urinary continence using a
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catheter. Seven interviewees remembered trying not to talk to anyone about their child’s issues to avoid being labelled as “the mother of that poor little child”. They explained that they limited their social life to what was strictly necessary “to protect my child”.
Three mothers out of 14 spoke of their lack of acceptance, years after their child was
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diagnosed: “Sometimes, I feel as though I couldn’t accept the disease. It feels as if life had dealt me a hefty blow”. The whole situation was described as “unbelievable”, like something unreal, a dream, especially in one case, where the interviewee reported feeling as if she found herself in a whirlwind
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that she could not stop or control, or as if she were suffocating. One of the mothers used the generic term “that thing” to refer to BEEC. Such choice of words suggests the need to distance oneself from
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the issue, and remain as unaware as possible of reality and the actions that must be taken. The main data from the interview were summarized in Table 3.
Discussion The birth of a child with a malformation, who needs invasive surgery after birth and then periodically throughout his/her life, generates a significant amount of stress [12,13]. Our research on the experience of mothers of children with BEEC has shown that emotions such as underlying anger, disappointment and fear are always present in the lives of the parents. The emotional states 8
ACCEPTED MANUSCRIPT that interviewees described corroborate results of previous scientific studies [6,8]. We decided to let these women voice their feelings, listening to them and suggesting specific topics for discussion, such as their personal lives and stories, which are particularly difficult to assess using standardized instruments. Our explorative qualitative interviews allowed access to specific aspects of these
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women’s stories in a way that indicated what content was of most interest for them. The results thus obtained confirm data previously found in the literature, and provide a clearer definition of the psychological dynamics and strategies implemented by mothers facing the birth of a child with
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BEEC. Our observations will be made available to the multidisciplinary medical team, with the aim of providing useful tools that may help improve timing in the administration of care, raising
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awareness about these conditions, which are corollary to the disease, and possible ways of coping with them.
When facing difficult situations, individuals often go through a series of emotions that come in specific stages: (1) denial; (2) anger; (3) bargaining; (4) depression; and finally (5) acceptance
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[14]. The analysis of the topics discussed during the semi-structured interviews showed that, even
after 6 or 10 years, some of the participants had yet to reach the final stage of acceptance. All mothers declared that every follow-up surgical operation their child underwent reminded them of
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distressing moments in the past, when their child was born. This allowed us to appreciate the difficulty of accepting a disease that continuously requires finding new coping mechanisms while
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the child grows. All of the mothers described feeling shocked at the time of the birth because they had not been able to deliver healthy children [15] and because of the malformation characteristics, and the uncertainty surrounding their child’s life and future [16,17]. Therefore, planning a support programme for women facing this kind of situation is of utmost importance. The medical team in charge of the child is seen in two opposite ways, namely both as a source of information used to gain greater awareness about the disease, and as an entity that must be avoided to receive no more bad news. Mothers indicated not seeking information and not having direct contact with the issue as a temporary coping mechanism. Greater emotional awareness of 9
ACCEPTED MANUSCRIPT both caregivers and parents, as regards the latter’s reaction to the malformation and the consequent surgery, could result in significant improvements in the family’s life and relation with the caregiving team. Multidisciplinary teams should therefore always include a psychologist monitoring
provide space and time for dealing with the child’s BEEC.
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emotional reactions to and interaction with healthcare operators, to facilitate communication and
Aspects regarding life with the partner were mentioned only in passing and almost
immediately dismissed by the interviewees, who preferred to bring back the focus on the birth of
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their child. Participants seemed eager to avoid discussing aspects linked to their children’s sexual life, which would certainly be influenced by BEEC once they became adolescents.
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The reactions that parents had regarding the world outside their family and the ways in which they established relationships and communicated with others are particularly significant. During the interviews, mothers described having conflicting wishes: on the one hand, they wanted to experience more freedom in their social life, while, on the other hand, they kept shutting
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themselves off, avoiding certain topics, and enjoying a certain degree of anonymity so as not to be labelled as parents of a child with a disability or with peculiar problems. The inadequate amount of information and the uncertainty of living day by day without clear answers on how the disease
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would evolve feed the desire to hide the hard truth of a disease requiring very invasive interventions, often involving unnatural procedures, such as the use of a catheterization. We believe that when a
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mother of a 6–10-year-old is still living her child’s birth as traumatic, unable to talk about her child’s condition and living a life of isolation because of it, this could represent a potential risk when the time of separation at adolescence comes. Indeed, psychological effects related to the trauma experienced by the mother, when added to the difficulties strictly related to the disease itself, may further deteriorate the emotional state of the child reaching adolescence. Admittedly, the study has various limits, including (1) the limited number of participants, (2) the absence of descriptions given by the fathers on how they dealt with the mothers’ experiences, and (3) the fact that the interviews took place in the hospital, which constituted a non-neutral 10
ACCEPTED MANUSCRIPT environment, having a significant emotional impact on the mothers. Unfortunately, the uneven geographical distribution of the participants made it impossible to conduct the interviews in more neutral locations.
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Conclusions We believe that giving birth to a child with BEEC triggers very complex experiences for the mother, and a need to change family expectations and dynamics. The results suggest that planning a support
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programme may have a significant impact on the way mothers deal with experiences of fear and anger influencing their relationship with the child, from his/her birth onwards. Using adaptive
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procedures to face the disease (1) would allow the mother, but also the child and the whole family, to lead a more serene life, with greater awareness of the disease, and (2) may help improving the surgery planning process with the multidisciplinary team.
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Acknowledgements
We would like to express our gratitude to all the women and mothers who took part in this study. We are very grateful for their willingness to open up and share their personal feelings, and we
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admire their strength and courage in facing everyday life. We would also like acknowledge the work of the three reviewers, whose insightful observations prompted us to re-examine and improve
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our research
Conflict of interest None.
Funding None.
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ACCEPTED MANUSCRIPT References
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ACCEPTED MANUSCRIPT Table 1 Distribution of background variables in the sample. N
14 (5 female and 9 male)
40.14
SD
3.97
Range
35-45
Years of schooling 7.85
SD
1.34
Range
6-10
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Mean
Mothers’ place of origin North Italy
5
Central Italy
5
1
In couple Employment status
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Employee
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Single
4
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South Italy Marital state
Unemployed
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Mean
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Age (years)
13 8 6
ACCEPTED MANUSCRIPT Table 2 Specific questions asked during the semi-structured interview. 1
Impact of the diagnosis
Tell me what you went through when your child was born
How did exstrophy affect you?
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How were you told about the disease?
What difficulties did you experience in the days following the birth of your child? 2
Relationship with the medical team
3
Relationship with the partner
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Did you find it difficult to communicate with the doctors?
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Were the doctors you met understanding and accommodating?
Has the relationship between you and your partner changed after the birth of your child? Has your intimate/sexual behaviour towards one another changed since your child with exstrophy was born?
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Have your life choices as a couple and as individuals been influenced by the birth of your child? Given that bladder exstrophy affects the genital organs, have you or your partner ever discussed emotional life and sexuality with your child?
Social life: relationships at work, with relatives and with friends
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4
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Have you been able to conciliate your work with your family since your child was born? Has your relationship with your network of friends changed since your child was born? Has your relationship with other family members changed since your child was born? Did you or your partner ever talk to friends and relatives about your child being born with exstrophy?
ACCEPTED MANUSCRIPT Table 3 Interview topics. N Impact of the diagnosis
Relationship with the medical team Ambivalence of feelings towards the medical team
12
Relationship with the partner In-depth description of the relationship and 2
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sexual life Social life
The mother starts working in a less demanding position
7
5
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Lack of family support Decrease in social activities
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14
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Traumatic experience
10