284 I.H. Zaal-Schuller et al. / Research in Developmental Disabilities 59 (2016) 283–293 What this paper adds This study compared the experiences of parents and the involved physicians during the EoLDM process for the same child with PIMD. A long-lasting treatment relationship facilitated the EoLDM process, although negative healthcare encounters could also lead to distrust. Previous healthcare encounters also led parents to anticipate on EoLDM in the future. Parents’ anticipation of the EoLDM process facilitated an earlier start of conversations about EoLDs, while the child was still in a stable condition. Parents highly valued the physicians’ advice. Moreover, they also valued information from parents who had already experienced the same process. Disturbing disagreements particularly occurred when decisions had to be made under time pressure with a physician who did not yet know the child. Parents and physicians agreed that disagreements sometimes improved the EoLDM process because it made physicians look for alternative treatments more suitable for the child. Physicians need to be aware that parents are the experts on the well-being of their child and should treat parents accordingly. This awareness implies that parents’ wishes should be taken into account during the entire process of EoLDM. 1. Introduction The majority of children with profound intellectual and multiple disabilities (PIMD) have complex, life-limiting conditions that are characterized by prognostic uncertainty (Bittles, Petterson, Sullivan, Hussain, Glasson, & Montgomery, 2002; Chaney & Eyman, 2000; Katz, 2003; Oeseburg, Dijkstra, Groothoff, Reijneveld, & Jansen, 2011; Strauss, Shavelle, & Anderson, 1998; Tamburro, Shaffer, Hahnlen, Felker, & Ceneviva, 2011; van Schrojenstein Lantman-de Valk et al., 1997). Parents are continuously confronted with challenging decisions in the light of this uncertainty. Children with PIMD lack decision-making capacity, and for this reason, parents and physicians have to make decisions on their behalf. This responsibility can be very burdensome, especially if it concerns an end-of-life decision (EoLD). EoLDs include decisions to withhold or withdraw lifeprolonging treatments (such as mechanical ventilation, tube feeding and dialysis) and to alleviate pain or other symptoms by using drugs with a possible life-shortening effect that may lead to or hasten death, whether intentionally or not (van der Heide et al., 2003). In The Netherlands, legislation around EoLDs for children is recorded in the Dutch Medical Treatment Act (WGBO) (2006). In children who lack decision-making capacity, informed consent from their parents is needed before treatment can be started. However, if a parent refuses to allow treatment but there is no question that this treatment is obviously necessary, the physician is allowed to treat the child without parental consent. In cases in which treatment clearly will not be beneficial to the patient, physicians are not obligated to provide treatment, even when parents request it. In practice, parents are given ample opportunity to participate in the end-of-life decision-making (EoLDM) process as recommended in national guidelines. ("Dutch Medical Treatment Act (WGBO)," 2006; NVK [Paediatric Association Of The Netherlands], 2013) Children with PIMD account for a large part of hospital care in paediatric wards today (Berry et al., 2011; Namachivayam et al., 2010; Simon et al., 2010). Therefore, it is remarkable that very little is known about the experiences of parents and physicians during the EoLDM process for children with PIMD in a clinical setting. Previous research, mainly conducted in neonatal and paediatric intensive care units, has shown that parents’ points of view regarding decisions to continue or discontinue treatment (Madrigal, Carroll, Hexem, Faerber, Morrison, & Feudtner, 2012; McHaffie, Lyon, & Hume, 2001; Meert, Thurston, & Sarnaik, 2000; Michelson, Koogler, Sullivan, Ortega, Hall, & Frader, 2009; Sharman, Meert, & Sarnaik, 2005; Sullivan, Monagle, & Gillam, 2014) are not always in line with the points of view of the involved physicians. This difference regularly leads to conflicts (de Vos et al., 2011; Michelson et al., 2009; Studdert et al., 2003; Verhagen et al., 2009). In general, parents of children with PIMD have numerous encounters with healthcare professionals. These encounters may influence the way parents will experience the process of EoLDM for their child. However, whether and how these encounters influence parents’ experiences has not yet been investigated. The purpose of this qualitative study was to compare the experiences of parents and physicians who were involved in the EoLDM process of the same child with PIMD in The Netherlands. This study aims to answer three questions: (1) How do parents of children with PIMD experience the process of clinical EoLDM?; (2) How do their physicians experience this process?; and (3) What are the similarities and differences between parents’ and physicians’ experiences? 2. Methods 2.1. Study design and participants We performed a qualitative, retrospective study by means of semi-structured interviews with the parents and physicians of 14 children with PIMD for whom an EoLD had been made within the last two years. Parents were approached in different ways. First, all of the parents who were members of a parent organization for children with cerebral palsy (BOSK) received a letter in which they were invited to participate in the study. Second, all of the parents of children with PIMD who visited one of two large specialized day care centres received the same invitational letter. Third, all of the parents who visited the annual national meeting for children with PIMD received this letter. Fourth, Intellectual Disability-physicians and paediatricians who worked in the North-Holland, South-Holland and Utrecht regions were asked by an informative email to approach eligible parents to participate in this study. Parents who fulfilled the following criteria were invited to participate: (1) their child had a PIMD and a life-limiting condition, (2) their child was aged between 1 and 21 years at the time an EoLD had been considered and (3) the parents had discussed this decision with their child’s physician at one or more moments in the
I.H. Zaal-Schuller et al. / Research in Developmental Disabilities 59 (2016) 283–293 285 past two years in a clinical setting. Parents of children who had died after an EoLD had been made were included as well as parents of children who were still alive. We strived to achieve a large variety of cases according to the following patient-related characteristics: age, gender, diagnosis, prognosis and co-morbidity. We also sought variety according to the following parent-related characteristics: age, length of physician-family relationship, religion and ethnic background. Parents who wished to participate in the study were asked to contact the first author by telephone or by e-mail to arrange an interview. Four of the parents were not eligible for this study because they had not been involved in the EoLDM process or because the decision had been made too long ago. All of the participants provided audiotaped informed consent before participating in the interview. At the end of the interview, the interviewer asked the parents for their consent to contact their child’s physician for an interview. Physicians received ample information about the study via e-mail. Two weeks later the interviewer contacted them by telephone to ask for their consent. This time frame was chosen to give physicians sufficient time to consider their participation. During this phone call, physicians were given additional information about the study. They were ensured absolute anonymity and they were given the opportunity to ask any remaining questions. The hospital’s Medical Ethics Review Committee approved the protocol for this study. 2.2. Data collection The first author, who was an Intellectual Disability-physician in training at the time, conducted the semi-structured interviews with parents and physicians using the same interview guide. This interview guide was compiled after a literature search for questionnaires about this topic (Carnevale et al., 2007; Partridge et al., 2005; Provoost et al., 2006; Sharman et al., 2005; Williams et al., 2009). After discussing the questionnaire with a second researcher (MdV), some questions were added, and the definitive questionnaire was composed. The questionnaire contained questions to describe the EoLDM process for each individual child from the perspective of the participants. The interview guide contained open-ended questions about the motives for discussing an EoLD, the steps preceding an EoLD, the main considerations for an EoLD, the occurrence and resolution of conflicting points of view, who made the final decision and what could have been done better. All of the interviews were audiotaped. All but two of the parents chose to be interviewed at home. Most physicians were interviewed in the hospital; three physicians preferred to be interviewed at home. Parents and physicians were asked to provide demographic information before the open-ended questions were presented to them. 2.3. Qualitative data analysis The tape-recorded interviews were transcribed verbatim. The transcripts were analysed using the qualitative data analysis software, MaxQDA. An initial subsample of three transcripts was independently coded by the main researcher (IZ) and a research assistant to identify important and/or striking experiences of parents and physicians prior to, during and after the process of EoLDM. Subsequently, these two researchers compared their codings and constructed a coding dictionary that contained a detailed overview of all of the experiences mentioned by the respondents. A third investigator (MdV) then read three more interviews to check and complete this dictionary. With this final dictionary, the main researcher coded all of the interview transcripts. Two investigators (MdV and DW) randomly checked these codings. The overall agreement was practically 100%. Finally, three investigators (IS, MdV and DW) elaborately compared the applied codes across all of the interviews and, after extensive discussion, integrated them into discerning patterns regarding the experiences of both parents and physicians. These patterns are presented in the results section. 3. Results 3.1. Characteristics of the participants Parents of fourteen children agreed to participate in this study. For three children, the parents were interviewed together as a couple; for the remaining children, only the mothers were interviewed. All of the parents agreed that the interviewer could approach their child’s physician, except one couple. The parents of this child were involved in a disagreement with their child’s physician, and they feared that an interview would lead to an escalation. Of the thirteen physicians who we approached, two physicians refused to be interviewed because they believed it could harm their relationship with the child’s parents. The characteristics of the participants can be found in Table 1 (children), Table 2 (parents) and Table 3 (physicians). 3.2. Experiences of parents and physicians In analysing and comparing the interviews with the included parents and physicians, six main themes emerged. The integrated findings of both parents and physicians are presented according to these themes in consecutive order. An overview of these main experiences is presented in Table 4.