- Email: [email protected]
Social support received by women with intellectual and developmental disabilities during pregnancy and childbirth: An exploratory qualitative study
Midwifery 37 (2016) 57–64
Contents lists available at ScienceDirect
Midwifery journal homepage: www.elsevier.com/midw
Social support received by women with intellectual and developmental disabilities during pregnancy and childbirth: An exploratory qualitative study Lynne A. Potvin, H.B.A (Specialization in Psychology and Birth Doula)a, Hilary K. Brown, PhD (Postdoctoral Fellow)b, Virginie Cobigo, PhD (Associate Professor)c,n a b c
University of Ottawa, School of Psychology, 136 Jean Jacques Lussier (5078), Ottawa, Canada ON K1N 6N5 Women's College Research Institute, University of Toronto, Department of Psychiatry, 76 Grenville Street, Toronto, Canada ON M5S 1B2 University of Ottawa, School of Psychology, 136 Jean Jacques Lussier (5083), Ottawa, Canada ON K1N 6N5
art ic l e i nf o
a b s t r a c t
Article history: Received 18 November 2015 Received in revised form 25 March 2016 Accepted 10 April 2016
Objective: this study aims to contribute to the development of a conceptual framework that will inform maternity care improvements for expectant mothers with intellectual and developmental disabilities (IDD) by exploring the structure, functions, and perceived quality of social support received by women with IDD during pregnancy and childbirth. Design/setting: using a grounded theory approach, we conducted an exploratory study set in Ontario, Canada in 2015. Participants: the sample included four adult women with IDD who had given birth in the last five years. Measurements: data were collected using semi-structured interviews. Findings: the structure of social support received by women with IDD consisted of both formal and informal sources, but few or no friendships. Women with IDD reported high levels of informational and instrumental support and low levels of emotional support and social companionship. However, a high level of available support was not always perceived as beneficial. Emergent core categories suggest that social support is perceived as most effective when three conditions are met: (1) support is accessible, (2) support is provided by individuals expressing positive attitudes towards the pregnancy, and (3) autonomy is valued. Key conclusions and implications for practice: our study confirms and identifies important gaps in the social support received by expectant mothers with IDD. Women with IDD currently lack accessible informational support, emotional support, and social companionship during pregnancy and childbirth. Additional findings regarding the structure and functions of social support are presented, and a preliminary conceptual framework of effective social support during pregnancy and childbirth, as perceived by women with IDD is also proposed. Findings suggest that increasing support accessibility should be a social and clinical priority; however, maternity care providers should be aware of stigmatizing attitudes and respect the autonomy of pregnant women with IDD as they prepare for motherhood. & 2016 Elsevier Ltd. All rights reserved.
Keywords: Intellectual disability Developmental disability Pregnancy Childbirth Social support Grounded theory
Introduction Intellectual and developmental disabilities (IDD) are neurodevelopmental disorders characterised by limitations in cognitive and adaptive functioning (American Psychiatric Association, 2013). According to health administrative data from Ontario, Canada, the prevalence rate of IDD per 100 is 0.80 (Lin et al., 2013). Historically, n
Corresponding author. E-mail addresses: [email protected] (L.A. Potvin), [email protected] (H.K. Brown), [email protected] (V. Cobigo). http://dx.doi.org/10.1016/j.midw.2016.04.005 0266-6138/& 2016 Elsevier Ltd. All rights reserved.
many women with IDD were involuntary sterilised and institutionalized (Kempton and Kahn, 1991). Today, legislation protects the reproductive rights of women with IDD in most developed nations, including Canada (Blackford, 1993), and the United Nations Convention on the Rights of Persons with Disabilities (2006) reaffirms the right of persons with IDD to “marriage, family, parenthood and relationships, on an equal basis with others” (article 23.1). This changing context has significant implications for childbearing in this population. Many women with IDD have sexual relationships and desire children (Aunos and Feldman, 2002). Moreover, an increasing
58
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
number of women with IDD are using maternity services (Royal College of Midwives, 2000). The general fertility rate in women with IDD is 20.3 live births per 1000 women with IDD (Brown et al., 2016); pregnancy is therefore not uncommon in this population. Women with IDD have high rates of adverse perinatal outcomes, including preeclampsia, caesarean section, preterm birth, and perinatal mortality (McConnell et al., 2008a; Höglund, 2012; Mitra et al., 2015), suggesting that they are a high-risk maternity population whose individual needs should be supported. Social support can be conceptualised in terms of structure and function. Structure refers to the number of relationships one has and the degree of interconnectedness between these relationships (Sherbourne and Stewart, 1991). Relationships may be formal (e.g., paid sources such as healthcare professionals) or informal (e.g., unpaid sources such as family members and friends) (Lyons and Zarit, 1999). Function refers to the nature of social support provided and the degree to which the relationship serves a purpose (Sherbourne and Stewart, 1991). Commonly cited functions of social support are informational support, instrumental support, emotional support, and social companionship (Sherbourne and Stewart, 1991; Willis and Shinar, 2000). Informational support includes the provision of knowledge, advice, and guidance. Instrumental support includes practical support such as transportation, financial aid, and help with household chores. Emotional support is defined by demonstrations of caring and acceptance like sympathetic listening. Social companionship refers to relationships that engage in social activities (Willis and Shinar, 2000). Perceived quality of social support refers to the subjective evaluation of support received by an individual, which may be positive or negative (Cohen and Syme, 1985; Bramston et al., 1999; Lunsky and Benson, 2001). Research has identified pregnancy as a potentially stressful life event requiring social readjustment (Holmes and Rahe, 1967); however, it is especially stressful if it is unplanned (Geller, 2004) or high risk (Heaman, 1998). Women with IDD appear to be vulnerable to unwanted pregnancy (Wingfield et al., 1994; Servais et al., 2002) and represent a high-risk maternity population (McConnell et al., 2008a; Höglund, 2012; Mitra et al., 2015). Conversely, social support serves a protective function against the negative effects of stress (Cohen and Syme, 1985; Cohen and Wills, 1985) and contributes to the health and well-being of expectant mothers (Oakley, 1988). Effective and continuous social support, in general, has been associated with a reduced risk of emotional distress during pregnancy (Glazier et al., 2004), shorter labour, fewer obstetrical interventions (Lantz et al., 2005), and a lower risk for post partum depression (Morikawa et al., 2015). Moreover, research specific to women with IDD has demonstrated that adapted informational support increases self-efficacy in preparation for motherhood and confidence in making informed choices during pregnancy (McGarry et al., 2015). Most adults with IDD experience low to moderate levels of social support (Lunsky and Benson, 1999). However, only a handful of studies, from the United Kingdom, Ireland, Sweden, and Australia, have described social support received by women with IDD during pregnancy and childbirth. Themes that have emerged from these studies include inadequately addressed needs for informational and practical support (Höglund and Larsson, 2013; Mayes et al., 2006; Redshaw et al., 2013; Walsh-Gallagher et al., 2012), feelings of loneliness (Walsh-Gallagher et al., 2012; Walsh-Gallagher et al., 2013) and social isolation (Llewellyn and McConnell, 2002), and distress over prejudicial beliefs held by family members and health care professionals towards the pregnancy (McConnell and Llewellyn, 2002; Mayes et al., 2006; McConnell et al., 2008b; Walsh-Gallagher et al., 2012; Höglund and Larsson, 2013; Walsh-Gallagher et al., 2013).
As such, this study aims to contribute to the development of a conceptual framework that will inform maternity care improvements for expectant mothers with IDD by exploring the structure, functions, and perceived quality of social support received by women with IDD during pregnancy and childbirth.
Method Ethics approval was obtained from the Office of Research Ethics and Integrity at the University of “X”. Design/setting We conducted a qualitative study using a grounded theory approach. Using pre-existing knowledge to understand the data, grounded theory uses constant comparison to identify and compare emerging conceptual elements, ask questions of the data, and code data into categories based on their theoretical properties (Robson, 2002; Holton, 2007). On the basis of this in-depth analysis, it is possible to generate explanatory models that are grounded in the data (Robson, 2002). The study was conducted in Ontario, Canada, in 2015. Participants Participants were recruited using convenience sampling from local French and English service agencies for adults with IDD that agreed to forward information packages to potential participants. The sample included women who had a diagnosis of IDD (as confirmed by the service agency) and who: (1) were 18 years of age or older, (2) had a pregnancy that resulted in a live birth in the last five years, and (3) possessed sufficient verbal capacity to answer questions in an interview setting (e.g., able to recall experiences and verbally relay them to the interviewer). Procedures Consent and confidentiality Consent was evaluated through a series of five questions designed to assess: (a) capacity to consent (i.e., understanding of research purpose, risks, and benefits), (b) voluntariness, (c) understanding of the right to withdraw consent at any time without consequence, and (d) understanding of the right to refuse to answer questions (Arscott et al., 1998). Third parties present during the interview (i.e., case workers) had to be invited by the women and were required to sign a confidentiality agreement prior to the interview. Avoiding distress There is a potential for eliciting psychological discomfort when discussing sensitive topics and sharing personal information; therefore, we developed the following procedure: (a) adopted strong interviewing skills such as sensitivity and empathy; (b) took breaks where needed; (c) terminated the interview if it became too distressing; (d) assured participants that they could decline to answer any questions and withdraw from the study at any time without consequence; (e) remained with the participant and shifted the interview to pleasant exchanges before terminating the interaction if distress was suspected (Corbin and Morse, 2003). Measurement Instrument Data were collected in recorded one-on-one semi-structured
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
interviews. Key components of the interview guide included demographic information, questions related to pregnancy and childbirth, a modified version of the Social Support Self-Report (SSSR) (Lunsky and Benson, 1997, 2001), and a social circles task (Lunsky and Benson, 1999). The SSSR is a self-reported measure of the structure and perceived quality of social support received by adults with IDD. Questions about structure are Yes/No and openended. Questions about perceived quality use a 3-point Likert scale (a lot, sometimes, not at all), which has been shown to be appropriate for individuals with IDD (Hartley and MacLean, 2006). The SSSR has high internal consistency (Cronbach's α ¼ 0.85) and is correlated with other measures of social support (r ¼0.75) (Lunsky and Benson, 1997). We added prompts to SSSR questions to elicit detailed information about social support received during pregnancy and childbirth. The social circles task is a visual activity that facilitates the provision of information about the structure of social support (Lunsky and Benson, 1999). Participants were invited to write the names of individuals who were in their life during pregnancy and childbirth inside a series of concentric circles based on perceptions of closeness to these individuals. Since the experience of social support may change from pregnancy to pregnancy, we invited women to discuss each of their pregnancies within the last five years. Addressing acquiescence bias Although semi-structured interviews have been used successfully with individuals with IDD, they are prone to acquiescent response patterns, especially when respondents are unsure about certain facts (Beail, 2002). To minimise this issue, the interviewer adhered to established guidelines for interviewing individuals with IDD (Finlay and Lyons, 2001; Tassé et al., 2005; D’Eath, 2005) (e.g., emphasised that there were no right or wrong answers, encouraged participants to ask for clarification of questions during the interview as needed, and followed most Yes/No questions with open-ended questions to elicit explanations for responses). The interviewer also employed journaling immediately following each interview, which provided an opportunity to reflect on the interview process and to identify possible biases. Interviewer characteristics The interviewer (first author) was an undergraduate Psychology student and research coordinator who was supervised and trained by a Clinical Psychologist whose research expertise is in IDD (third author). The interviewer had experience conducting telephone interviews with adults with IDD and is a birth doula. Data analysis The first author used memo writing and constant comparison throughout data collection and analysis to track and build theoretical categories (Lempert, 2007). Following data collection, audio-taped interviews were transcribed verbatim. Qualitative data analyses for responses to open-ended questions were carried out using NVivo 10. First, text segments from interviews were coded line by line. Initial coding was considered tentative and was modified throughout analysis (Holloway and Wheeler, 2010). To strengthen the reliability and validity of the coding structure and to minimise researcher bias, the second and third authors audited the coding process twice during analysis. Following discussion of initially developed codes, emerging codes were used to create groups of concepts marked by similar traits (open-coding). These groups of concepts were then reassembled into theoretical categories, and patterns underlying their relationships were further explored to generate a preliminary conceptual framework (axial
59
coding) (Holloway and Wheeler, 2010). Direct quotes from participants, who were assigned pseudonyms, were used to illustrate theoretical categories. To maintain the voice of individual participants, we did not translate French quotes to English but provide an interpretation of those quotes in the text.
Findings Participant characteristics Four women participated in the study. Two of them had more than one pregnancy within the last five years, for a total of seven eligible pregnancies. Women's age at the time of the interview ranged from 23 to 32 years. Two women resided in an urban area, one in a semi-rural area, and one in a rural area. Three women chose to be interviewed at the service agency involved in recruitment; the fourth was interviewed at home. All women requested that their caseworker be present during the interview. Two of the interviews were conducted in French, and two were conducted in English. On average, each interview took 1.5 hours to complete. Structure of social support The size of women's social support networks during pregnancy ranged from 7 to 19 individuals. Formal support was provided by paid professionals (i.e., doctors, nurses, caseworkers). Informal support was provided mostly by family members (i.e., parents, siblings, children, extended family) and by the partner and his family. Very little informal support was provided by individuals outside the family. All women reported having few or no friendships; individuals referred to as friends were women who were pregnant or had children. Functions of social support Informational support was primarily provided by formal sources. Doctors provided information during antenatal visits, while caseworkers explained complex medical advice provided by doctors. Caseworkers also served as bridges to other sources of informational support (e.g., antenatal and parenting classes, dieticians): Catherine: “She's [caseworker] the one that […] like told me about the health unit, told me about the parenting courses, told me about all the stuff that I never knew, but that I needed. I would have never knew or found out about it until she told me.” Informational support was also provided by other pregnant women and mothers. The cousin of Catherine's partner helped her prepare for motherhood by teaching her how to bathe and hold a newborn: Catherine: “If they had kids, I asked questions to people who had the kids. I don’t know, I guess cause well […] they had kids, they know what they’re talking about […] I guess that's what I did, like, if somebody I knew had kids that's who I’ll be asking.” Instrumental support was similarly provided by formal and informal sources. However, types of instrumental support provided depended on the source. Doctors provided medical care during pregnancy and childbirth, whereas nurses mostly provided medical care during childbirth. Caseworkers, family members, and the partner's family similarly provided tangible help (e.g., preparing
60
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
the infant's room, acquiring items for the infant). Transportation was mainly provided by the caseworker and the partner's family. Alexandra received significant support from her caseworker who accompanied her to all of her appointments and helped her plan the infant's room, including the room colour and crib: Alexandra:
expectant and new mothers; she lost touch with that friend after childbirth. Friendships were also created with other mothers. Catherine described accompanying her oldest child to a daycare where she received companionship from a mother whose child also attended the programme: Catherine:
«C’est elle qui m’accompagnait tout le temps, [à]mes rendezvous… Je veux dire c’est elle qui, quand j’avais besoin, you know, préparer la chambre […] C’est elle qui m’a aidé à planifier la couleur de la chambre […] la couchette […] je veux dire elle a fait sa part avec moi.»
“We went to daycare and that, like for my little one, ‘cause they told me before she started school she’d have to get used to be around other kids cause she was the only one really. So we brought her to the daycare here in town and she played with the kids and that […] There was a girl named T* […] And she has a daughter named D*.”
Catherine's mother helped with the infant shower, and her partner's family purchased infant furniture: Catherine: “And my mom [helped out] all the time, like if I needed anything for my baby shower and everything […] His [partner's] mother went out and bought us like the crib, the change table, the dresser, all that stuff.” Emotional support was lacking for most participants. Camille, Alexandra, and Rose had few people to talk to during pregnancy. When Rose found out that she was pregnant with her first infant, she did not have anyone to talk to about her fears: Rose: “I didn’t know what to do, you know, I was scared. Like am I going to be able to do it on my own, you know? I didn’t have nobody […] I did not have a good relationship with my parents at the time either. So it was hard, but…” When Camille was asked if she had anyone to care for her when she felt sick during her pregnancy or give her a hug when she felt sad, she replied that she did not; she did it all alone: Camille: «Je n’avais pas de ça. Je n’avais pas de ça moi […] Je le faisais toute seule. » Where emotional support was available, it was provided mostly by informal sources, such as family members. Rose and Camille considered their older children as their primary sources of emotional support in subsequent pregnancies; they brought comfort during difficult times. Alexandra stated that she did not have many friends or anyone to confide in during her pregnancy, with the exception of her parents: Alexandra: «Moi pour te le dire, tu as touché un point aussi que moi, je n’ai pas des amis beaucoup là […] Non, à qui me confier, à qui demander… umm je veux dire non.[…]…heureusement j’avais mes parents à qui me… leur dire comment je me sentais.» To a lesser extent, formal sources also provided emotional support, particularly caseworkers (during pregnancy) and nurses (during childbirth): Catherine: “But, mainly it was a nurse […] but she was there the whole time and she was awesome.[…] I never saw a nurse that was that good before […] Yeah, she was making us all work together to have the baby.” Social companionship was reported by few women. The limited social companionship that was provided came from partners, children, pregnant friends, and other women who have children. In general, friendships created during pregnancy with other pregnant women tended to be restricted to that period. Camille talked about making a friend while attending a programme for
Sometimes, social companionship was provided by formal sources, primarily the caseworker. While Alexandra had no friend with whom to engage in leisurely activities, she did receive social companionship from her caseworker; she talked about going for coffee and doing physical exercises with her: Alexandra: «Je fais mes exercices […] une fois de temps en temps je le fais avec [personnel de soutien], je vais avec elle pour un café, ou discuter un petit peu.» Relationships with caseworkers were more stable and longterm than friendships with pregnant women. Perceived quality of social support Three factors related to women's perceived quality of available social support emerged from the data: the accessibility of the desired structure and functions of social support; the attitudes of support persons; and autonomy in determining social support. Accessibility related to distance and transportation to sources of support, as well as the clarity of presented information about pregnancy and childbirth. All the women's families lived in different cities, but many of the women resided in proximity to their partners’ families. Consequently, participants often reached out to whomever was easiest to access. Catherine primarily received support from her partner's family during pregnancy but would have preferred to be supported by her own family: Catherine: “I just mainly went to whoever's closer and I felt comfortable with […] they’re [partner's family] right in this town, so they at least come visit at least once a week or every day, so yes I am closer to them because they’re always there, but if I was to go to anybody I would go to my family […] but they’re not there, so I have to go with what I got.” Alexandra's parents lived in another country. Despite her strained relationship with her mother-in-law, she nonetheless reached out to her in-laws for support because they were easily accessible: Alexandra: «Quand j’avais besoin d’aide, j’appelais au téléphone la personne qui était libre, elle se présentait. Soit c’était mon beaupère, soit c’était la belle-mère, soit c’est les deux… » These distance and transportation issues also related to the accessibility of formal supports. Camille was encouraged by her caseworker to attend antenatal classes during her second pregnancy. However, she reported not attending due to a lack of transportation and of a support person to accompany her. There were also issues related to the understandability of information. Alexandra only attended two antenatal classes because the classes were not available in her first language (French).
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
Although Rose attended antenatal classes during her first pregnancy, she preferred to have a nurse explain the information to her one-on-one and to acquire information about pregnancy and childbirth herself through the Internet: Rose: “They were talking to people as a group. The group, the [prenatal] class […] I’m better to understand one-on-one. But the stuff I brought back, the nurse was able to explain it to me when she came.” Clarity of information was also an issue at antenatal visits. Alexandra reported receiving test requisition papers and pamphlets from her doctor which she had difficulty understanding until she met with her caseworker to ask questions. Attitudes related to women's perceptions of their support persons’ opinions and feelings about the pregnancy. Support received was perceived as unhelpful and undesirable if it was provided by individuals expressing negative attitudes. Rose perceived support from her doctor unfavourably due to his attitudes: Rose: “One thing that I didn’t like hearing [from her doctor] was that I was young. I know I’m young. I just need your help to get through it now, ya know? I didn’t plan to have a baby although I wasn’t taking precautions. I did not plan to have a baby at that age […] So I didn’t need people's negative thoughts and stuff.” Alexandra mentioned her mother-in-law and sister-in-law's negativity towards her pregnancy several times during the interview. They told her that getting pregnant was foolish, and that she “wouldn’t be able to” parent. Although no one openly told her that she should terminate her pregnancy, these negative comments contributed to fears that someone would broach the subject with her: Alexandra: « Mais j’avais peur d’entendre l’un ou l’autre me dire ‘va l’avorter’ […] Parce que j’ai aimé ce petit… la minute où j’ai entendu, j’ai senti que j’étais enceinte, c’était comme ‘no way là!’. Je ne veux pas que quelqu’un me le dit, non… » However, some support persons also expressed positive attitudes at the announcement of pregnancy. Most partners and some family members welcomed the pregnancies: Catherine: “Everybody was happy! […] They [in-laws] were really, really, really happy. To have the first grandkids and that.” Moreover, caseworkers were an important source of encouragement during pregnancy. Alexandra felt encouraged and supported by her caseworker. Alexandra: « Elle m’encourageait, elle me supportait… » Autonomy related to the perception that individual support needs and self-determination were respected. Most of the women reported instances when their preferences were not valued. Sometimes this lack of autonomy related to the women's or their partner's desire for independence. Alexandra became frustrated when her wish to go to her medical appointment unaccompanied was not respected. She explained that she and her caseworker had a disagreement over the issue; Alexandra wanted to prove that she was capable of seeing her doctor by herself after 7 months of going to appointments together: Alexandra: «Il y avait un petit, comment je peux dire… [un] malentendu
61
là? Dans le sens que je voulais lui [personnel de soutien] prouver que je suis capable d’aller toute seule [chez le médecin] […]Je veux dire, ça fait quoi, 7 mois que tu me suis là, je suis capable de le faire toute seule! Elle était, comment on dit, fâchée contre moi. Elle me dit ‘Non tu devrais m’attendre, on avait une entente’. ‘Come on, je suis capable!’» Catherine did not perceive support provided by her partner's aunt positively because she imposed her help even though Catherine did not request it: Catherine: “Like somebody is trying to help you or whatever[…] there's okay I need your help, but it's okay I don’t need your help. She's just trying, and trying, kind of thing […] Sometimes it's nice, but sometimes it was annoying […]” Conversely, although Camille wanted her caseworker's support during her pregnancy, her partner refused this service; he wanted the couple to be autonomous: Camille: « Parce que je l’aimais beaucoup quand elle était là pour [la première grossesse], pis je voulais qu’elle soit là, mais [il] était un gars qui voulait être autonome […] » Sometimes this lack of autonomy related to the women's desire to receive a certain type of support from a particular person but receiving it from someone else. Although Catherine received support from several individuals during childbirth, she would have preferred to have been supported by her sister. During labour, there were too many family members involved, leading to preoccupations with others’ feelings: Catherine: “So my sister didn’t end up coming in the room, but I really wanted her to. So the only one that was in there was like my mom and his mom […] The only one that I really wanted in my room [was] my sister […] and my mom, but my mom didn’t have to be like […] as long as I had my sister there I was comfortable.”
Discussion Summary of findings During pregnancy, women in this study received social support from both formal and informal sources. However, results highlight the central role of formal support, primarily provided by the caseworker. Although pregnant women with IDD created shortterm friendships with other pregnant women, they generally lacked informal relationships outside the family structure. Moreover, although high levels of informational and instrumental support and low levels of emotional support and social companionship were reported, high levels of available support were not always perceived as beneficial. On the basis of these findings, we developed a preliminary conceptual framework of effective social support during pregnancy and childbirth, as perceived by women with IDD. Fig. 1 illustrates the emergent core categories which suggest that social support is perceived as most effective when desired support is accessible, support is provided by individuals expressing positive attitudes towards the pregnancy, and autonomy is valued (e.g., individual support needs and self-determination are respected).
62
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
Availability Support received Desired Support
Functions of Support
Structure of Support
Accessibility, Attitudes & Autonomy (+)or (-) attitudesof support persons
Value of autonomy
Perception of support received
Perceived Quality of Support
Fig. 1. A preliminary conceptual framework of effective social support during pregnancy and childbirth as perceived by women with intellectual and developmental disabilities.
Consistency with previous research and potential explanations Our finding of impoverished informal support outside of the families of women with IDD is consistent with previous studies demonstrating that adults with IDD generally have restricted social networks, with few or no friendships, which mainly comprise family members and paid caregivers (Lunsky and Benson, 1999). Although few friendships were reported, all women in our study created short-term friendships with pregnant women and mothers. One possible explanation is that the shared experience of pregnancy, or motherhood, may have facilitated these relationships. We found that women with IDD generally received little emotional support and social companionship as they prepared for motherhood. This is consistent with previous findings of social isolation among pregnant women with IDD (Llewellyn and McConnell, 2002). This finding may be related to negative reactions towards childbearing in this population (McConnell and Llewellyn, 2002; Mayes et al., 2006; McConnell et al., 2008b; Walsh-Gallagher et al., 2012; Höglund and Larsson, 2013; WalshGallagher et al., 2013). Similarly to previous research (Lunsky and Benson, 1999), our study illustrates the centrality of the caseworker-client relationship in the provision of social companionship amongst adults with IDD. Women in our study received support from their caseworkers for an extended period of time prior to pregnancy and therefore had an ongoing relationship with them. However, research has also demonstrated that paid carers often view themselves as facilitators to their clients’ friendships rather than social companions themselves (Pockney, 2006). The accessibility issues reported in our study are consistent with previous findings. As in our study, lack of transportation is commonly cited as a barrier to social inclusion and support for people with IDD (Abbott and McConkey, 2006). Moreover, our finding that information about pregnancy and childbirth was not always clear for women with IDD parallels previous research suggesting that adapted antenatal informational support for women with IDD is lacking (McConnell et al., 2008a; Walsh-Gallagher et al., 2012; Redshaw et al., 2013). This finding is important as research has demonstrated that women with IDD often view childbearing as “overwhelming and difficult to understand” (Höglund and Larsson, 2013, p.704). Negative attitudes toward pregnant women with IDD found in our study are consistent with a history of opposition to women
with IDD becoming mothers (Booth and Booth, 1995; Aunos and Feldman, 2002; Mayes et al., 2006; McConnell et al., 2008b). Pregnancy in this population is often treated as a mistake, and expectant women with IDD often feel pressured to terminate their pregnancies (Booth and Booth, 1995; Mayes et al., 2006). Negative attitudes may have contributed to social isolation during pregnancy: pregnant women with IDD tend to exclude unsupportive individuals from their networks (Mayes et al., 2006). Conversely, women in this study also reported that some support persons expressed positive attitudes towards their pregnancy; this seemed to facilitate access to support and to increase satisfaction with support. Others’ attitudes were also linked to autonomy and service uptake. Our finding that some women with IDD and their partners struggled for autonomy during pregnancy is consistent with previous literature demonstrating that decision-making opportunities are often restricted in this population (Höglund and Larsson, 2013): the ability of women with IDD to parent is commonly scrutinised (McConnell and Llewellyn, 2002; Mayes et al., 2006; Greenwood and Wilkinson, 2013), they are afforded less control over their lives (Mayes et al., 2006), and are brought under “constant surveillance” (Booth and Booth, 1995, p. 31). Moreover, women with IDD may react with hostility and resistance to professionals who do not validate their role as mothers, and respect their needs (Wilson et al., 2013). This may explain why some participants felt a need to prove themselves as “able” and refused some services. Limitations Several issues may limit the transferability of our findings. While convenience sampling is an appropriate sampling strategy for hard-to-reach study populations (Morse, 2007), our sample excluded women who were not currently receiving services for adults with IDD. Moreover, participants had to possess sufficient recall and verbal capacity to engage in an interview, thus excluding women who were non-verbal or who had a lower level of functioning. We did not have specific information pertaining to diagnosis or level of disability; however, all of the women currently had custody of their children and lived independently in the community. Results therefore may not reflect the experiences of women who did not maintain custody and who resided with caregivers. The sample size (n¼ 4) is also an important limitation.
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
Although providing rich preliminary data, we cannot claim to have reached theoretical saturation. Therefore, we must emphasise the preliminary nature of our conceptual model; other equally significant concepts could have emerged from the data with the addition of other participants. Response biases may also limit results. Although measures to circumvent acquiescent responses were taken, persons with IDD are prone to respond in such patterns (Heal and Sigelman, 1995). Additionally, although individuals with IDD have the ability to accurately recall lived experiences (Beail, 2002), semi-structured interviews are vulnerable to recall bias. Every woman in this study requested to have her caseworker present during the interview, potentially biasing responses to questions about formal support. However, the centrality of the caseworker-client relationship was also reflected in this choice. Implications Our findings suggest that improvements in the accessibility of social support for women with IDD are warranted. Given that women with IDD struggled with complex medical information provided by doctors and in antenatal classes, maternity care providers such as doctors, midwives, and nurse practitioners should consider longer antenatal appointments to allow sufficient time to effectively communicate information to pregnant women with IDD (McConnell et al., 2008a). Midwives, doulas, and nurses, who tend to offer longer appointments than doctors, may have a valuable role in providing continuous support and maternity care to women with IDD. Previous studies have shown the value of midwifery support, particularly in situations where informal support is low (Wilson et al., 2013). Women with IDD consider doulas to be reliable sources of pregnancy-related information (McGarry et al., 2015). Moreover, doulas provide important social and emotional support during labour (Lantz et al., 2005). Maternity care providers should also consider caseworkers (or community-based support agency staff) as key resources; they serve as bridges to other sources of support and assist women with IDD with understanding information and complying with healthcare providers’ recommendations. Furthermore, Hodnett et al., 2013 suggest that involving a chosen member of the family or friend appears to increase satisfaction with childbirth. Informal support therefore, should also be facilitated. Our study demonstrates that stereotypes and negative attitudes still exist towards childbearing in this population. All pregnant women with IDD have the right to respectful and nonjudgmental support. Therefore, healthcare practitioners should be provided with appropriate knowledge and resources regarding pregnancy in women with disabilities in order to minimise negative or stigmatizing attitudes. As autonomy and decision-making opportunities were valued by women with IDD during pregnancy and childbirth, respecting individual support needs and adopting a collaborative rather than directive support approach are encouraged.
Conclusion Interviewing mothers with IDD allowed us to propose a preliminary conceptual framework of effective social support during pregnancy and childbirth as perceived by women with IDD. Although increasing support availability for women with IDD during pregnancy and childbirth should be a social and clinical priority, our results suggest that social support is perceived as most effective when it is perceived as accessible, it is received from individuals expressing positive attitudes, and autonomy is valued.
63
Conflict of interest We have no conflicts of interest to declare.
Acknowledgements We are grateful to the volunteer research assistants – Barbara Mackenzie Barnett, Préscillia Dupont and Charlotte Wagner Searle – who transcribed the audiotaped interviews.
References Abbott, S., McConkey, R., 2006. The barriers to social inclusion as perceived by people with intellectual disabilities. Journal of Intellectual Disabilities 10, 275–287. American Psychiatric Association, 2013. Diagnostic and Statistical Manual of Mental Disorders, 5th edn DC: American Psychiatric Publishing, Washington. Arscott, K., Dagnan, D., Kroese, B.S., 1998. Consent to psychological research by people with an intellectual disability. Journal of Applied Research in Intellectual Disabilities 11, 77–83. Aunos, M., Feldman, M.A., 2002. Attitudes towards sexuality, sterilization and parenting rights of persons with intellectual disabilities. Journal of Applied Research in Intellectual Disabilities 15, 285–296. Brown, H.K., Lunsky, Y., Cobigo, V., Wilton, D., Vigod, S., 2016. Pregnancy in women with intellectual and developmental disabilities. Journal of Obstetrics and Gynaecology Canada 38, 9–16. Beail, N., 2002. Interrogative suggestibility, memory and intellectual disability. Journal of Applied Research in Intellectual Disabilities 15, 129–137. Blackford, K.A., 1993. Erasing mothers with disabilities through Canadian familyrelated policy. Disability, Handicap and Society 8, 281–294. Booth, T., Booth, W., 1995. Unto us a child is born: the trials and rewards of parenthood for people with learning difficulties. Australia and New Zealand Journal of Developmental Disabilities 20, 25–39. Bramston, P., Fogarty, G., Cummins, R.A., 1999. The nature of stressors reported by people with an intellectual disability. Journal of Applied Research in Intellectual Disabilities 12, 1–10. Cohen, S., Wills, T.A., 1985. Stress, social support, and the buffering hypothesis. Psychological Bulletin 98 (2), 310–357. Cohen S., Syme S.L., 1985. Issues in the Study and Application of Social Support. Social Support and Health. Academic Press, San Francisco, pp. 3–22. Corbin, J., Morse, J.M., 2003. The unstructured interactive interview: issues of reciprocity and risks when dealing with sensitive topics. Qualitative Inquiry 9, 335–354. D’Eath, M., 2005. Guidelines for Researchers when Interviewing People with an Intellectual Disability. National Federation of Voluntary Bodies, Galway. Finlay, W.M.L., Lyons, E., 2001. Methodological issues in interviewing and using self-report questionnaires with people with mental retardation. Psychological Assessment 13, 319–335. Geller, P.A., 2004. Pregnancy as a stressful life event. CNS Spectrums 9, 188–197. Glazier, R.H., Elgar, F.J., Goel, V., Holzapfel, S., 2004. Stress, social support, and emotional distress in a community sample of pregnant women. Journal of Psychosomatic Obstetrics and Gynaecology 25, 247–255. Greenwood, N.W., Wilkinson, J., 2013. Sexual and reproductive health care for women with intellectual disabilities: a primary care perspective. International Journal of Family Medicine 2013, 642472. Höglund, B., 2012. Pregnancy, Childbirth and Midwifery Care Among Women with Intellectual Disability in Sweden: Epidemiological and Descriptive Studies (Unpublished doctoral dissertation). Uppsala University, Sweden. Höglund, B., Larsson, M., 2013. Struggling for motherhood with an intellectual disability – A qualitative study of women's experiences in Sweden. Midwifery 29, 698–704. Hartley, S.L., MacLean, J., W.E., 2006. A review of the reliability and validity of Likert-type scales for people with intellectual disability. Journal of Intellectual Disability Research 50, 813–827. http://dx.doi.org/10.1111/ j.1365-2788.2006.00844.x. Heal, L.W., Sigelman, C.K., 1995. Response biases in interviews of individuals with limited mental ability. Journal of Intellectual Disability Research 39, 331–340. Heaman, M., 1998. Psychosocial impact of high-risk pregnancy: hospital and home care. Clinical Obstetrics and Gynecology 41, 626–639. Hodnett, E.D., Gates, S., Hofmeyr, G.J., and Sakala, C., 2013. Continuous support for women during childbirth (Cochrane Review). The Cochrane Library, Issue 7. Retrieved from 〈http://onlinelibrary.wiley.com/doi/10.1002/14651858. CD003766.pub5/epdf〉. Holloway, I., Wheeler, S., 2010. Qualitative Research in Nursing and Health Care, 3rd edn Wiley-Blackwell Publishing, Oxford. Holmes, T., Rahe, R., 1967. The social readjustment rating scale. Journal of Psychosomatic Research 11, 213–218. Holton, J.A., 2007. The coding process and its challenges. In: Bryant, A., Charmaz, K. (Eds.), The SAGE Handbook of Grounded Theory. SAGE Publications Ltd.,
64
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
London, pp. 265–290. http://dx.doi.org/10.4135/9781848607941.n13. Kempton, W., Kahn, E., 1991. Sexuality and people with intellectual disabilities: a historical perspective. Sexuality and Disability 9, 93–111. Lantz, P.M., Low, L.K., Varkey, S., Watson, R.L., 2005. Doulas as childbirth paraprofessionals: results from a national survey. Women's Health Issues 15, 109–116. Lempert, L.B., 2007. Asking questions of the data: memo writing in the grounded theory tradition. In: Bryant, A., Charmaz, K. (Eds.), The SAGE Handbook of Grounded Theory. SAGE Publications Ltd., London, pp. 245–265. Lin, E., Balogh, R., Cobigo, V., Ouellette-Kuntz, H., Wilton, A.S., Lunsky, Y., 2013. Using administrative health data to identify individuals with intellectual and developmental disabilities: a comparison of algorithms. Journal of Intellectual Disability Research 57, 462–477. http://dx.doi.org/10.1111/jir.12002. Llewellyn, G., McConnell, D., 2002. Mothers with learning difficulties and their support networks. Journal of Intellectual Disability Research 46, 17–34. Lunsky, Y., Benson, B.A., 1997. Reliability of ratings of consumers with mental retardation and their staff on multiple measures of social support. American Journal on Mental Retardation 102, 280–284. Lunsky, Y., Benson, B.A., 1999. Social circles of adults with mental retardation as viewed by their caregivers. Journal of Developmental and Physical Disabilities 11, 115–129. Lunsky, Y., Benson, B.A., 2001. Association between perceived social support and strain, and positive and negative outcome for adults with mild intellectual disability. Journal of Intellectual Disability Research 45, 106–114. Lyons, K.S., Zarit, S.H., 1999. Formal and informal support: the great divide. International Journal of Geriatric Psychiatry 14, 183–196. Mayes, R., Llewellyn, G., McConnell, D., 2006. Misconception: the experience of pregnancy for women with intellectual disabilities. Scandinavian Journal of Disability Research 8, 120–131. http://dx.doi.org/10.1080/15017410600774178. McConnell, D., Llewellyn, G., 2002. Stereotypes, parents with intellectual disability and child protection. Journal of Social Welfare and Family Law 24, 297–317. http://dx.doi.org/10.1080/09649060210161294. McConnell, D., Mayes, R., Llewellyn, G., 2008a. Women with intellectual disability at risk of adverse pregnancy and birth outcomes. Journal of Intellectual Disability Research 52, 529–535. http://dx.doi.org/10.1111/j.1365-2788.2008.01061.x. McConnell, D., Mayes, R., Llewellyn, G., 2008b. Pre-partum distress in women with intellectual disabilities. Journal of Intellectual and Developmental Disability 33 (2), 177–183. McGarry, A., Stenfert Kroese, B., Cox, R., 2015. How do women with an intellectual disability experience the support of a doula during their pregnancy, childbirth and after the birth of their child? Journal of Applied Research in Intellectual Disabilities . http://dx.doi.org/10.1111/jar.12155 [Epub ahead of print]. Mitra, M., Parish, S.L., Clements, K.M., Cui, X., Diop, H., 2015. Pregnancy outcomes among women with intellectual and developmental disabilities. American Journal of Preventive Medicine 48, 300–308.
Morikawa, M., Okada, T., Ando, M., Aleksic, B., Kunimoto, S., Nakamura, Y., Ozaki, N., 2015. Relationship between social support during pregnancy and postpartum depressive state: a prospective cohort study. Scientific Reports 5, Retrieved from 〈〈http://www.nature.com/articles/srep10520〉〉. Morse, J.M., 2007. Sampling in grounded theory. In: Bryant, A., Charmaz, K. (Eds.), The SAGE Handbook of Grounded Theory. SAGE Publications Ltd., London, pp. 229–245. http://dx.doi.org/10.4135/9781848607941.n11. Oakley, A., 1988. Is social support good for the health of mothers and babies? Journal of Reproductive and Infant Psychology 6, 3–21. Pockney, R., 2006. Friendship or facilitation: people with learning disabilities and their paid carers. Sociological Research Online 11, Retrieved from 〈〈http://www. socresonline.org.uk/11/3/pockney.html〉〉. Redshaw, M., Malouf, R., Gao, H., Gray, R., 2013. Women with disability: the experience of maternity care during pregnancy, labour and birth and the postnatal period. BMC Pregnancy Childbirth 13, 174, Retrieved from 〈〈http://www. biomedcentral.com/1471-2393/13/174〉〉. Robson, C., 2002. Real World Research. A Resource for Social Scientists and Practitioner-Researchers, 2nd edn Wiley, Oxford. Royal College of Midwives, 2000. Position Paper 11a. Maternity Care for Women with Disabilities. Royal College of Midwives, London. Servais, L., Jacques, D., Leach, R., Conod, L., Hoyois, P., Dan, B., Roussaux, J.P., 2002. Contraception of women with intellectual disability: prevalence and determinants. Journal of Intellectual Disability Research 46, 108–119. Sherbourne, C.D., Stewart, A.L., 1991. The MOS social support survey. Social Science and Medicine 32, 705–714. Tassé, M.J., Schalock, R., Thompson, J.R., and Wehmeyer, M., 2005. Guidelines for interviewing people with disabilities: Supports intensity scale. American Association on Intellectual and Developmental Disabilities. Washington, DC. United Nations, 2006. Convention on the Rights of Persons with Disabilities (CRPD). United Nations, New York, NY, Retrieved from 〈http://www.un.org/disabilities/ convention/conventionfull.shtml〉. Walsh-Gallagher, D., Sinclair, M., Mc Conkey, R., 2012. The ambiguity of disabled women's experiences of pregnancy, childbirth and motherhood: a phenomenological understanding. Midwifery 28, 156–162. Walsh-Gallagher, D., Mc Conkey, R., Sinclair, M., Clarke, R., 2013. Normalising birth for women with a disability: the challenges facing practitioners. Midwifery 29, 294–299. Willis, T.A., Shinar, O., 2000. Measuring perceived and received social support. In: Cohen, S., Underwood, L.G., Gottlieb, B.H. (Eds.), Social Support Measurement and Intervention: A Guide for Health and Social Scientists. Oxford University Press, New York, pp. 86–135. Wilson, S., McKenzie, K., Quayle, E., Murray, G.C., 2013. The postnatal support needs of mothers with an intellectual disability. Midwifery 29, 592–598. Wingfield, M., Healy, D.L., Nicholson, A., 1994. Gynaecological care for women with intellectual disability. The Medical Journal of Australia 160, 536–538.
Contents lists available at ScienceDirect
Midwifery journal homepage: www.elsevier.com/midw
Social support received by women with intellectual and developmental disabilities during pregnancy and childbirth: An exploratory qualitative study Lynne A. Potvin, H.B.A (Specialization in Psychology and Birth Doula)a, Hilary K. Brown, PhD (Postdoctoral Fellow)b, Virginie Cobigo, PhD (Associate Professor)c,n a b c
University of Ottawa, School of Psychology, 136 Jean Jacques Lussier (5078), Ottawa, Canada ON K1N 6N5 Women's College Research Institute, University of Toronto, Department of Psychiatry, 76 Grenville Street, Toronto, Canada ON M5S 1B2 University of Ottawa, School of Psychology, 136 Jean Jacques Lussier (5083), Ottawa, Canada ON K1N 6N5
art ic l e i nf o
a b s t r a c t
Article history: Received 18 November 2015 Received in revised form 25 March 2016 Accepted 10 April 2016
Objective: this study aims to contribute to the development of a conceptual framework that will inform maternity care improvements for expectant mothers with intellectual and developmental disabilities (IDD) by exploring the structure, functions, and perceived quality of social support received by women with IDD during pregnancy and childbirth. Design/setting: using a grounded theory approach, we conducted an exploratory study set in Ontario, Canada in 2015. Participants: the sample included four adult women with IDD who had given birth in the last five years. Measurements: data were collected using semi-structured interviews. Findings: the structure of social support received by women with IDD consisted of both formal and informal sources, but few or no friendships. Women with IDD reported high levels of informational and instrumental support and low levels of emotional support and social companionship. However, a high level of available support was not always perceived as beneficial. Emergent core categories suggest that social support is perceived as most effective when three conditions are met: (1) support is accessible, (2) support is provided by individuals expressing positive attitudes towards the pregnancy, and (3) autonomy is valued. Key conclusions and implications for practice: our study confirms and identifies important gaps in the social support received by expectant mothers with IDD. Women with IDD currently lack accessible informational support, emotional support, and social companionship during pregnancy and childbirth. Additional findings regarding the structure and functions of social support are presented, and a preliminary conceptual framework of effective social support during pregnancy and childbirth, as perceived by women with IDD is also proposed. Findings suggest that increasing support accessibility should be a social and clinical priority; however, maternity care providers should be aware of stigmatizing attitudes and respect the autonomy of pregnant women with IDD as they prepare for motherhood. & 2016 Elsevier Ltd. All rights reserved.
Keywords: Intellectual disability Developmental disability Pregnancy Childbirth Social support Grounded theory
Introduction Intellectual and developmental disabilities (IDD) are neurodevelopmental disorders characterised by limitations in cognitive and adaptive functioning (American Psychiatric Association, 2013). According to health administrative data from Ontario, Canada, the prevalence rate of IDD per 100 is 0.80 (Lin et al., 2013). Historically, n
Corresponding author. E-mail addresses: [email protected] (L.A. Potvin), [email protected] (H.K. Brown), [email protected] (V. Cobigo). http://dx.doi.org/10.1016/j.midw.2016.04.005 0266-6138/& 2016 Elsevier Ltd. All rights reserved.
many women with IDD were involuntary sterilised and institutionalized (Kempton and Kahn, 1991). Today, legislation protects the reproductive rights of women with IDD in most developed nations, including Canada (Blackford, 1993), and the United Nations Convention on the Rights of Persons with Disabilities (2006) reaffirms the right of persons with IDD to “marriage, family, parenthood and relationships, on an equal basis with others” (article 23.1). This changing context has significant implications for childbearing in this population. Many women with IDD have sexual relationships and desire children (Aunos and Feldman, 2002). Moreover, an increasing
58
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
number of women with IDD are using maternity services (Royal College of Midwives, 2000). The general fertility rate in women with IDD is 20.3 live births per 1000 women with IDD (Brown et al., 2016); pregnancy is therefore not uncommon in this population. Women with IDD have high rates of adverse perinatal outcomes, including preeclampsia, caesarean section, preterm birth, and perinatal mortality (McConnell et al., 2008a; Höglund, 2012; Mitra et al., 2015), suggesting that they are a high-risk maternity population whose individual needs should be supported. Social support can be conceptualised in terms of structure and function. Structure refers to the number of relationships one has and the degree of interconnectedness between these relationships (Sherbourne and Stewart, 1991). Relationships may be formal (e.g., paid sources such as healthcare professionals) or informal (e.g., unpaid sources such as family members and friends) (Lyons and Zarit, 1999). Function refers to the nature of social support provided and the degree to which the relationship serves a purpose (Sherbourne and Stewart, 1991). Commonly cited functions of social support are informational support, instrumental support, emotional support, and social companionship (Sherbourne and Stewart, 1991; Willis and Shinar, 2000). Informational support includes the provision of knowledge, advice, and guidance. Instrumental support includes practical support such as transportation, financial aid, and help with household chores. Emotional support is defined by demonstrations of caring and acceptance like sympathetic listening. Social companionship refers to relationships that engage in social activities (Willis and Shinar, 2000). Perceived quality of social support refers to the subjective evaluation of support received by an individual, which may be positive or negative (Cohen and Syme, 1985; Bramston et al., 1999; Lunsky and Benson, 2001). Research has identified pregnancy as a potentially stressful life event requiring social readjustment (Holmes and Rahe, 1967); however, it is especially stressful if it is unplanned (Geller, 2004) or high risk (Heaman, 1998). Women with IDD appear to be vulnerable to unwanted pregnancy (Wingfield et al., 1994; Servais et al., 2002) and represent a high-risk maternity population (McConnell et al., 2008a; Höglund, 2012; Mitra et al., 2015). Conversely, social support serves a protective function against the negative effects of stress (Cohen and Syme, 1985; Cohen and Wills, 1985) and contributes to the health and well-being of expectant mothers (Oakley, 1988). Effective and continuous social support, in general, has been associated with a reduced risk of emotional distress during pregnancy (Glazier et al., 2004), shorter labour, fewer obstetrical interventions (Lantz et al., 2005), and a lower risk for post partum depression (Morikawa et al., 2015). Moreover, research specific to women with IDD has demonstrated that adapted informational support increases self-efficacy in preparation for motherhood and confidence in making informed choices during pregnancy (McGarry et al., 2015). Most adults with IDD experience low to moderate levels of social support (Lunsky and Benson, 1999). However, only a handful of studies, from the United Kingdom, Ireland, Sweden, and Australia, have described social support received by women with IDD during pregnancy and childbirth. Themes that have emerged from these studies include inadequately addressed needs for informational and practical support (Höglund and Larsson, 2013; Mayes et al., 2006; Redshaw et al., 2013; Walsh-Gallagher et al., 2012), feelings of loneliness (Walsh-Gallagher et al., 2012; Walsh-Gallagher et al., 2013) and social isolation (Llewellyn and McConnell, 2002), and distress over prejudicial beliefs held by family members and health care professionals towards the pregnancy (McConnell and Llewellyn, 2002; Mayes et al., 2006; McConnell et al., 2008b; Walsh-Gallagher et al., 2012; Höglund and Larsson, 2013; Walsh-Gallagher et al., 2013).
As such, this study aims to contribute to the development of a conceptual framework that will inform maternity care improvements for expectant mothers with IDD by exploring the structure, functions, and perceived quality of social support received by women with IDD during pregnancy and childbirth.
Method Ethics approval was obtained from the Office of Research Ethics and Integrity at the University of “X”. Design/setting We conducted a qualitative study using a grounded theory approach. Using pre-existing knowledge to understand the data, grounded theory uses constant comparison to identify and compare emerging conceptual elements, ask questions of the data, and code data into categories based on their theoretical properties (Robson, 2002; Holton, 2007). On the basis of this in-depth analysis, it is possible to generate explanatory models that are grounded in the data (Robson, 2002). The study was conducted in Ontario, Canada, in 2015. Participants Participants were recruited using convenience sampling from local French and English service agencies for adults with IDD that agreed to forward information packages to potential participants. The sample included women who had a diagnosis of IDD (as confirmed by the service agency) and who: (1) were 18 years of age or older, (2) had a pregnancy that resulted in a live birth in the last five years, and (3) possessed sufficient verbal capacity to answer questions in an interview setting (e.g., able to recall experiences and verbally relay them to the interviewer). Procedures Consent and confidentiality Consent was evaluated through a series of five questions designed to assess: (a) capacity to consent (i.e., understanding of research purpose, risks, and benefits), (b) voluntariness, (c) understanding of the right to withdraw consent at any time without consequence, and (d) understanding of the right to refuse to answer questions (Arscott et al., 1998). Third parties present during the interview (i.e., case workers) had to be invited by the women and were required to sign a confidentiality agreement prior to the interview. Avoiding distress There is a potential for eliciting psychological discomfort when discussing sensitive topics and sharing personal information; therefore, we developed the following procedure: (a) adopted strong interviewing skills such as sensitivity and empathy; (b) took breaks where needed; (c) terminated the interview if it became too distressing; (d) assured participants that they could decline to answer any questions and withdraw from the study at any time without consequence; (e) remained with the participant and shifted the interview to pleasant exchanges before terminating the interaction if distress was suspected (Corbin and Morse, 2003). Measurement Instrument Data were collected in recorded one-on-one semi-structured
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
interviews. Key components of the interview guide included demographic information, questions related to pregnancy and childbirth, a modified version of the Social Support Self-Report (SSSR) (Lunsky and Benson, 1997, 2001), and a social circles task (Lunsky and Benson, 1999). The SSSR is a self-reported measure of the structure and perceived quality of social support received by adults with IDD. Questions about structure are Yes/No and openended. Questions about perceived quality use a 3-point Likert scale (a lot, sometimes, not at all), which has been shown to be appropriate for individuals with IDD (Hartley and MacLean, 2006). The SSSR has high internal consistency (Cronbach's α ¼ 0.85) and is correlated with other measures of social support (r ¼0.75) (Lunsky and Benson, 1997). We added prompts to SSSR questions to elicit detailed information about social support received during pregnancy and childbirth. The social circles task is a visual activity that facilitates the provision of information about the structure of social support (Lunsky and Benson, 1999). Participants were invited to write the names of individuals who were in their life during pregnancy and childbirth inside a series of concentric circles based on perceptions of closeness to these individuals. Since the experience of social support may change from pregnancy to pregnancy, we invited women to discuss each of their pregnancies within the last five years. Addressing acquiescence bias Although semi-structured interviews have been used successfully with individuals with IDD, they are prone to acquiescent response patterns, especially when respondents are unsure about certain facts (Beail, 2002). To minimise this issue, the interviewer adhered to established guidelines for interviewing individuals with IDD (Finlay and Lyons, 2001; Tassé et al., 2005; D’Eath, 2005) (e.g., emphasised that there were no right or wrong answers, encouraged participants to ask for clarification of questions during the interview as needed, and followed most Yes/No questions with open-ended questions to elicit explanations for responses). The interviewer also employed journaling immediately following each interview, which provided an opportunity to reflect on the interview process and to identify possible biases. Interviewer characteristics The interviewer (first author) was an undergraduate Psychology student and research coordinator who was supervised and trained by a Clinical Psychologist whose research expertise is in IDD (third author). The interviewer had experience conducting telephone interviews with adults with IDD and is a birth doula. Data analysis The first author used memo writing and constant comparison throughout data collection and analysis to track and build theoretical categories (Lempert, 2007). Following data collection, audio-taped interviews were transcribed verbatim. Qualitative data analyses for responses to open-ended questions were carried out using NVivo 10. First, text segments from interviews were coded line by line. Initial coding was considered tentative and was modified throughout analysis (Holloway and Wheeler, 2010). To strengthen the reliability and validity of the coding structure and to minimise researcher bias, the second and third authors audited the coding process twice during analysis. Following discussion of initially developed codes, emerging codes were used to create groups of concepts marked by similar traits (open-coding). These groups of concepts were then reassembled into theoretical categories, and patterns underlying their relationships were further explored to generate a preliminary conceptual framework (axial
59
coding) (Holloway and Wheeler, 2010). Direct quotes from participants, who were assigned pseudonyms, were used to illustrate theoretical categories. To maintain the voice of individual participants, we did not translate French quotes to English but provide an interpretation of those quotes in the text.
Findings Participant characteristics Four women participated in the study. Two of them had more than one pregnancy within the last five years, for a total of seven eligible pregnancies. Women's age at the time of the interview ranged from 23 to 32 years. Two women resided in an urban area, one in a semi-rural area, and one in a rural area. Three women chose to be interviewed at the service agency involved in recruitment; the fourth was interviewed at home. All women requested that their caseworker be present during the interview. Two of the interviews were conducted in French, and two were conducted in English. On average, each interview took 1.5 hours to complete. Structure of social support The size of women's social support networks during pregnancy ranged from 7 to 19 individuals. Formal support was provided by paid professionals (i.e., doctors, nurses, caseworkers). Informal support was provided mostly by family members (i.e., parents, siblings, children, extended family) and by the partner and his family. Very little informal support was provided by individuals outside the family. All women reported having few or no friendships; individuals referred to as friends were women who were pregnant or had children. Functions of social support Informational support was primarily provided by formal sources. Doctors provided information during antenatal visits, while caseworkers explained complex medical advice provided by doctors. Caseworkers also served as bridges to other sources of informational support (e.g., antenatal and parenting classes, dieticians): Catherine: “She's [caseworker] the one that […] like told me about the health unit, told me about the parenting courses, told me about all the stuff that I never knew, but that I needed. I would have never knew or found out about it until she told me.” Informational support was also provided by other pregnant women and mothers. The cousin of Catherine's partner helped her prepare for motherhood by teaching her how to bathe and hold a newborn: Catherine: “If they had kids, I asked questions to people who had the kids. I don’t know, I guess cause well […] they had kids, they know what they’re talking about […] I guess that's what I did, like, if somebody I knew had kids that's who I’ll be asking.” Instrumental support was similarly provided by formal and informal sources. However, types of instrumental support provided depended on the source. Doctors provided medical care during pregnancy and childbirth, whereas nurses mostly provided medical care during childbirth. Caseworkers, family members, and the partner's family similarly provided tangible help (e.g., preparing
60
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
the infant's room, acquiring items for the infant). Transportation was mainly provided by the caseworker and the partner's family. Alexandra received significant support from her caseworker who accompanied her to all of her appointments and helped her plan the infant's room, including the room colour and crib: Alexandra:
expectant and new mothers; she lost touch with that friend after childbirth. Friendships were also created with other mothers. Catherine described accompanying her oldest child to a daycare where she received companionship from a mother whose child also attended the programme: Catherine:
«C’est elle qui m’accompagnait tout le temps, [à]mes rendezvous… Je veux dire c’est elle qui, quand j’avais besoin, you know, préparer la chambre […] C’est elle qui m’a aidé à planifier la couleur de la chambre […] la couchette […] je veux dire elle a fait sa part avec moi.»
“We went to daycare and that, like for my little one, ‘cause they told me before she started school she’d have to get used to be around other kids cause she was the only one really. So we brought her to the daycare here in town and she played with the kids and that […] There was a girl named T* […] And she has a daughter named D*.”
Catherine's mother helped with the infant shower, and her partner's family purchased infant furniture: Catherine: “And my mom [helped out] all the time, like if I needed anything for my baby shower and everything […] His [partner's] mother went out and bought us like the crib, the change table, the dresser, all that stuff.” Emotional support was lacking for most participants. Camille, Alexandra, and Rose had few people to talk to during pregnancy. When Rose found out that she was pregnant with her first infant, she did not have anyone to talk to about her fears: Rose: “I didn’t know what to do, you know, I was scared. Like am I going to be able to do it on my own, you know? I didn’t have nobody […] I did not have a good relationship with my parents at the time either. So it was hard, but…” When Camille was asked if she had anyone to care for her when she felt sick during her pregnancy or give her a hug when she felt sad, she replied that she did not; she did it all alone: Camille: «Je n’avais pas de ça. Je n’avais pas de ça moi […] Je le faisais toute seule. » Where emotional support was available, it was provided mostly by informal sources, such as family members. Rose and Camille considered their older children as their primary sources of emotional support in subsequent pregnancies; they brought comfort during difficult times. Alexandra stated that she did not have many friends or anyone to confide in during her pregnancy, with the exception of her parents: Alexandra: «Moi pour te le dire, tu as touché un point aussi que moi, je n’ai pas des amis beaucoup là […] Non, à qui me confier, à qui demander… umm je veux dire non.[…]…heureusement j’avais mes parents à qui me… leur dire comment je me sentais.» To a lesser extent, formal sources also provided emotional support, particularly caseworkers (during pregnancy) and nurses (during childbirth): Catherine: “But, mainly it was a nurse […] but she was there the whole time and she was awesome.[…] I never saw a nurse that was that good before […] Yeah, she was making us all work together to have the baby.” Social companionship was reported by few women. The limited social companionship that was provided came from partners, children, pregnant friends, and other women who have children. In general, friendships created during pregnancy with other pregnant women tended to be restricted to that period. Camille talked about making a friend while attending a programme for
Sometimes, social companionship was provided by formal sources, primarily the caseworker. While Alexandra had no friend with whom to engage in leisurely activities, she did receive social companionship from her caseworker; she talked about going for coffee and doing physical exercises with her: Alexandra: «Je fais mes exercices […] une fois de temps en temps je le fais avec [personnel de soutien], je vais avec elle pour un café, ou discuter un petit peu.» Relationships with caseworkers were more stable and longterm than friendships with pregnant women. Perceived quality of social support Three factors related to women's perceived quality of available social support emerged from the data: the accessibility of the desired structure and functions of social support; the attitudes of support persons; and autonomy in determining social support. Accessibility related to distance and transportation to sources of support, as well as the clarity of presented information about pregnancy and childbirth. All the women's families lived in different cities, but many of the women resided in proximity to their partners’ families. Consequently, participants often reached out to whomever was easiest to access. Catherine primarily received support from her partner's family during pregnancy but would have preferred to be supported by her own family: Catherine: “I just mainly went to whoever's closer and I felt comfortable with […] they’re [partner's family] right in this town, so they at least come visit at least once a week or every day, so yes I am closer to them because they’re always there, but if I was to go to anybody I would go to my family […] but they’re not there, so I have to go with what I got.” Alexandra's parents lived in another country. Despite her strained relationship with her mother-in-law, she nonetheless reached out to her in-laws for support because they were easily accessible: Alexandra: «Quand j’avais besoin d’aide, j’appelais au téléphone la personne qui était libre, elle se présentait. Soit c’était mon beaupère, soit c’était la belle-mère, soit c’est les deux… » These distance and transportation issues also related to the accessibility of formal supports. Camille was encouraged by her caseworker to attend antenatal classes during her second pregnancy. However, she reported not attending due to a lack of transportation and of a support person to accompany her. There were also issues related to the understandability of information. Alexandra only attended two antenatal classes because the classes were not available in her first language (French).
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
Although Rose attended antenatal classes during her first pregnancy, she preferred to have a nurse explain the information to her one-on-one and to acquire information about pregnancy and childbirth herself through the Internet: Rose: “They were talking to people as a group. The group, the [prenatal] class […] I’m better to understand one-on-one. But the stuff I brought back, the nurse was able to explain it to me when she came.” Clarity of information was also an issue at antenatal visits. Alexandra reported receiving test requisition papers and pamphlets from her doctor which she had difficulty understanding until she met with her caseworker to ask questions. Attitudes related to women's perceptions of their support persons’ opinions and feelings about the pregnancy. Support received was perceived as unhelpful and undesirable if it was provided by individuals expressing negative attitudes. Rose perceived support from her doctor unfavourably due to his attitudes: Rose: “One thing that I didn’t like hearing [from her doctor] was that I was young. I know I’m young. I just need your help to get through it now, ya know? I didn’t plan to have a baby although I wasn’t taking precautions. I did not plan to have a baby at that age […] So I didn’t need people's negative thoughts and stuff.” Alexandra mentioned her mother-in-law and sister-in-law's negativity towards her pregnancy several times during the interview. They told her that getting pregnant was foolish, and that she “wouldn’t be able to” parent. Although no one openly told her that she should terminate her pregnancy, these negative comments contributed to fears that someone would broach the subject with her: Alexandra: « Mais j’avais peur d’entendre l’un ou l’autre me dire ‘va l’avorter’ […] Parce que j’ai aimé ce petit… la minute où j’ai entendu, j’ai senti que j’étais enceinte, c’était comme ‘no way là!’. Je ne veux pas que quelqu’un me le dit, non… » However, some support persons also expressed positive attitudes at the announcement of pregnancy. Most partners and some family members welcomed the pregnancies: Catherine: “Everybody was happy! […] They [in-laws] were really, really, really happy. To have the first grandkids and that.” Moreover, caseworkers were an important source of encouragement during pregnancy. Alexandra felt encouraged and supported by her caseworker. Alexandra: « Elle m’encourageait, elle me supportait… » Autonomy related to the perception that individual support needs and self-determination were respected. Most of the women reported instances when their preferences were not valued. Sometimes this lack of autonomy related to the women's or their partner's desire for independence. Alexandra became frustrated when her wish to go to her medical appointment unaccompanied was not respected. She explained that she and her caseworker had a disagreement over the issue; Alexandra wanted to prove that she was capable of seeing her doctor by herself after 7 months of going to appointments together: Alexandra: «Il y avait un petit, comment je peux dire… [un] malentendu
61
là? Dans le sens que je voulais lui [personnel de soutien] prouver que je suis capable d’aller toute seule [chez le médecin] […]Je veux dire, ça fait quoi, 7 mois que tu me suis là, je suis capable de le faire toute seule! Elle était, comment on dit, fâchée contre moi. Elle me dit ‘Non tu devrais m’attendre, on avait une entente’. ‘Come on, je suis capable!’» Catherine did not perceive support provided by her partner's aunt positively because she imposed her help even though Catherine did not request it: Catherine: “Like somebody is trying to help you or whatever[…] there's okay I need your help, but it's okay I don’t need your help. She's just trying, and trying, kind of thing […] Sometimes it's nice, but sometimes it was annoying […]” Conversely, although Camille wanted her caseworker's support during her pregnancy, her partner refused this service; he wanted the couple to be autonomous: Camille: « Parce que je l’aimais beaucoup quand elle était là pour [la première grossesse], pis je voulais qu’elle soit là, mais [il] était un gars qui voulait être autonome […] » Sometimes this lack of autonomy related to the women's desire to receive a certain type of support from a particular person but receiving it from someone else. Although Catherine received support from several individuals during childbirth, she would have preferred to have been supported by her sister. During labour, there were too many family members involved, leading to preoccupations with others’ feelings: Catherine: “So my sister didn’t end up coming in the room, but I really wanted her to. So the only one that was in there was like my mom and his mom […] The only one that I really wanted in my room [was] my sister […] and my mom, but my mom didn’t have to be like […] as long as I had my sister there I was comfortable.”
Discussion Summary of findings During pregnancy, women in this study received social support from both formal and informal sources. However, results highlight the central role of formal support, primarily provided by the caseworker. Although pregnant women with IDD created shortterm friendships with other pregnant women, they generally lacked informal relationships outside the family structure. Moreover, although high levels of informational and instrumental support and low levels of emotional support and social companionship were reported, high levels of available support were not always perceived as beneficial. On the basis of these findings, we developed a preliminary conceptual framework of effective social support during pregnancy and childbirth, as perceived by women with IDD. Fig. 1 illustrates the emergent core categories which suggest that social support is perceived as most effective when desired support is accessible, support is provided by individuals expressing positive attitudes towards the pregnancy, and autonomy is valued (e.g., individual support needs and self-determination are respected).
62
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
Availability Support received Desired Support
Functions of Support
Structure of Support
Accessibility, Attitudes & Autonomy (+)or (-) attitudesof support persons
Value of autonomy
Perception of support received
Perceived Quality of Support
Fig. 1. A preliminary conceptual framework of effective social support during pregnancy and childbirth as perceived by women with intellectual and developmental disabilities.
Consistency with previous research and potential explanations Our finding of impoverished informal support outside of the families of women with IDD is consistent with previous studies demonstrating that adults with IDD generally have restricted social networks, with few or no friendships, which mainly comprise family members and paid caregivers (Lunsky and Benson, 1999). Although few friendships were reported, all women in our study created short-term friendships with pregnant women and mothers. One possible explanation is that the shared experience of pregnancy, or motherhood, may have facilitated these relationships. We found that women with IDD generally received little emotional support and social companionship as they prepared for motherhood. This is consistent with previous findings of social isolation among pregnant women with IDD (Llewellyn and McConnell, 2002). This finding may be related to negative reactions towards childbearing in this population (McConnell and Llewellyn, 2002; Mayes et al., 2006; McConnell et al., 2008b; Walsh-Gallagher et al., 2012; Höglund and Larsson, 2013; WalshGallagher et al., 2013). Similarly to previous research (Lunsky and Benson, 1999), our study illustrates the centrality of the caseworker-client relationship in the provision of social companionship amongst adults with IDD. Women in our study received support from their caseworkers for an extended period of time prior to pregnancy and therefore had an ongoing relationship with them. However, research has also demonstrated that paid carers often view themselves as facilitators to their clients’ friendships rather than social companions themselves (Pockney, 2006). The accessibility issues reported in our study are consistent with previous findings. As in our study, lack of transportation is commonly cited as a barrier to social inclusion and support for people with IDD (Abbott and McConkey, 2006). Moreover, our finding that information about pregnancy and childbirth was not always clear for women with IDD parallels previous research suggesting that adapted antenatal informational support for women with IDD is lacking (McConnell et al., 2008a; Walsh-Gallagher et al., 2012; Redshaw et al., 2013). This finding is important as research has demonstrated that women with IDD often view childbearing as “overwhelming and difficult to understand” (Höglund and Larsson, 2013, p.704). Negative attitudes toward pregnant women with IDD found in our study are consistent with a history of opposition to women
with IDD becoming mothers (Booth and Booth, 1995; Aunos and Feldman, 2002; Mayes et al., 2006; McConnell et al., 2008b). Pregnancy in this population is often treated as a mistake, and expectant women with IDD often feel pressured to terminate their pregnancies (Booth and Booth, 1995; Mayes et al., 2006). Negative attitudes may have contributed to social isolation during pregnancy: pregnant women with IDD tend to exclude unsupportive individuals from their networks (Mayes et al., 2006). Conversely, women in this study also reported that some support persons expressed positive attitudes towards their pregnancy; this seemed to facilitate access to support and to increase satisfaction with support. Others’ attitudes were also linked to autonomy and service uptake. Our finding that some women with IDD and their partners struggled for autonomy during pregnancy is consistent with previous literature demonstrating that decision-making opportunities are often restricted in this population (Höglund and Larsson, 2013): the ability of women with IDD to parent is commonly scrutinised (McConnell and Llewellyn, 2002; Mayes et al., 2006; Greenwood and Wilkinson, 2013), they are afforded less control over their lives (Mayes et al., 2006), and are brought under “constant surveillance” (Booth and Booth, 1995, p. 31). Moreover, women with IDD may react with hostility and resistance to professionals who do not validate their role as mothers, and respect their needs (Wilson et al., 2013). This may explain why some participants felt a need to prove themselves as “able” and refused some services. Limitations Several issues may limit the transferability of our findings. While convenience sampling is an appropriate sampling strategy for hard-to-reach study populations (Morse, 2007), our sample excluded women who were not currently receiving services for adults with IDD. Moreover, participants had to possess sufficient recall and verbal capacity to engage in an interview, thus excluding women who were non-verbal or who had a lower level of functioning. We did not have specific information pertaining to diagnosis or level of disability; however, all of the women currently had custody of their children and lived independently in the community. Results therefore may not reflect the experiences of women who did not maintain custody and who resided with caregivers. The sample size (n¼ 4) is also an important limitation.
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
Although providing rich preliminary data, we cannot claim to have reached theoretical saturation. Therefore, we must emphasise the preliminary nature of our conceptual model; other equally significant concepts could have emerged from the data with the addition of other participants. Response biases may also limit results. Although measures to circumvent acquiescent responses were taken, persons with IDD are prone to respond in such patterns (Heal and Sigelman, 1995). Additionally, although individuals with IDD have the ability to accurately recall lived experiences (Beail, 2002), semi-structured interviews are vulnerable to recall bias. Every woman in this study requested to have her caseworker present during the interview, potentially biasing responses to questions about formal support. However, the centrality of the caseworker-client relationship was also reflected in this choice. Implications Our findings suggest that improvements in the accessibility of social support for women with IDD are warranted. Given that women with IDD struggled with complex medical information provided by doctors and in antenatal classes, maternity care providers such as doctors, midwives, and nurse practitioners should consider longer antenatal appointments to allow sufficient time to effectively communicate information to pregnant women with IDD (McConnell et al., 2008a). Midwives, doulas, and nurses, who tend to offer longer appointments than doctors, may have a valuable role in providing continuous support and maternity care to women with IDD. Previous studies have shown the value of midwifery support, particularly in situations where informal support is low (Wilson et al., 2013). Women with IDD consider doulas to be reliable sources of pregnancy-related information (McGarry et al., 2015). Moreover, doulas provide important social and emotional support during labour (Lantz et al., 2005). Maternity care providers should also consider caseworkers (or community-based support agency staff) as key resources; they serve as bridges to other sources of support and assist women with IDD with understanding information and complying with healthcare providers’ recommendations. Furthermore, Hodnett et al., 2013 suggest that involving a chosen member of the family or friend appears to increase satisfaction with childbirth. Informal support therefore, should also be facilitated. Our study demonstrates that stereotypes and negative attitudes still exist towards childbearing in this population. All pregnant women with IDD have the right to respectful and nonjudgmental support. Therefore, healthcare practitioners should be provided with appropriate knowledge and resources regarding pregnancy in women with disabilities in order to minimise negative or stigmatizing attitudes. As autonomy and decision-making opportunities were valued by women with IDD during pregnancy and childbirth, respecting individual support needs and adopting a collaborative rather than directive support approach are encouraged.
Conclusion Interviewing mothers with IDD allowed us to propose a preliminary conceptual framework of effective social support during pregnancy and childbirth as perceived by women with IDD. Although increasing support availability for women with IDD during pregnancy and childbirth should be a social and clinical priority, our results suggest that social support is perceived as most effective when it is perceived as accessible, it is received from individuals expressing positive attitudes, and autonomy is valued.
63
Conflict of interest We have no conflicts of interest to declare.
Acknowledgements We are grateful to the volunteer research assistants – Barbara Mackenzie Barnett, Préscillia Dupont and Charlotte Wagner Searle – who transcribed the audiotaped interviews.
References Abbott, S., McConkey, R., 2006. The barriers to social inclusion as perceived by people with intellectual disabilities. Journal of Intellectual Disabilities 10, 275–287. American Psychiatric Association, 2013. Diagnostic and Statistical Manual of Mental Disorders, 5th edn DC: American Psychiatric Publishing, Washington. Arscott, K., Dagnan, D., Kroese, B.S., 1998. Consent to psychological research by people with an intellectual disability. Journal of Applied Research in Intellectual Disabilities 11, 77–83. Aunos, M., Feldman, M.A., 2002. Attitudes towards sexuality, sterilization and parenting rights of persons with intellectual disabilities. Journal of Applied Research in Intellectual Disabilities 15, 285–296. Brown, H.K., Lunsky, Y., Cobigo, V., Wilton, D., Vigod, S., 2016. Pregnancy in women with intellectual and developmental disabilities. Journal of Obstetrics and Gynaecology Canada 38, 9–16. Beail, N., 2002. Interrogative suggestibility, memory and intellectual disability. Journal of Applied Research in Intellectual Disabilities 15, 129–137. Blackford, K.A., 1993. Erasing mothers with disabilities through Canadian familyrelated policy. Disability, Handicap and Society 8, 281–294. Booth, T., Booth, W., 1995. Unto us a child is born: the trials and rewards of parenthood for people with learning difficulties. Australia and New Zealand Journal of Developmental Disabilities 20, 25–39. Bramston, P., Fogarty, G., Cummins, R.A., 1999. The nature of stressors reported by people with an intellectual disability. Journal of Applied Research in Intellectual Disabilities 12, 1–10. Cohen, S., Wills, T.A., 1985. Stress, social support, and the buffering hypothesis. Psychological Bulletin 98 (2), 310–357. Cohen S., Syme S.L., 1985. Issues in the Study and Application of Social Support. Social Support and Health. Academic Press, San Francisco, pp. 3–22. Corbin, J., Morse, J.M., 2003. The unstructured interactive interview: issues of reciprocity and risks when dealing with sensitive topics. Qualitative Inquiry 9, 335–354. D’Eath, M., 2005. Guidelines for Researchers when Interviewing People with an Intellectual Disability. National Federation of Voluntary Bodies, Galway. Finlay, W.M.L., Lyons, E., 2001. Methodological issues in interviewing and using self-report questionnaires with people with mental retardation. Psychological Assessment 13, 319–335. Geller, P.A., 2004. Pregnancy as a stressful life event. CNS Spectrums 9, 188–197. Glazier, R.H., Elgar, F.J., Goel, V., Holzapfel, S., 2004. Stress, social support, and emotional distress in a community sample of pregnant women. Journal of Psychosomatic Obstetrics and Gynaecology 25, 247–255. Greenwood, N.W., Wilkinson, J., 2013. Sexual and reproductive health care for women with intellectual disabilities: a primary care perspective. International Journal of Family Medicine 2013, 642472. Höglund, B., 2012. Pregnancy, Childbirth and Midwifery Care Among Women with Intellectual Disability in Sweden: Epidemiological and Descriptive Studies (Unpublished doctoral dissertation). Uppsala University, Sweden. Höglund, B., Larsson, M., 2013. Struggling for motherhood with an intellectual disability – A qualitative study of women's experiences in Sweden. Midwifery 29, 698–704. Hartley, S.L., MacLean, J., W.E., 2006. A review of the reliability and validity of Likert-type scales for people with intellectual disability. Journal of Intellectual Disability Research 50, 813–827. http://dx.doi.org/10.1111/ j.1365-2788.2006.00844.x. Heal, L.W., Sigelman, C.K., 1995. Response biases in interviews of individuals with limited mental ability. Journal of Intellectual Disability Research 39, 331–340. Heaman, M., 1998. Psychosocial impact of high-risk pregnancy: hospital and home care. Clinical Obstetrics and Gynecology 41, 626–639. Hodnett, E.D., Gates, S., Hofmeyr, G.J., and Sakala, C., 2013. Continuous support for women during childbirth (Cochrane Review). The Cochrane Library, Issue 7. Retrieved from 〈http://onlinelibrary.wiley.com/doi/10.1002/14651858. CD003766.pub5/epdf〉. Holloway, I., Wheeler, S., 2010. Qualitative Research in Nursing and Health Care, 3rd edn Wiley-Blackwell Publishing, Oxford. Holmes, T., Rahe, R., 1967. The social readjustment rating scale. Journal of Psychosomatic Research 11, 213–218. Holton, J.A., 2007. The coding process and its challenges. In: Bryant, A., Charmaz, K. (Eds.), The SAGE Handbook of Grounded Theory. SAGE Publications Ltd.,
64
L.A. Potvin et al. / Midwifery 37 (2016) 57–64
London, pp. 265–290. http://dx.doi.org/10.4135/9781848607941.n13. Kempton, W., Kahn, E., 1991. Sexuality and people with intellectual disabilities: a historical perspective. Sexuality and Disability 9, 93–111. Lantz, P.M., Low, L.K., Varkey, S., Watson, R.L., 2005. Doulas as childbirth paraprofessionals: results from a national survey. Women's Health Issues 15, 109–116. Lempert, L.B., 2007. Asking questions of the data: memo writing in the grounded theory tradition. In: Bryant, A., Charmaz, K. (Eds.), The SAGE Handbook of Grounded Theory. SAGE Publications Ltd., London, pp. 245–265. Lin, E., Balogh, R., Cobigo, V., Ouellette-Kuntz, H., Wilton, A.S., Lunsky, Y., 2013. Using administrative health data to identify individuals with intellectual and developmental disabilities: a comparison of algorithms. Journal of Intellectual Disability Research 57, 462–477. http://dx.doi.org/10.1111/jir.12002. Llewellyn, G., McConnell, D., 2002. Mothers with learning difficulties and their support networks. Journal of Intellectual Disability Research 46, 17–34. Lunsky, Y., Benson, B.A., 1997. Reliability of ratings of consumers with mental retardation and their staff on multiple measures of social support. American Journal on Mental Retardation 102, 280–284. Lunsky, Y., Benson, B.A., 1999. Social circles of adults with mental retardation as viewed by their caregivers. Journal of Developmental and Physical Disabilities 11, 115–129. Lunsky, Y., Benson, B.A., 2001. Association between perceived social support and strain, and positive and negative outcome for adults with mild intellectual disability. Journal of Intellectual Disability Research 45, 106–114. Lyons, K.S., Zarit, S.H., 1999. Formal and informal support: the great divide. International Journal of Geriatric Psychiatry 14, 183–196. Mayes, R., Llewellyn, G., McConnell, D., 2006. Misconception: the experience of pregnancy for women with intellectual disabilities. Scandinavian Journal of Disability Research 8, 120–131. http://dx.doi.org/10.1080/15017410600774178. McConnell, D., Llewellyn, G., 2002. Stereotypes, parents with intellectual disability and child protection. Journal of Social Welfare and Family Law 24, 297–317. http://dx.doi.org/10.1080/09649060210161294. McConnell, D., Mayes, R., Llewellyn, G., 2008a. Women with intellectual disability at risk of adverse pregnancy and birth outcomes. Journal of Intellectual Disability Research 52, 529–535. http://dx.doi.org/10.1111/j.1365-2788.2008.01061.x. McConnell, D., Mayes, R., Llewellyn, G., 2008b. Pre-partum distress in women with intellectual disabilities. Journal of Intellectual and Developmental Disability 33 (2), 177–183. McGarry, A., Stenfert Kroese, B., Cox, R., 2015. How do women with an intellectual disability experience the support of a doula during their pregnancy, childbirth and after the birth of their child? Journal of Applied Research in Intellectual Disabilities . http://dx.doi.org/10.1111/jar.12155 [Epub ahead of print]. Mitra, M., Parish, S.L., Clements, K.M., Cui, X., Diop, H., 2015. Pregnancy outcomes among women with intellectual and developmental disabilities. American Journal of Preventive Medicine 48, 300–308.
Morikawa, M., Okada, T., Ando, M., Aleksic, B., Kunimoto, S., Nakamura, Y., Ozaki, N., 2015. Relationship between social support during pregnancy and postpartum depressive state: a prospective cohort study. Scientific Reports 5, Retrieved from 〈〈http://www.nature.com/articles/srep10520〉〉. Morse, J.M., 2007. Sampling in grounded theory. In: Bryant, A., Charmaz, K. (Eds.), The SAGE Handbook of Grounded Theory. SAGE Publications Ltd., London, pp. 229–245. http://dx.doi.org/10.4135/9781848607941.n11. Oakley, A., 1988. Is social support good for the health of mothers and babies? Journal of Reproductive and Infant Psychology 6, 3–21. Pockney, R., 2006. Friendship or facilitation: people with learning disabilities and their paid carers. Sociological Research Online 11, Retrieved from 〈〈http://www. socresonline.org.uk/11/3/pockney.html〉〉. Redshaw, M., Malouf, R., Gao, H., Gray, R., 2013. Women with disability: the experience of maternity care during pregnancy, labour and birth and the postnatal period. BMC Pregnancy Childbirth 13, 174, Retrieved from 〈〈http://www. biomedcentral.com/1471-2393/13/174〉〉. Robson, C., 2002. Real World Research. A Resource for Social Scientists and Practitioner-Researchers, 2nd edn Wiley, Oxford. Royal College of Midwives, 2000. Position Paper 11a. Maternity Care for Women with Disabilities. Royal College of Midwives, London. Servais, L., Jacques, D., Leach, R., Conod, L., Hoyois, P., Dan, B., Roussaux, J.P., 2002. Contraception of women with intellectual disability: prevalence and determinants. Journal of Intellectual Disability Research 46, 108–119. Sherbourne, C.D., Stewart, A.L., 1991. The MOS social support survey. Social Science and Medicine 32, 705–714. Tassé, M.J., Schalock, R., Thompson, J.R., and Wehmeyer, M., 2005. Guidelines for interviewing people with disabilities: Supports intensity scale. American Association on Intellectual and Developmental Disabilities. Washington, DC. United Nations, 2006. Convention on the Rights of Persons with Disabilities (CRPD). United Nations, New York, NY, Retrieved from 〈http://www.un.org/disabilities/ convention/conventionfull.shtml〉. Walsh-Gallagher, D., Sinclair, M., Mc Conkey, R., 2012. The ambiguity of disabled women's experiences of pregnancy, childbirth and motherhood: a phenomenological understanding. Midwifery 28, 156–162. Walsh-Gallagher, D., Mc Conkey, R., Sinclair, M., Clarke, R., 2013. Normalising birth for women with a disability: the challenges facing practitioners. Midwifery 29, 294–299. Willis, T.A., Shinar, O., 2000. Measuring perceived and received social support. In: Cohen, S., Underwood, L.G., Gottlieb, B.H. (Eds.), Social Support Measurement and Intervention: A Guide for Health and Social Scientists. Oxford University Press, New York, pp. 86–135. Wilson, S., McKenzie, K., Quayle, E., Murray, G.C., 2013. The postnatal support needs of mothers with an intellectual disability. Midwifery 29, 592–598. Wingfield, M., Healy, D.L., Nicholson, A., 1994. Gynaecological care for women with intellectual disability. The Medical Journal of Australia 160, 536–538.