- Email: [email protected]
Social stigma and compounded losses: quality-of-life issues for multiple-birth families
FERTILITY AND STERILITY威 VOL. 80, NO. 2, AUGUST 2003 Copyright ©2003 American Society for Reproductive Medicine Published by Elsevier Inc. Printed on acid-free paper in U.S.A.
Social stigma and compounded losses: quality-of-life issues for multiple-birth families Marcia A. Ellison, Ph.D., and Janet E. Hall, M.D. Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts
Received August 19, 2002; revised and accepted March 8, 2003. Supported by the National Institutes of Health, Bethesda, Maryland (T32HD 07396), and Harvard Medical School’s Center of Excellence in Women’s Health Fund, Boston, Massachusetts (213-98-0009). Presented at the Harvard Medical School’s Center of Excellence in Women’s Health, Women’s Health Research Conference, Boston, Massachusetts, October 9, 2002. Reprint requests: Marcia A. Ellison, Ph.D., Reproductive Endocrine Unit, Massachusetts General Hospital, 55 Fruit Street, Bartlett Hall Ext. 5, Boston, Massachusetts 02114-2696 (FAX: 617-726-5357; E-mail: [email protected]. 0015-0282/03/$30.00 doi:10.1016/S0015-0282(03) 00659-0
Objective: To determine the quality-of-life domains most impacted by multiple births. Design: Focus groups, qualitative research. Setting: Human volunteers in a medical research environment. Patient(s): Forty-three mothers, 29 raising multiple-birth children, 13 raising singletons, identified from random and convenience samples. Intervention(s): None. Main Outcome Measure(s): Maternal self-reports of the psychosocial sequelae of multiple or singleton births, based on qualitative data analysis of transcribed group discussions. Result(s): The quality-of-life domains that were most impacted by raising multiple birth children were social stigma, pregnancy loss, marital satisfaction, children’s health, unmet family needs, parenting stress, maternal depression, and the infertility experience. Conclusion(s): Qualitative methods identified two novel quality-of-life domains in iatrogenic multiple birth families: social stigma and compounded losses. An unexpected finding was the potential for increased marital solidification as parents coped with the inordinate stresses of multiple births. As anticipated, children’s health, unmet family needs, maternal depression, and parental stress were key areas of concern. In addition, the infertility experience had a lasting impact. These findings are significant, given that at least 38% of all assisted conceptions result in a multiple birth. This study lays the groundwork for further research on the impact of iatrogenic multiple births. (Fertil Steril威 2003;80:405–14. ©2003 by American Society for Reproductive Medicine.) Key Words: Multiple births, infertility, social stigma, qualitative research
Since the 1970s, twin birth rates in the United States have increased 55% (from 18.9 to 29.3 per 1,000), while rates of triplet and higher order multiple births increased 423% (from 37 to 193.5 per 100,000) (1, 2). At least 70% of these increases are the result of assisted reproduction (3– 4). Although the increased maternal– child health risks associated with assisted reproduction have been documented (5– 10), relatively little is known about the concomitant social risks that these families may face, particularly those raising multiple-birth children. The results of studies of singleton in vitro fertilization (IVF) families have produced contradictory results. Two longitudinal studies documented increased parental warmth and parental involvement among IVF families at the
initial study point (11, 12). However, this outcome appears to be associated with the infertility experience, as adoptive parents also scored higher on these indices than parents of naturally conceived children. At the follow-up study points, as the children transitioned to adolescence, IVF parents in the first study (11) reported greater enjoyment of their parental roles than parents of adopted or naturally conceived children. But some IVF parents were also more emotionally enmeshed, or overly involved, with their children (13). The other longitudinal study (12) found that the IVF parents were less sensitive to their now adolescent children than those with adopted or naturally conceived children. Yet at this time point IVF parents exhibited greater warmth than adoptive parents (14). 405
In contrast, two additional longitudinal studies found significant and persistent levels of psychosocial impairment among first-time IVF parents as compared with fertile controls, including lower parental self-esteem and an impaired sense of maternal competence. These families also perceived their children as more vulnerable and were more childfocused than the control families who had not experienced infertility (15, 16). Given these contradictory findings, it is difficult to determine how much of these outcomes is the result of the experience of infertility versus fertility treatment. The potential social risks for families who have faced infertility may be exacerbated by a multiple birth. A seminal study, that found increased parenting stress in families of IVF versus spontaneous twins, was confounded by parity status (17); most IVF mothers were primiparas, whereas mothers of spontaneous twins had already made their transition to parenthood. Yet a study that controlled for parity status also documented increased stress as well as decreased psychosocial well-being among parents of IVF versus spontaneous twins (18). Multiple births, independent of fertility treatment, present increased stressors for families. For example, studies indicate that the unremitting stresses of raising multiple-birth children may increase the risk of nonperinatal maternal depression. A study, conducted in the early 1970s, before the advent of iatrogenic multiple births, found that mothers of twins were more vulnerable to depression than mothers of closely spaced singleton births (19). Two studies that documented high rates of persistent maternal depression among triplet mothers are provocative. However, these studies did not include a control group and because of their small samples are not generalizable (20, 21). Thus, the role of assisted reproduction as an independent predictor of longterm psychosocial sequelae and the impact of multiple births on the family have yet to be determined.
MATERIALS AND METHODS The purpose of this study was to determine the domains of family quality-of-life that are most impacted by multiple births. Given the equivocal findings on multiple-birth family outcomes to date, it was important to determine the domains of family life that were most impacted by multiple births. Clearly, multiple birth mothers could provide insights and identify outcomes that might have been overlooked in previous studies. Thus, this study used focus groups to identify the quality-of-life domains that were most relevant to multiple-birth mothers. Qualitative research strategies, such as focus groups, are particularly well suited to elucidating the key concerns of a particular population (22–24).
cultures. The initial focus groups (n ⫽ 33) included mothers regardless of their fertility status (i.e., spontaneous versus assisted conceptions). The homogeneity of the groups, and thus the comfort of the participants, was enhanced by separating participants by birth type. To control for idiosyncratic group responses (25), two groups were conducted for each of the three birth types: [1] high-risk multiple births (ⱕ2,500 grams, or ⬍37 weeks’ gestation), [2] low-risk multiple births, and [3] singleton births. The mothers of singletons (n ⫽ 10) and low-risk multiplebirth children (n ⫽ 12) were recruited from a random sample of mothers raising children 4 to 5 or 9 to 10 years of age. This sample, identified from the State of Massachusetts Birth Registry, was restricted to children of normal birth weight and gestational age. As a result, to control for sample BIAs, we recruited high-risk multiple-birth mothers (n ⫽ 11) from a convenience sample of peer support groups; their children ranged from 1 to 5 years of age. Preliminary analysis of these six focus groups indicated that infertility remained central to women’s experiences. Therefore, to control for the infertility experience, two additional groups were limited to participants who had conceived through assisted conception. These participants, also identified from a convenience sample of peer support groups, were raising singleton (n ⫽ 4) or multiple birth children (n ⫽ 6), from 8 months to 11 years of age.
Focus Group Protocol All study protocols were reviewed and approved by the institutional review board at Massachusetts General Hospital. Potential participants received a letter or e-mail flyer that described the study and provided contact information. Respondents were screened by telephone to ensure that they met the study’s eligibility requirements (i.e., children’s conception and birth type, children’s age). Recruited participants were mailed a packet that included a cover letter that described the study and the focus group protocol, and two copies of an informed consent form. This gave all identified women time to review the study protocol and materials before attending the group session, thus increasing the voluntary nature of their participation. Signed consent was obtained before each group discussion.
Study Participants
Each session was held in a private conference room at Massachusetts General Hospital. Because of the potentially sensitive nature of the discussion topics, the groups were kept small and were conducted on a first-name-only basis. This protected the privacy of the participants and fostered a sense of trust. The initial series of semistructured discussions followed a schedule of topics derived from the literature, including family stresses, family needs, social programs, social support, children’s health, marital satisfaction, and impact on women’s sense of self.
Study eligibility was limited to mothers, as women continue to be the primary childcare givers in Euro-American
The two follow-up groups explored topics that emerged from the previous sessions, such as women’s evaluations of
406
Ellison and Hall
Quality-of-Life: Multiple birth families
Vol. 80, No. 2, August 2003
TABLE 1 Focus group demographics. Demographic Birth type Singletons Twins Triplets Quadruplets Maternal age at child birth 20–24 25–29 30–34 35–39 40⫹
Initial groups (n ⫽ 33)
Follow-up groups (n ⫽ 10)
30% (10) 24% (8) 40% (13) 6% (2)
33% (3) — 56% (6) 11% (1)
3% (1) 18% (6) 27% (9) 43% (14) 9% (3)
— 10% (1) 40% (4) 30% (3) 20% (2)
Ellison. Quality-of-Life: Multiple birth families. Fertil Steril 2003.
assisted reproductive medicine and their treatment outcomes. Each session, which lasted 2 to 3 hours, was audiotaped and transcribed verbatim, using pseudonyms to protect participant confidentiality.
Data Analysis The statistical package SPSS was used to manage and analyze the descriptive statistics. The narrative data were managed using a computerized qualitative data analysis package (QSR NUD*IST Vivo). As is advised in qualitative research, data analysis was ongoing throughout the data collection process, which focused the direction of subsequent discussions. The study investigators reviewed the transcripts to establish interrater agreement about the initially identified themes. Themes were systematically identified using the standard grounded theory procedures of open and axial coding (26). Open coding identified key themes, which were assigned an identification label or “code.” Each code was then examined separately for recurrent patterns or motifs, and to determine the spectrum of responses. Axial coding, the reaggregation of data conceptually rather than temporally or literally, was used to determine the links between each theme. These procedures identified core quality-of-life domains.
RESULTS Demographics The characteristics of the study participants by birth type and maternal age are presented in Table 1. The study (n ⫽ 43) comprised 29 multiple birth mothers and 13 singleton mothers. Over half (58%) of the participants had undergone assisted conception, of which 78% resulted in a multiple birth. Sixty percent of the participants in the initial six focus groups (n ⫽ 33) were primiparas. Their children ranged from FERTILITY & STERILITY威
1 to 11 years of age. Thirty-two of the mothers were white, one was Asian American; 32 were currently married, one was separated. All but three had a college degree (58%), and 33% had an advanced degree. Combined pretax household incomes ranged from at least $100,000 (42%), to $60,000 – 99,000 (29%), to under $60,000 (18%). In the two follow-up groups (n ⫽ 10), eight participants were white, one was African American, and one was Hispanic. All were currently married and living with their husbands, although two of the multiple-birth mothers mentioned a potential separation. All of the participants had completed a 4-year college degree; and 33% had an advanced degree. Reported pretax combined household incomes ranged from $100,000 or more (25%), to $80,000 –99,999 (38%), to $60,000 –79,999 (25%), to under $60,000 (12%). These demographics reflect state and national statistics of multiple-birth families, which tend to be white with high income and education levels (27, 28). Similar to national statistics, the multiple-birth mothers in this study reported significantly higher rates of low birth weight (P⬍.001), prematurity (P⬍.001), chronic children’s health problems (P⫽.003), and assisted conception (P⬍.001), than the singleton mothers (Fisher’s exact test, two-sided, P ⫽ .05; Fig. 1).
Qualitative Data Analysis Preliminary data analysis identified eight core domains of quality of life that were most impacted by multiple births: [1] social stigma, [2] pregnancy and neonatal losses, [3] marital satisfaction, [4] children’s health, [5] meeting family needs, [6] parenting stress, [7] maternal depression, and [8] the infertility treatment experience. Data illustrating each of these domains are presented. Two additional themes, children’s development and school placement issues, were not included in the final analysis, as they were beyond the scope of the current research focus. Infertility treatment issues continued to be relevant for both singleton and multiple-birth mothers, but the other identified quality-of-life domains were more marked in multiple-birth mothers. Therefore, unless noted otherwise, the quotations are typical and representative of multiple-birth mothers’ responses to a particular topic. Because informal speech is less concise than the written word, quotations have been edited to preserve their intended meaning. Identifying information, including names, has been changed to protect participant confidentiality.
Social Stigma A devalued or spoiled identity is the hallmark of social stigma. All multiple-birth mothers had experienced social stigma related to their fertility treatment. Social stigma may be perceived or enacted through social interactions. As an example, participants discussed at length how family and friends had asked unsolicited and insensitive questions about their fertility status. The focal point of these invasive ques407
FIGURE 1
FIGURE 2
Children’s health outcomes by birth type (n ⫽ 43): f ⫽ low birth weight; u ⫽ premature; f ⫽ chronic health problem; f ⫽ assisted conception.
Follow-up focus groups (n ⫽ 10). Pregnancy losses and neonatal deaths: f ⫽ miscarriages; f ⫽ stillbirths; f ⫽ neonatal death; f ⫽ selective reduction.
Ellison. Quality-of-Life: Multiple birth families. Fertil Steril 2003.
Ellison. Quality-of-Life: Multiple birth families. Fertil Steril 2003.
tions was their children’s conception, specifically whether it was “normal” or “natural,” and by extension, whether they and their families were normal or natural. It always surprised me that people feel very free to ask such a personal question. It took me back. “Were you using something?” That would be the question. My response became “cheap wine.” (Pamela, mother of twins) We really wanted kids and we had gone on Clomid. But the way that his parents and family think and the way that my family is, we didn’t want them to know. All the talk. I don’t want them looking at my kids like they have four heads. When we had the triplets we didn’t tell them about our treatment, but a family member asked, “What kind of drugs were they on?” (Ellen, mother of triplets)
One of the core features of social stigma is a discredited moral status (29). A number of participants described social interactions, often with strangers, that resulted in moral judgments. The punitive comments they described reflect an assumption that assisted conception breaches sacred or secular norms. Thus, fertility treatment and its consequences may be cast as moral violations of God or nature. Because my son has cerebral palsy people say things like, “That’s what you get for messing with Mother Nature.” (Laura, mother of triplets) A woman I didn’t know asked me very nicely if I had fertility treatment. When I said yes her response was, “How dare you mess around with God’s will. It’s not natural. If you weren’t getting pregnant on your own there was a reason.” (Tony, mother of twins).
they’ll be impressed. If I say no they’ll think there’s something wrong with me. My mother-in-law is much more comfortable since we lost three of our children. Now everything’s normal. Nobody has to know that I had IVF. Nobody has to know that I had multiples. For her this is the best thing that could have happened. Now we look like a normal family. (Terry, mother of quadruplets)
Pregnancy and Neonatal Losses Similar to the high rates of miscarriage among women seeking fertility treatment (31), 60% of the participants in the two follow-up groups that controlled for infertility (n ⫽ 6 out of 10) had experienced a pregnancy or neonatal loss (Fig. 2). I got to almost 27 weeks after pulmonary edema, three blood transfusions, and burning a hole in my esophagus from acid reflux. We thought we had licked it but I got an infection. It killed one of my children in utero. Right after we had passing ceremonies for her, my son had off the chart head bleeds. He lived about two weeks. One of the triplets survived. She has a form of CP but she’s the greatest kid. (Laurie, mother of triplets) I tried Clomid. It didn’t work. We tried IUI. That didn’t work. Then, our first in vitro worked. One of them split, so I had a singleton and a set of identical twins. One twin died in utero. The other had a brain bleed and has CP, but every day she does something new. The singleton survived; she’s huge. They’re great kids. (Michele, mother of triplets)
These losses created acute emotional needs that were not always recognized nor well met.
Another potential hallmark of social stigma is a “courtesy stigma” which may extend, like a contagion, to social intimates (29, 30). As a result, children conceived through assisted conception, as well as extended family members, may be vulnerable to social stigma.
Everything happened on weekends. I delivered at 25 weeks. The social worker asked if I wanted a chaplain to say a service for the baby that died. During the service he kept calling the baby the wrong name. After that the social worker said, “Well, it’s Friday and it’s a long weekend, so I’ll talk to you on Tuesday.” (Lynn, mother of triplets)
When I was pregnant with quads, everyone assumed I had ART. Some people would say, “Was it natural?” If I say yes
In addition, for women who have experienced infertility, selective reduction poses a poignant personal and moral
408
Ellison and Hall
Quality-of-Life: Multiple birth families
Vol. 80, No. 2, August 2003
dilemma. In this double-bind situation, a couple must make a decision regarding a voluntary medical procedure that requires them to sacrifice one fetus for the greater good of their surviving fetuses, while they risk losing their entire pregnancy. Not many people know this, but mine were in vitro. I went through all that. They used to implant sixteen eggs because they wanted to get good results. We didn’t think anything of it. We figured, hey, the more the merrier. We just want to be able to get pregnant. I got pregnant with five. (Wendy, mother of twins) Yeah, they’re playing the odds. Did you have selective reduction? (Sandy, mother of triplets) Nothing was selective about it but that’s the word they use. He really didn’t give me much of a choice. He said to me, “You should go to two.” He explained the ramifications. It was a hard decision because you’ve had infertility for eight or nine years and you have all these great kids inside of you and then you’re doing something that goes against all that. I thought, if I can give these two the chance, I had to go for that. (Wendy)
Moreover, selective reduction may be at odds with desires to have a larger family, or may conflict with patients’ religious or moral beliefs. Their personal histories, including their treatment experiences, may also strongly influence their decision. Thus, a number of participants resisted their doctors’ advice and carried a multifetal pregnancy to term. Once our first IUI wasn’t successful, we tried IVF and I miscarried. In the second IVF cycle I lost twins. The third cycle was quadruplets; that was just too many. They advised us to reduce. I couldn’t do that to one of my children. How do you decide which one? We terminated that cycle. The fourth cycle was our triplets. We have two embryos left; we’re donating them to research. (Mandy, mother of triplets)
Marital Impacts The impact of multiple births on a marriage varied considerably. Many participants described losing touch with their husband as the incessant demands of raising multiplebirth children engulfed their marriage. We’re definitely just passing in the night. He goes to bed a half-hour after he gets home, because he gets up so early. It’s hard to take the little time he spends with the family and take it all for myself. Maybe we both tend to put the children first. We tend to put our marriage on the back burner. (Randy, mother of twins)
Couples unable to equitably divide family and household labor, or who were unable to work together as a team, described the slow erosion of their marriage. Ever since my multiple pregnancy my husband can’t handle what happened to our marriage. Our perfect little family, we were going down this perfect little path and then we get bombarded with triplets. It wasn’t part of the plan. Having children was part of the plan but he can’t handle three at one time. (Victoria, mother of triplets)
FERTILITY & STERILITY威
We had the good life before kids. Money was not an issue. We took lots of vacations. With our first child it was okay. With the triplets our life came to a screeching halt. I was on bedrest. He had to take over our son’s and my care. Then we had to make it through NICU. Now that the children are toddlers he can’t handle the havoc on the house and the noise level after working 12 hours. They’re not quiet and bathed and in their jammies. As often as I tried, it’s not that way. It’s just too overwhelming for him. (Kate, mother of triplets)
In contrast, the inordinate stresses of raising multiplebirth infants strengthened some marriages. In the face of such unremitting stress, some participants concluded that they would need to become a team if their marriages were to survive. This kind of teamwork typically resulted in greater paternal participation in childcare and household tasks. Twins changed everything for us. My husband suddenly parented a lot more than he had with our first child. Caring for twin infants was really difficult. He’s probably a much better parent because we have twins. (Audrey, mother of twins) You learn to work as a team or you don’t survive. It becomes a team effort; to survive you really must. I think it made us closer because of the team effort. You find out whether you really care about each other or not. (Megan, mother of twins)
Children’s Health Children’s health problems ranged from minor fine motor skill problems or the difficulty of dealing with the dominoeffect of normal childhood illnesses, to severe chronic illnesses and disabilities, particularly among triplets and quadruplets. I love my children but I didn’t plan on having four kids at once and needing overnight nurses for children with special needs, like quadriplegia, feeding tubes, blindness, and permanent brain shunts. (Carol, mother of quadruplets)
In another group session a mother of healthy twins commented, What happens when these kids go home and they have CP, and they’re blind, and they have to be fed through a feeding tube? What services are there for these special needs families? (Megan, mother of twins)
Mothers raising high-risk multiple-birth children, whose children are most likely to have special needs, were vocal about their families’ unmet needs and long-term vulnerabilities. They noted that even in well-trained tertiary care settings, they were not always informed about available support services. Moreover, the needs and increased emotional vulnerability that often arise in families whose children’s health is fragile were not always recognized or attended to. I was in the hospital for two weeks before I delivered and for five days after. The social worker came to meet me as I was discharged and told me that she had me on her list to visit. Then, it was almost four months after we had been
409
taking two cars a day to visit our children in NICU that she told us about parking coupons for families with children in NICU. (Jane, mother of triplets) Our family is comfortable financially but when my daughter coded they took her by ambulance to the hospital. When we got there they kicked me out of the room. Four people carried me out. I said that it was in my Bill of Rights and that I did not have to leave my baby. If my child is going to die I want to be there. (Pam, mother of triplets)
Meeting Family Needs The birth of a child increases the needs of all families. However, these needs increase exponentially for families with twins, triplets, or more. This includes daily needs such as diapers and formula, as well as the need for more car seats, specialized strollers, and cars and homes large enough to accommodate an immediate influx of children. Maybe a lot of us decide to have two kids, three kids, eight kids. But it’s different when, boom, you have two at once. It’s really no different than your neighbor next door that has two kids. But now you have to spend $35 on formula as opposed to $17 for one child. Can you imagine what it costs for daycare, or to send your kids to private nursery school or preschool times two or three? (Megan, mother of twins) I had to move to a larger home. The three little ones were in the living room. I had to go from my Honda Civic to a Chevy Suburban. (Nancy, mother of triplets)
For multiple-birth parents, childcare expenses also increase exponentially. Even a night out for dinner and a movie may require finding, hiring, and paying two or three babysitters to care for multiple-birth children and any additional children in the household. Moreover, their childcare expenses may have already been exacerbated by weeks or months of full bedrest prescribed during a multiple pregnancy. I had to give up my job when I was pregnant with the triplets. Then my husband was laid off. We have huge hospital bills. We’ve had four ambulances to the emergency room in four weeks. We’re still in shellshock after losing one of our children less than a year ago. (Jane, mother of triplets)
Increases in childcare expenses may effectively resolve the dilemma many couples face about the trade-off of daycare versus a second income. However, while some participants felt they were fortunate to be stay-at-home mothers, for others giving up their career meant losing part of their identity and independence. I left my job. That was my thing; that was my time. Now my time is spent for everybody else, and not for me. Every now and then I can eke out time with a friend, but otherwise there’s nothing for me. My husband has his job; he can socialize at work. My social life, my world, is my kids. (Randy, mother of twins)
Parenting Stress Sleep deprivation was a ubiquitous complaint, but the other most significant stressor was managing the daily, re410
Ellison and Hall
Quality-of-Life: Multiple birth families
petitive routines of family life. Participants described the difficulty of the logistics and juggling that were required to meet their families’ needs and schedules. The biggest stresses are getting people where they need to go and finding time to do things. They still need help toileting themselves, the whole routine of meals and snacks and then cleaning up, getting everyone dressed and planning what we’re going to do, and then trying to do it. And in between getting the laundry and groceries done, paying bills. So when we do have money that’s not even the whole answer. It’s getting the time to do any of this. (Phyllis, mother of triplets) When they were newborns and toddlers, the logistics was very hard. You’re the only one with them, carrying them places, getting them in and out of cars, dressing them and getting them out at the same time. That was the hard part. You can’t do anything. You can’t go to the bathroom. You can’t eat. That was just for the first year or two. I think that’s the big difference to raising singletons. (Randy, mother of twins)
In addition, while one might assume that multiple births are more stressful for first-time parents, multiple births may also pose a unique stressor for families with previous children who reported struggling to meet the needs of their older children. It was hard to integrate the triplets into an existing family. Our daughter was an only child. My husband, who was also an only child, said, “I really think two is better. Let’s go for two.” The second ended up being triplets. For us it was having him for five years, and then bringing in what felt like this group. Kabam. It changes everything. Cribs everywhere. It was crazy. It was hard for us to be sensitive to how our first child was feeling about it. We each chose a name for one of them. (Phyllis, mother of triplets) Having my first-born and then having triplets, I sometimes feel guilty because the first one doesn’t get enough attention. I feel selfish that I wanted a sibling for the older child. Today my older son said to me, “You didn’t give me any attention today.” That hurts; that hurts a lot. (Victoria, mother of triplets)
Iatrogenic multiple births may generate another unique parenting stressor. Unlike mothers whose twins, triplets, or more were spontaneous, mothers who sought fertility treatment, although they did not ask for a multiple gestation, may feel that they cannot complain about being exhausted and overwhelmed. Infertility affects how you parent your children. I used to think, how could I complain? I was exhausted. I was working. But I would always smile because I drove everyone crazy for eight years trying to have kids. Who am I to start complaining? Then I read a book for multiple birth families that said, “It’s okay to complain because your issues are real. It may have taken a little bit longer than some other people to get pregnant but it’s okay to be tired. You don’t have to
Vol. 80, No. 2, August 2003
smile all the time about being up half the night.” (Megan, mother of twins)
Maternal Depression Although anecdotal evidence of high rates of depression among these mothers was discussed, few participants reported nonperinatal depression. However, a number of participants who experienced postnatal depression felt that their condition had been intensified by their multiple births. I told one of my neighbors, “I’ve got to get somebody in here at night. I need to sleep.” I had postpartum depression. I was just a wreck. I couldn’t handle it. I didn’t know what to do. You’re just trying to get through it. You come out of this fog and you know there are services available but you don’t know you’ll need them until you’re in it. I knew I was having twins. I didn’t know what was going to happen once they got here. I thought, great, everything would be nice. It was just too much. (Jillian, mother of twins) My husband doesn’t love me anymore. He hates that I’ve gained all this weight. We went to counseling. Because of our finances we can’t separate now. Our marriage began to fall apart after the triplets were born. I never got enough sleep. I became depressed and thought about suicide. Now I’m on antidepressants. (Cam, mother of triplets)
Infertility Treatment Singleton and multiple birth mothers described lengthy attempts to become pregnant that typically accelerated from ovulation induction to IUI, culminating in IVF or IVF/ICSI. Although every participant was grateful she had been able to receive treatment and conceive, their most common treatment concerns were a perceived lack of follow-up for those whose treatment was limited to ovulation induction, and the timing of the decision about the number of eggs to be transferred for those undergoing assisted reproductive technologies. Massachusetts has the highest concentration of higher order multiples in the world and the stats are going up because of the flagrant use of ovulation enhancing drugs. Let’s be blunt. They prescribe them like candy. Some doctors give them and are not following up with ultrasounds. (Shiela, mother of twins) I have a girl in my triplet moms support group who didn’t know she was carrying triplets for 20 weeks. Because Clomid is supposed to have a very low risk of multiples they didn’t do an ultrasound. (Connie, mother of triplets)
As participants often recounted, “Clinicians give you those statistics and you think that your chance is small.” Thus, both patients’ and physicians’ desires for a live birth may inadvertently contribute to the continuing rise of iatrogenic multiple births. I had in vitro. I didn’t have to do selective reduction. I had four implanted and two had taken. But when I was in the operating room for the implantation, they said, “You’ve got two “B⫹’s” and these look like two “B’s.” I’m like, “Okay, so we’ll take those two.” He said, “You decide, you tell us
FERTILITY & STERILITY威
which ones you want. Do you want to do two? Do you want to do three? Do you want to try four?” Of course, I said, “I don’t know.” What a time to make the decision. (Jillian, mother of twins) Look where you are, though. You’re in the operating room. You’re vulnerable. (Nancy, mother of triplets)
Many respondents also felt there was a lack of congruency between their own and their physician’s goals. Some participants were willing to risk a multiple pregnancy by having more than one embryo transferred, but others felt that physicians were more likely to play the odds to increase clinic success rates. My husband and I had in vitro. Because my sister-in-law had already had twins, I told the doctor, “I only want one egg at a time.” When they implant they give you Valium to knock you out. He said, “All right, we’re going to put two in, because the odds are that you’re not going to get pregnant.” He talked my husband into doing it. We’d already discussed it. I only wanted one at a time; I didn’t want to take the chance. I conceived twins. Now we’re doing in vitro again. I told them, “Don’t you dare. Only one at a time or I will make you personally responsible. I’m not taking any chances.” (Susan, mother of twins)
The “physician–patient disconnect” that some participants described may be exacerbated by the fact that many fertility experts are men. Thus, there was concern that male physicians may be unaware of the long-term ramifications of their actions on family life, particularly for multiple-birth families or those whose children’s health and well-being are compromised by chronic illness or disabilities. I had triplets but I didn’t work once they were born, and only work part-time now. When my sister went through IVF and her doctor said, “We have three eggs. Do you want two or three?” She said, “I’ll take three. My sister has triplets. I can do it.” She and her husband work full-time. It’s been very difficult for her. I think it’s wrong to ask if they want two or three; they need to be counseled about the ramifications of having so many babies at once. The people that are doing the actual implantation have no idea what it’s like on the outside. (Nancy, mother of triplets) Most of them are men. (Jillian, mother of twins) They have no idea what life is like. They’re, “Oh, my god, I did it. I got the three in. She has three babies. She’s going to deliver.” Boom. Get discharged from the hospital. There’s no back-up services. Nothing. (Nancy)
However, one of the most critical issues in patients’ evaluations of their treatment experience and its outcome was the timing of the patient–physician dialogue about selective reduction. I walked out of the ultrasound where I was told I was having triplets, directly into the doctor’s office, where almost the first thing said to me was, “Should we reduce?” That hadn’t crossed my mind. I didn’t need to hear it the second after I learned I was having triplets. Who could make a rational decision at that point? I hadn’t even accepted the fact that I was having them. I
411
was a little offended. Give me some time to digest the information, and then I could deal with making the decision about reduction. (Alice, mother of triplets)
In contrast, another participant, who was referred to a perinatologist, felt she and her husband were given ample time and information to reach an informed decision congruent with their moral beliefs. No matter how I or anybody else feels morally about that subject, academically and emotionally it was great information to be given. Every bit of information possible was presented in an unemotional and very informational way. My husband and I did not share those conversations with anyone among our family and friends because we wanted to make sure our decision was totally ours. We didn’t need anyone else’s opinions or comments. (Shelly, mother of triplets)
DISCUSSION The focus group data indicated key quality-of-life domains that are particularly relevant to multiple-birth families. This qualitative research strategy revealed areas of concern that have not been focal points in the literature to date: social stigma, perinatal losses, potential positive marital outcomes, and the enduring impacts of the infertility experience. Each of these novel findings demonstrate the strengths of qualitative research strategies in eliciting the point of view and core concerns of an identified population. This is particularly useful when there are insufficient data to generate meaningful hypothesis, or to further illustrate and elucidate quantified outcomes. This study’s most significant limitation is its size, a common limitation of qualitative research. However, this small nonrepresentative sample revealed novel findings. While qualitative research strategies may not yield statistically significant data, they may provide theoretical significance and extend our understanding of a particular phenomenon. Another limitation of this study is that the sample was skewed toward triplets and higher order multiples. This overrepresentation may be advantageous, as birth rates for iatrogenic triplets and higher order multiples have increased the most dramatically over the past 30 years (2). A final limitation is that focus groups have a tendency toward consensus (24), an effect that was diminished by purposively eliciting contradictory responses during the group sessions. The social stigma that multiple-birth mothers experience was the most novel finding of this study. This suggests that in the United States multiple births have become equated with infertility and assisted reproduction. Therefore, regardless of the etiology of their conception, multiple-birth mothers become vulnerable to public evaluations of their fertility status and medical histories. The social interactions the participants described, of feeling devalued, different, or morally judged, underscore the need to prepare patients to effectively cope with the social 412
Ellison and Hall
Quality-of-Life: Multiple birth families
stigma they may experience. These interactions also illuminate the lack of public understanding about the medical condition of infertility and its psychosocial consequences. Public information campaigns could increase public awareness of infertility and its consequences, demystify assisted reproduction, and clarify that treatment may help people achieve what most of the fertile population takes for granted, a family. The compounded pregnancy losses this study identified were particularly salient given that participants felt these experiences were frequently minimized or overlooked; also important are the potential long-term psychosocial implications these losses may have on families. Early IVF pregnancy losses are associated with the greater likelihood of a subsequent full-term pregnancy (32), but the psychological toll associated with pregnancy losses has been documented (33, 34). Thus, involuntary losses, such as miscarriages, stillbirths, or neonatal deaths, increase women’s psychosocial risks, including increased vulnerability to future major depression (35, 36). Selective reduction may also increase women’s psychosocial vulnerability. Younger women, who are more likely to conceive a multiple gestation if two or more embryos are transferred (37), report poorer psychosocial outcomes following selective reduction (38). Other factors that influence poor outcomes, and thus could be assessed during treatment, are high levels of religiosity and growing up in or desiring a large family (38). Yet for most women the long-term impacts of selective reduction, similar to those associated with elective abortion, appear to be transient (38). This may be due to the degree of volition involved in both procedures. Another unexpected finding was the heterogeneity of the impact of multiple births on marital satisfaction and stability. Anecdotal reports suggest that multiple births result in higher rates of divorce and marital dissatisfaction (39). In this current study, participants described the impact of multiple births on their marriage with the following words: “boom,” “screeching,” “bombarded,” “havoc,” and “kabam.” This conveys the implosion of marriage and family life in the initial wake of a multiple birth. It also vividly describes the aftermath that couples face as they struggle to maintain their marital ties amid the inordinate demands of raising multiple-birth infants. However, the extremity and unremitting nature of these stressors may lead couples to renegotiate their relationship and roles, which can result in increased marital satisfaction and stability. This study suggests that a husband’s or partner’s willingness to share in household and childcare activities may be a significant factor in stabilizing marriages after the birth of multiple children. This finding is at odds with a recent study that found women’s marital satisfaction was more dependent on their spouse’s emotional support (40). Thus, the increased need for paternal participation in houseVol. 80, No. 2, August 2003
hold and childcare activities may indicate a unique need for multiple-birth mothers.
decision that they could live with and incorporate into their own religious and moral beliefs, as well as their personal histories (46).
Multiple births also present dilemmas for physicians in the United States, who face the double jeopardy of providing fertility treatment in a market-driven medical environment that is marked by competition for success rates and a lack of universal health care (41). Even with the refinement of assisted reproductive techniques (42) and physicians’ willingness to reduce the number of embryos transferred (43), the rate of twin births in the United States is still double (28 per 1000) that of countries with nationalized coverage for fertility treatment such as Sweden, England, and Australia (14 per 1000).
Patients who are well informed about multiple births and selective reduction before treatment begins may be better able to take the time they need to make their decisions, and feel that they can do so in partnership with their physicians. This may assuage the feeling some participants described of being pressured to reduce, or being pushed to make a decision prematurely. In addition, the continued controversy among treatment providers (41, 47–51) regarding practice guidelines for ovulation induction continues to put families at risk for multiple births.
This suggests that U.S. clinics and physicians can still further reduce iatrogenic multiple births. Given the inadequate information patients often have regarding medical treatment and its outcomes (44, 45), the timing of patient– physician dialogues and decision making appears to be critical. The candid stories that participants shared indicate that well-timed physician–patient dialogues, that increase a sense of partnership, might further decrease multiple births while improving patient evaluations of treatment outcomes.
In conclusion, this study’s qualitative approach minimized a priori assumptions about the impact of multiple births on family quality of life. This strategy resulted in novel findings. Its results extend our understanding of the ramifications of a common treatment outcome. The key findings were the social stigma and compounded pregnancy losses associated with multiple births. Another unexpected finding was the potential increase in marital stability as parents cope with the stresses of multiple births.
Participants reported that their sense of invincibility and desire for a child made it easy for them to discount statistics about their potential outcomes. This tendency might be countered through frank discussions about the maternal– child health risks and the social risks of multiple births, the realities of selective reduction, and the need to limit the number of embryos transferred or cancel cycles. These discussions may enhance patients’ informed decision making and decrease the perception of a patient–physician disconnect.
As anticipated, children’s health, unmet family needs, maternal depression, and parental stress were also key areas of concern, which supports previous studies on multiplebirth families. Finally, the infertility experience had a lasting impact. Participants’ evaluations of their fertility treatment identified several means of improving patient–physician interactions. This study provides critical data on quality-of-life concerns for multiple-birth families and lays the groundwork for further quantitative and qualitative study of this unique subpopulation.
Moreover, while physicians may try to avoid paternalistic, unilateral decision making, the time and place of shared and informed decision making appears to be critical. For example, the participants felt it was inappropriate that they were asked to make decisions about the number of embryos to be transferred during the transfer itself, while they were particularly vulnerable. In addition, the participants indicated that they needed adequate time to process information. For instance, this study’s findings indicate that the diagnosis of a multiple gestation and the possibility of selective reduction represent separate issues for patients. The participants needed time to process their diagnosis before discussing the realities, as opposed to the theoretical possibilities, of selective reduction. Furthermore, within the cultural context of the protracted abortion controversy in the United States, selective reduction also increases a couple’s vulnerability to social stigma. Thus, selective reduction may create an even more poignant moral dilemma for patients. This was evident in participants’ avid assertions that they needed time and information to make a FERTILITY & STERILITY威
Acknowledgments: The authors thank the women who participated in the focus groups for their candid stories and insights. The authors would also like to thank Triplets Moms and More, and RESOLVE for making their group members aware of this study. In addition, the authors thank Janet Rich-Edwards, Ph.D., Harvard Pilgrim Health Care, Harvard Medical School, for her work in some of the initial coding of the focus group data. Finally, the authors thank the Harvard Medical School’s Center of Excellence in Women’s Health for the grant from the Fund for Women’s Health that funded this study, and the NIH for Dr. Ellison’s postdoctoral fellowship grant (T32HD 07396).
References 1. Martin JA, Hamilton BE, Ventura SJ, Menacker F, Park MM. Births: final data for 2000. Natl Vital Stat Rep 2000;50:1–24. 2. Ventura SJ, Martin JA, Curtin SC, Mathews TJ, Park MM. Births: final data for 1998. Natl Vital Stat Rep 2000;48:1–17. 3. Callahan TL, Hall JE, Ettner SL, Christiansen CL, Greene MF, Crowley WF Jr. The economic impact of multiple-gestation pregnancies and the contribution of assisted reproductive techniques to their incidence. N Engl J Med 1994;331:244 –9.
413
4. Schieve LA, Jeng G, Wilcox L. Use of assisted reproductive technology—United States, 1996 and 1998. Morb Mortal Wkly Rep 2002;51: 97–101. 5. Schieve LA, Meikle SF, Ferre C, Peterson HB, Jeng G, Wilcox LS. Low and very low birth weight in infants conceived with use of assisted reproductive technology. N Engl J Med 2002;346:731–7. 6. Hansen M, Kurinczuk JJ, Bower C, Webb S. The risk of major birth defects after intracytoplasmic sperm injection and in vitro fertilization. N Engl J Med 2002;346:725–30. 7. Stro¨ mberg B, Dahlquist G, Ericson A, Finnstro¨ m O, Ko¨ ster M, Stjernqvist K. Neurological sequelae in children born after in-vitro fertilisation: a population-based study. Lancet 2002;359:461–5. 8. Tarlatzis BC, Grimbizis G. Pregnancy and child outcomes after assisted reproduction techniques. Hum Reprod 1999;14S:231–42. 9. Sattar N, Greer I. Pregnancy complications and maternal cardiovascular risk: opportunities for intervention and screening? BMJ 2002;325:157– 60. 10. Lynch A, McDuffie R, Murphy J, Faber K, Orleans M. Preeclampsia in multiple gestations: role of assisted reproductive technologies. Obstet Gynecol 2002;99:445–51. 11. Golombok S, Cook R, Bish A, Murray C. Families created by the new reproductive technologies: quality of parenting and social and emotional development of the children. Child Dev 1995;66:285–8. 12. Golombok S, Brewaeys A, Cook R, Giavazzi MT, Guerra D, Dexeus S, et al. European study of assisted reproduction families: family functioning and child development. Hum Reprod 1996;11:2324 –31. 13. Golombok S, MacCallum F, Goodman E. The “test-tube” generation: parent-child relationships and the psychological well-being of in vitro fertilization children at adolescence. Child Dev 2001;72:599 –608. 14. Golombok S, Brewaeys A, Giavazzi MT, Guerra D, MacCallum F, Rust J. The European study of assisted reproduction families: the transition to adolescence. Hum Reprod 2002;17:830 –40. 15. McMahon CA, Ungerer JA, Tennant C, Saunders D. Psychosocial adjustment and the quality of the mother-child relationship at four months postpartum after conception by in vitro fertilization. Fertil Steril 1997;68:492–500. 16. Gibson FL, Ungerer JA, Tennant CC, Saunders DM. Parental adjustment and attitudes to parenting after in vitro fertilization. Fertil Steril 2000;73:565–74. 17. Cook R, Bradley S, Golombok S. A preliminary study of parental stress and child behavior in families with twins conceived by in-vitro fertilization. Hum Reprod 1998;13:3244 –6. 18. Colpin HL, Vandemeulebroecke L. Parenting stress and psychosocial well-being among parents with twins conceived naturally or by reproductive technology. Hum Reprod 1999;14:3133–7. 19. Thorpe K, Golding J, MacGillivray I, Greenwood R. Comparison of prevalence of depression in mothers of twins and mothers of singletons. BMJ 1991;302:875–7. 20. Robin M, Bydlowski M, Cahen F, Josse D. Maternal reactions to the birth of triplets. Acta Genet Med Gemellol (Roma) 1991;40:41–51. 21. Garel M, Salobir C, Blondel B. Psychological consequences of having triplets: a 4-year follow-up study. Fertil Steril 1997;67:1162–65. 22. Bartel DA, Engler AJ, Natale JE, Misra V, Lewin A, Joseph JG. Working with families of suddenly and critically ill children: physician perspectives. Arch Pediatr Adolesc Med 2000;154:1127–33. 23. Anderson RT, Barbara AM, Weisman C, Hudson Scholle S, Binko J, Gwinner V, et al. A qualitative analysis of women’s satisfaction with primary care from a panel of focus groups in the National Centers of Excellence in Women’s Health. J Womens Health Gend Based Med 2001;10:637–47. 24. Ellison M. Authoritative knowledge and single women’s unintentional pregnancies, abortion, adoption and single motherhood: social stigma and structural violence. Med Anthropol Q 2003;17:in press. 25. Krueger RA. Focus groups: a practical guide for applied research, 2nd ed. Thousand Oaks, CA: Sage, 1994. 26. Strauss AL, Corbin J. Basics of qualitative research: techniques and procedures for developing grounded theory. Thousand Oaks, CA: Sage, 1998.
414
Ellison and Hall
Quality-of-Life: Multiple birth families
27. Cohen BB, Friedman DJ, Zhang Z, Trudeau EB, Walker DK, McKenna PA, et al. Impact of multiple births on low birthweight: Massachusetts, 1989 –1996. MMWR Morb Mortal Wkly Rep 1999;48:289 –92. 28. Stephen EH, Chandra A. Use of infertility services in the United States: 1995. Fam Plann Perspect 2000;32:132–7. 29. Goffman E. Stigma: notes on the management of spoiled identity. New York: Simon & Schuster, 1963. 30. Ellison MA. Disclosing selves/exposing culture: the gender work of single women’s crisis pregnancies—abortion, birthmothers, and single mothers. Ph.D. disseration, University of California, Los Angeles, 2000. 31. Centers for Disease Control. Live birth rates for ART cycles using fresh, nondonor eggs or embryos, by women’s age and history of miscarriage, among women with no previous births, 1999. Available online at http://www.cdc.gov/nccdphp/drh/ART99/images. 32. Bates J, Ginsburg ES. Early pregnancy loss in in vitro fertilization (IVF) is a positive predictor of subsequent IVF success. Fertil Steril 2002;77:337–41. 33. Shapiro CH. Infertility and pregnancy loss. San Francisco: Jossey-Bass, 1988. 34. Berson A. Quality-of-life issues for patients with reproductive loss. Clin Consult Obstet Gynecol 1994;6:100 –8. 35. Franche RL, Mikail SF. The impact of perinatal loss on adjustment to subsequent pregnancy. Soc Sci Med 1999;48:1613–23. 36. Bernazzani O, Bifulco A. Motherhood as a vulnerability factor in major depression: the role of negative pregnancy experiences. Soc Sci Med 2003;56:1249 – 60. 37. Schieve L, Peterson HB, Meidle SF, Jeng G, Danel I, Wilcox LS. Live-birth rates and multiple-birth risk using in vitro fertilization. JAMA 1999;282:1832–8. 38. Schreiner-Engel P, Walther VN, Mindes J, Lynch L, Berkowitz RL. First-trimester multifetal pregnancy reduction: acute and persistent psychologic reactions. Am J Obstet Gynecol 1995;172:541–7. 39. Malmstrom PM, Biale R. An agenda for meeting the special needs of multiple birth families. Acta Genet Med Gemellol (Roma) 1990;39: 507–14. 40. Erickson RJ. Reconceptualizing family work: the effect of emotion work on perceptions of marital quality. J Marriage Fam 1993;55:888 – 900. 41. Jones HW. Multiple births: how are we doing? Fertil Steril 2003;79: 17–21. 42. Cramer DW, Liberman RF, Powers D, Hornstein MD, McShane P, Barbieri RL. Recent trends in assisted reproductive techniques and associated outcomes. Obstet Gynecol 2000;95:61–6. 43. Hock DL, Seifer DB, Kontopoulos E, Ananth CV. Practice patterns among board-certified reproductive endocrinologists regarding highorder multiple gestations: a United States national survey. Obstet Gynecol 2002;99:763–70. 44. Deber RB, Kraetschmer N, Irvine J. What role do patients wish to play in treatment decision making? Arch Intern Med 1996;156:1414 –20. 45. Gleicher N, Campbell DP, Chan CL, Karande V, Rao R, Balin M, et al. The desire for multiple births in couples with infertility problems contradicts present practice patterns. Hum Reprod 1995;10:1079 –84. 46. Ginsburg FD. Contested lives: the abortion debate in an American community. Berkeley, CA: University of California Press, 1989. 47. Dickey RP. A year of inaction on high-order multiple pregnancies due to ovulation induction. Fertil Steril 2003;79:14 –6. 48. Fritz MA, Ory SJ. Practice guidelines cannot be justified in the absence of sufficient evidence: inaction is far more appropriate than indefensible action. Fertil Steril 2003;79:22–4. 49. Rosenwaks Z, Chung PH. High-order multiple pregnancy: is it a matter of inaction or a consequence of practice patterns? Fertil Steril 2003; 79:25–6. 50. Daya S. Inappropriate use of cohort studies to formulate treatment guidelines. Fertil Steril 2003;79:27. 51. Dickey RP. It has really been 15 years of inaction on high-order multiple pregnancies due to ovulation induction. Fertil Steril 2003;79: 28 –9.
Vol. 80, No. 2, August 2003
Social stigma and compounded losses: quality-of-life issues for multiple-birth families Marcia A. Ellison, Ph.D., and Janet E. Hall, M.D. Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts
Received August 19, 2002; revised and accepted March 8, 2003. Supported by the National Institutes of Health, Bethesda, Maryland (T32HD 07396), and Harvard Medical School’s Center of Excellence in Women’s Health Fund, Boston, Massachusetts (213-98-0009). Presented at the Harvard Medical School’s Center of Excellence in Women’s Health, Women’s Health Research Conference, Boston, Massachusetts, October 9, 2002. Reprint requests: Marcia A. Ellison, Ph.D., Reproductive Endocrine Unit, Massachusetts General Hospital, 55 Fruit Street, Bartlett Hall Ext. 5, Boston, Massachusetts 02114-2696 (FAX: 617-726-5357; E-mail: [email protected]. 0015-0282/03/$30.00 doi:10.1016/S0015-0282(03) 00659-0
Objective: To determine the quality-of-life domains most impacted by multiple births. Design: Focus groups, qualitative research. Setting: Human volunteers in a medical research environment. Patient(s): Forty-three mothers, 29 raising multiple-birth children, 13 raising singletons, identified from random and convenience samples. Intervention(s): None. Main Outcome Measure(s): Maternal self-reports of the psychosocial sequelae of multiple or singleton births, based on qualitative data analysis of transcribed group discussions. Result(s): The quality-of-life domains that were most impacted by raising multiple birth children were social stigma, pregnancy loss, marital satisfaction, children’s health, unmet family needs, parenting stress, maternal depression, and the infertility experience. Conclusion(s): Qualitative methods identified two novel quality-of-life domains in iatrogenic multiple birth families: social stigma and compounded losses. An unexpected finding was the potential for increased marital solidification as parents coped with the inordinate stresses of multiple births. As anticipated, children’s health, unmet family needs, maternal depression, and parental stress were key areas of concern. In addition, the infertility experience had a lasting impact. These findings are significant, given that at least 38% of all assisted conceptions result in a multiple birth. This study lays the groundwork for further research on the impact of iatrogenic multiple births. (Fertil Steril威 2003;80:405–14. ©2003 by American Society for Reproductive Medicine.) Key Words: Multiple births, infertility, social stigma, qualitative research
Since the 1970s, twin birth rates in the United States have increased 55% (from 18.9 to 29.3 per 1,000), while rates of triplet and higher order multiple births increased 423% (from 37 to 193.5 per 100,000) (1, 2). At least 70% of these increases are the result of assisted reproduction (3– 4). Although the increased maternal– child health risks associated with assisted reproduction have been documented (5– 10), relatively little is known about the concomitant social risks that these families may face, particularly those raising multiple-birth children. The results of studies of singleton in vitro fertilization (IVF) families have produced contradictory results. Two longitudinal studies documented increased parental warmth and parental involvement among IVF families at the
initial study point (11, 12). However, this outcome appears to be associated with the infertility experience, as adoptive parents also scored higher on these indices than parents of naturally conceived children. At the follow-up study points, as the children transitioned to adolescence, IVF parents in the first study (11) reported greater enjoyment of their parental roles than parents of adopted or naturally conceived children. But some IVF parents were also more emotionally enmeshed, or overly involved, with their children (13). The other longitudinal study (12) found that the IVF parents were less sensitive to their now adolescent children than those with adopted or naturally conceived children. Yet at this time point IVF parents exhibited greater warmth than adoptive parents (14). 405
In contrast, two additional longitudinal studies found significant and persistent levels of psychosocial impairment among first-time IVF parents as compared with fertile controls, including lower parental self-esteem and an impaired sense of maternal competence. These families also perceived their children as more vulnerable and were more childfocused than the control families who had not experienced infertility (15, 16). Given these contradictory findings, it is difficult to determine how much of these outcomes is the result of the experience of infertility versus fertility treatment. The potential social risks for families who have faced infertility may be exacerbated by a multiple birth. A seminal study, that found increased parenting stress in families of IVF versus spontaneous twins, was confounded by parity status (17); most IVF mothers were primiparas, whereas mothers of spontaneous twins had already made their transition to parenthood. Yet a study that controlled for parity status also documented increased stress as well as decreased psychosocial well-being among parents of IVF versus spontaneous twins (18). Multiple births, independent of fertility treatment, present increased stressors for families. For example, studies indicate that the unremitting stresses of raising multiple-birth children may increase the risk of nonperinatal maternal depression. A study, conducted in the early 1970s, before the advent of iatrogenic multiple births, found that mothers of twins were more vulnerable to depression than mothers of closely spaced singleton births (19). Two studies that documented high rates of persistent maternal depression among triplet mothers are provocative. However, these studies did not include a control group and because of their small samples are not generalizable (20, 21). Thus, the role of assisted reproduction as an independent predictor of longterm psychosocial sequelae and the impact of multiple births on the family have yet to be determined.
MATERIALS AND METHODS The purpose of this study was to determine the domains of family quality-of-life that are most impacted by multiple births. Given the equivocal findings on multiple-birth family outcomes to date, it was important to determine the domains of family life that were most impacted by multiple births. Clearly, multiple birth mothers could provide insights and identify outcomes that might have been overlooked in previous studies. Thus, this study used focus groups to identify the quality-of-life domains that were most relevant to multiple-birth mothers. Qualitative research strategies, such as focus groups, are particularly well suited to elucidating the key concerns of a particular population (22–24).
cultures. The initial focus groups (n ⫽ 33) included mothers regardless of their fertility status (i.e., spontaneous versus assisted conceptions). The homogeneity of the groups, and thus the comfort of the participants, was enhanced by separating participants by birth type. To control for idiosyncratic group responses (25), two groups were conducted for each of the three birth types: [1] high-risk multiple births (ⱕ2,500 grams, or ⬍37 weeks’ gestation), [2] low-risk multiple births, and [3] singleton births. The mothers of singletons (n ⫽ 10) and low-risk multiplebirth children (n ⫽ 12) were recruited from a random sample of mothers raising children 4 to 5 or 9 to 10 years of age. This sample, identified from the State of Massachusetts Birth Registry, was restricted to children of normal birth weight and gestational age. As a result, to control for sample BIAs, we recruited high-risk multiple-birth mothers (n ⫽ 11) from a convenience sample of peer support groups; their children ranged from 1 to 5 years of age. Preliminary analysis of these six focus groups indicated that infertility remained central to women’s experiences. Therefore, to control for the infertility experience, two additional groups were limited to participants who had conceived through assisted conception. These participants, also identified from a convenience sample of peer support groups, were raising singleton (n ⫽ 4) or multiple birth children (n ⫽ 6), from 8 months to 11 years of age.
Focus Group Protocol All study protocols were reviewed and approved by the institutional review board at Massachusetts General Hospital. Potential participants received a letter or e-mail flyer that described the study and provided contact information. Respondents were screened by telephone to ensure that they met the study’s eligibility requirements (i.e., children’s conception and birth type, children’s age). Recruited participants were mailed a packet that included a cover letter that described the study and the focus group protocol, and two copies of an informed consent form. This gave all identified women time to review the study protocol and materials before attending the group session, thus increasing the voluntary nature of their participation. Signed consent was obtained before each group discussion.
Study Participants
Each session was held in a private conference room at Massachusetts General Hospital. Because of the potentially sensitive nature of the discussion topics, the groups were kept small and were conducted on a first-name-only basis. This protected the privacy of the participants and fostered a sense of trust. The initial series of semistructured discussions followed a schedule of topics derived from the literature, including family stresses, family needs, social programs, social support, children’s health, marital satisfaction, and impact on women’s sense of self.
Study eligibility was limited to mothers, as women continue to be the primary childcare givers in Euro-American
The two follow-up groups explored topics that emerged from the previous sessions, such as women’s evaluations of
406
Ellison and Hall
Quality-of-Life: Multiple birth families
Vol. 80, No. 2, August 2003
TABLE 1 Focus group demographics. Demographic Birth type Singletons Twins Triplets Quadruplets Maternal age at child birth 20–24 25–29 30–34 35–39 40⫹
Initial groups (n ⫽ 33)
Follow-up groups (n ⫽ 10)
30% (10) 24% (8) 40% (13) 6% (2)
33% (3) — 56% (6) 11% (1)
3% (1) 18% (6) 27% (9) 43% (14) 9% (3)
— 10% (1) 40% (4) 30% (3) 20% (2)
Ellison. Quality-of-Life: Multiple birth families. Fertil Steril 2003.
assisted reproductive medicine and their treatment outcomes. Each session, which lasted 2 to 3 hours, was audiotaped and transcribed verbatim, using pseudonyms to protect participant confidentiality.
Data Analysis The statistical package SPSS was used to manage and analyze the descriptive statistics. The narrative data were managed using a computerized qualitative data analysis package (QSR NUD*IST Vivo). As is advised in qualitative research, data analysis was ongoing throughout the data collection process, which focused the direction of subsequent discussions. The study investigators reviewed the transcripts to establish interrater agreement about the initially identified themes. Themes were systematically identified using the standard grounded theory procedures of open and axial coding (26). Open coding identified key themes, which were assigned an identification label or “code.” Each code was then examined separately for recurrent patterns or motifs, and to determine the spectrum of responses. Axial coding, the reaggregation of data conceptually rather than temporally or literally, was used to determine the links between each theme. These procedures identified core quality-of-life domains.
RESULTS Demographics The characteristics of the study participants by birth type and maternal age are presented in Table 1. The study (n ⫽ 43) comprised 29 multiple birth mothers and 13 singleton mothers. Over half (58%) of the participants had undergone assisted conception, of which 78% resulted in a multiple birth. Sixty percent of the participants in the initial six focus groups (n ⫽ 33) were primiparas. Their children ranged from FERTILITY & STERILITY威
1 to 11 years of age. Thirty-two of the mothers were white, one was Asian American; 32 were currently married, one was separated. All but three had a college degree (58%), and 33% had an advanced degree. Combined pretax household incomes ranged from at least $100,000 (42%), to $60,000 – 99,000 (29%), to under $60,000 (18%). In the two follow-up groups (n ⫽ 10), eight participants were white, one was African American, and one was Hispanic. All were currently married and living with their husbands, although two of the multiple-birth mothers mentioned a potential separation. All of the participants had completed a 4-year college degree; and 33% had an advanced degree. Reported pretax combined household incomes ranged from $100,000 or more (25%), to $80,000 –99,999 (38%), to $60,000 –79,999 (25%), to under $60,000 (12%). These demographics reflect state and national statistics of multiple-birth families, which tend to be white with high income and education levels (27, 28). Similar to national statistics, the multiple-birth mothers in this study reported significantly higher rates of low birth weight (P⬍.001), prematurity (P⬍.001), chronic children’s health problems (P⫽.003), and assisted conception (P⬍.001), than the singleton mothers (Fisher’s exact test, two-sided, P ⫽ .05; Fig. 1).
Qualitative Data Analysis Preliminary data analysis identified eight core domains of quality of life that were most impacted by multiple births: [1] social stigma, [2] pregnancy and neonatal losses, [3] marital satisfaction, [4] children’s health, [5] meeting family needs, [6] parenting stress, [7] maternal depression, and [8] the infertility treatment experience. Data illustrating each of these domains are presented. Two additional themes, children’s development and school placement issues, were not included in the final analysis, as they were beyond the scope of the current research focus. Infertility treatment issues continued to be relevant for both singleton and multiple-birth mothers, but the other identified quality-of-life domains were more marked in multiple-birth mothers. Therefore, unless noted otherwise, the quotations are typical and representative of multiple-birth mothers’ responses to a particular topic. Because informal speech is less concise than the written word, quotations have been edited to preserve their intended meaning. Identifying information, including names, has been changed to protect participant confidentiality.
Social Stigma A devalued or spoiled identity is the hallmark of social stigma. All multiple-birth mothers had experienced social stigma related to their fertility treatment. Social stigma may be perceived or enacted through social interactions. As an example, participants discussed at length how family and friends had asked unsolicited and insensitive questions about their fertility status. The focal point of these invasive ques407
FIGURE 1
FIGURE 2
Children’s health outcomes by birth type (n ⫽ 43): f ⫽ low birth weight; u ⫽ premature; f ⫽ chronic health problem; f ⫽ assisted conception.
Follow-up focus groups (n ⫽ 10). Pregnancy losses and neonatal deaths: f ⫽ miscarriages; f ⫽ stillbirths; f ⫽ neonatal death; f ⫽ selective reduction.
Ellison. Quality-of-Life: Multiple birth families. Fertil Steril 2003.
Ellison. Quality-of-Life: Multiple birth families. Fertil Steril 2003.
tions was their children’s conception, specifically whether it was “normal” or “natural,” and by extension, whether they and their families were normal or natural. It always surprised me that people feel very free to ask such a personal question. It took me back. “Were you using something?” That would be the question. My response became “cheap wine.” (Pamela, mother of twins) We really wanted kids and we had gone on Clomid. But the way that his parents and family think and the way that my family is, we didn’t want them to know. All the talk. I don’t want them looking at my kids like they have four heads. When we had the triplets we didn’t tell them about our treatment, but a family member asked, “What kind of drugs were they on?” (Ellen, mother of triplets)
One of the core features of social stigma is a discredited moral status (29). A number of participants described social interactions, often with strangers, that resulted in moral judgments. The punitive comments they described reflect an assumption that assisted conception breaches sacred or secular norms. Thus, fertility treatment and its consequences may be cast as moral violations of God or nature. Because my son has cerebral palsy people say things like, “That’s what you get for messing with Mother Nature.” (Laura, mother of triplets) A woman I didn’t know asked me very nicely if I had fertility treatment. When I said yes her response was, “How dare you mess around with God’s will. It’s not natural. If you weren’t getting pregnant on your own there was a reason.” (Tony, mother of twins).
they’ll be impressed. If I say no they’ll think there’s something wrong with me. My mother-in-law is much more comfortable since we lost three of our children. Now everything’s normal. Nobody has to know that I had IVF. Nobody has to know that I had multiples. For her this is the best thing that could have happened. Now we look like a normal family. (Terry, mother of quadruplets)
Pregnancy and Neonatal Losses Similar to the high rates of miscarriage among women seeking fertility treatment (31), 60% of the participants in the two follow-up groups that controlled for infertility (n ⫽ 6 out of 10) had experienced a pregnancy or neonatal loss (Fig. 2). I got to almost 27 weeks after pulmonary edema, three blood transfusions, and burning a hole in my esophagus from acid reflux. We thought we had licked it but I got an infection. It killed one of my children in utero. Right after we had passing ceremonies for her, my son had off the chart head bleeds. He lived about two weeks. One of the triplets survived. She has a form of CP but she’s the greatest kid. (Laurie, mother of triplets) I tried Clomid. It didn’t work. We tried IUI. That didn’t work. Then, our first in vitro worked. One of them split, so I had a singleton and a set of identical twins. One twin died in utero. The other had a brain bleed and has CP, but every day she does something new. The singleton survived; she’s huge. They’re great kids. (Michele, mother of triplets)
These losses created acute emotional needs that were not always recognized nor well met.
Another potential hallmark of social stigma is a “courtesy stigma” which may extend, like a contagion, to social intimates (29, 30). As a result, children conceived through assisted conception, as well as extended family members, may be vulnerable to social stigma.
Everything happened on weekends. I delivered at 25 weeks. The social worker asked if I wanted a chaplain to say a service for the baby that died. During the service he kept calling the baby the wrong name. After that the social worker said, “Well, it’s Friday and it’s a long weekend, so I’ll talk to you on Tuesday.” (Lynn, mother of triplets)
When I was pregnant with quads, everyone assumed I had ART. Some people would say, “Was it natural?” If I say yes
In addition, for women who have experienced infertility, selective reduction poses a poignant personal and moral
408
Ellison and Hall
Quality-of-Life: Multiple birth families
Vol. 80, No. 2, August 2003
dilemma. In this double-bind situation, a couple must make a decision regarding a voluntary medical procedure that requires them to sacrifice one fetus for the greater good of their surviving fetuses, while they risk losing their entire pregnancy. Not many people know this, but mine were in vitro. I went through all that. They used to implant sixteen eggs because they wanted to get good results. We didn’t think anything of it. We figured, hey, the more the merrier. We just want to be able to get pregnant. I got pregnant with five. (Wendy, mother of twins) Yeah, they’re playing the odds. Did you have selective reduction? (Sandy, mother of triplets) Nothing was selective about it but that’s the word they use. He really didn’t give me much of a choice. He said to me, “You should go to two.” He explained the ramifications. It was a hard decision because you’ve had infertility for eight or nine years and you have all these great kids inside of you and then you’re doing something that goes against all that. I thought, if I can give these two the chance, I had to go for that. (Wendy)
Moreover, selective reduction may be at odds with desires to have a larger family, or may conflict with patients’ religious or moral beliefs. Their personal histories, including their treatment experiences, may also strongly influence their decision. Thus, a number of participants resisted their doctors’ advice and carried a multifetal pregnancy to term. Once our first IUI wasn’t successful, we tried IVF and I miscarried. In the second IVF cycle I lost twins. The third cycle was quadruplets; that was just too many. They advised us to reduce. I couldn’t do that to one of my children. How do you decide which one? We terminated that cycle. The fourth cycle was our triplets. We have two embryos left; we’re donating them to research. (Mandy, mother of triplets)
Marital Impacts The impact of multiple births on a marriage varied considerably. Many participants described losing touch with their husband as the incessant demands of raising multiplebirth children engulfed their marriage. We’re definitely just passing in the night. He goes to bed a half-hour after he gets home, because he gets up so early. It’s hard to take the little time he spends with the family and take it all for myself. Maybe we both tend to put the children first. We tend to put our marriage on the back burner. (Randy, mother of twins)
Couples unable to equitably divide family and household labor, or who were unable to work together as a team, described the slow erosion of their marriage. Ever since my multiple pregnancy my husband can’t handle what happened to our marriage. Our perfect little family, we were going down this perfect little path and then we get bombarded with triplets. It wasn’t part of the plan. Having children was part of the plan but he can’t handle three at one time. (Victoria, mother of triplets)
FERTILITY & STERILITY威
We had the good life before kids. Money was not an issue. We took lots of vacations. With our first child it was okay. With the triplets our life came to a screeching halt. I was on bedrest. He had to take over our son’s and my care. Then we had to make it through NICU. Now that the children are toddlers he can’t handle the havoc on the house and the noise level after working 12 hours. They’re not quiet and bathed and in their jammies. As often as I tried, it’s not that way. It’s just too overwhelming for him. (Kate, mother of triplets)
In contrast, the inordinate stresses of raising multiplebirth infants strengthened some marriages. In the face of such unremitting stress, some participants concluded that they would need to become a team if their marriages were to survive. This kind of teamwork typically resulted in greater paternal participation in childcare and household tasks. Twins changed everything for us. My husband suddenly parented a lot more than he had with our first child. Caring for twin infants was really difficult. He’s probably a much better parent because we have twins. (Audrey, mother of twins) You learn to work as a team or you don’t survive. It becomes a team effort; to survive you really must. I think it made us closer because of the team effort. You find out whether you really care about each other or not. (Megan, mother of twins)
Children’s Health Children’s health problems ranged from minor fine motor skill problems or the difficulty of dealing with the dominoeffect of normal childhood illnesses, to severe chronic illnesses and disabilities, particularly among triplets and quadruplets. I love my children but I didn’t plan on having four kids at once and needing overnight nurses for children with special needs, like quadriplegia, feeding tubes, blindness, and permanent brain shunts. (Carol, mother of quadruplets)
In another group session a mother of healthy twins commented, What happens when these kids go home and they have CP, and they’re blind, and they have to be fed through a feeding tube? What services are there for these special needs families? (Megan, mother of twins)
Mothers raising high-risk multiple-birth children, whose children are most likely to have special needs, were vocal about their families’ unmet needs and long-term vulnerabilities. They noted that even in well-trained tertiary care settings, they were not always informed about available support services. Moreover, the needs and increased emotional vulnerability that often arise in families whose children’s health is fragile were not always recognized or attended to. I was in the hospital for two weeks before I delivered and for five days after. The social worker came to meet me as I was discharged and told me that she had me on her list to visit. Then, it was almost four months after we had been
409
taking two cars a day to visit our children in NICU that she told us about parking coupons for families with children in NICU. (Jane, mother of triplets) Our family is comfortable financially but when my daughter coded they took her by ambulance to the hospital. When we got there they kicked me out of the room. Four people carried me out. I said that it was in my Bill of Rights and that I did not have to leave my baby. If my child is going to die I want to be there. (Pam, mother of triplets)
Meeting Family Needs The birth of a child increases the needs of all families. However, these needs increase exponentially for families with twins, triplets, or more. This includes daily needs such as diapers and formula, as well as the need for more car seats, specialized strollers, and cars and homes large enough to accommodate an immediate influx of children. Maybe a lot of us decide to have two kids, three kids, eight kids. But it’s different when, boom, you have two at once. It’s really no different than your neighbor next door that has two kids. But now you have to spend $35 on formula as opposed to $17 for one child. Can you imagine what it costs for daycare, or to send your kids to private nursery school or preschool times two or three? (Megan, mother of twins) I had to move to a larger home. The three little ones were in the living room. I had to go from my Honda Civic to a Chevy Suburban. (Nancy, mother of triplets)
For multiple-birth parents, childcare expenses also increase exponentially. Even a night out for dinner and a movie may require finding, hiring, and paying two or three babysitters to care for multiple-birth children and any additional children in the household. Moreover, their childcare expenses may have already been exacerbated by weeks or months of full bedrest prescribed during a multiple pregnancy. I had to give up my job when I was pregnant with the triplets. Then my husband was laid off. We have huge hospital bills. We’ve had four ambulances to the emergency room in four weeks. We’re still in shellshock after losing one of our children less than a year ago. (Jane, mother of triplets)
Increases in childcare expenses may effectively resolve the dilemma many couples face about the trade-off of daycare versus a second income. However, while some participants felt they were fortunate to be stay-at-home mothers, for others giving up their career meant losing part of their identity and independence. I left my job. That was my thing; that was my time. Now my time is spent for everybody else, and not for me. Every now and then I can eke out time with a friend, but otherwise there’s nothing for me. My husband has his job; he can socialize at work. My social life, my world, is my kids. (Randy, mother of twins)
Parenting Stress Sleep deprivation was a ubiquitous complaint, but the other most significant stressor was managing the daily, re410
Ellison and Hall
Quality-of-Life: Multiple birth families
petitive routines of family life. Participants described the difficulty of the logistics and juggling that were required to meet their families’ needs and schedules. The biggest stresses are getting people where they need to go and finding time to do things. They still need help toileting themselves, the whole routine of meals and snacks and then cleaning up, getting everyone dressed and planning what we’re going to do, and then trying to do it. And in between getting the laundry and groceries done, paying bills. So when we do have money that’s not even the whole answer. It’s getting the time to do any of this. (Phyllis, mother of triplets) When they were newborns and toddlers, the logistics was very hard. You’re the only one with them, carrying them places, getting them in and out of cars, dressing them and getting them out at the same time. That was the hard part. You can’t do anything. You can’t go to the bathroom. You can’t eat. That was just for the first year or two. I think that’s the big difference to raising singletons. (Randy, mother of twins)
In addition, while one might assume that multiple births are more stressful for first-time parents, multiple births may also pose a unique stressor for families with previous children who reported struggling to meet the needs of their older children. It was hard to integrate the triplets into an existing family. Our daughter was an only child. My husband, who was also an only child, said, “I really think two is better. Let’s go for two.” The second ended up being triplets. For us it was having him for five years, and then bringing in what felt like this group. Kabam. It changes everything. Cribs everywhere. It was crazy. It was hard for us to be sensitive to how our first child was feeling about it. We each chose a name for one of them. (Phyllis, mother of triplets) Having my first-born and then having triplets, I sometimes feel guilty because the first one doesn’t get enough attention. I feel selfish that I wanted a sibling for the older child. Today my older son said to me, “You didn’t give me any attention today.” That hurts; that hurts a lot. (Victoria, mother of triplets)
Iatrogenic multiple births may generate another unique parenting stressor. Unlike mothers whose twins, triplets, or more were spontaneous, mothers who sought fertility treatment, although they did not ask for a multiple gestation, may feel that they cannot complain about being exhausted and overwhelmed. Infertility affects how you parent your children. I used to think, how could I complain? I was exhausted. I was working. But I would always smile because I drove everyone crazy for eight years trying to have kids. Who am I to start complaining? Then I read a book for multiple birth families that said, “It’s okay to complain because your issues are real. It may have taken a little bit longer than some other people to get pregnant but it’s okay to be tired. You don’t have to
Vol. 80, No. 2, August 2003
smile all the time about being up half the night.” (Megan, mother of twins)
Maternal Depression Although anecdotal evidence of high rates of depression among these mothers was discussed, few participants reported nonperinatal depression. However, a number of participants who experienced postnatal depression felt that their condition had been intensified by their multiple births. I told one of my neighbors, “I’ve got to get somebody in here at night. I need to sleep.” I had postpartum depression. I was just a wreck. I couldn’t handle it. I didn’t know what to do. You’re just trying to get through it. You come out of this fog and you know there are services available but you don’t know you’ll need them until you’re in it. I knew I was having twins. I didn’t know what was going to happen once they got here. I thought, great, everything would be nice. It was just too much. (Jillian, mother of twins) My husband doesn’t love me anymore. He hates that I’ve gained all this weight. We went to counseling. Because of our finances we can’t separate now. Our marriage began to fall apart after the triplets were born. I never got enough sleep. I became depressed and thought about suicide. Now I’m on antidepressants. (Cam, mother of triplets)
Infertility Treatment Singleton and multiple birth mothers described lengthy attempts to become pregnant that typically accelerated from ovulation induction to IUI, culminating in IVF or IVF/ICSI. Although every participant was grateful she had been able to receive treatment and conceive, their most common treatment concerns were a perceived lack of follow-up for those whose treatment was limited to ovulation induction, and the timing of the decision about the number of eggs to be transferred for those undergoing assisted reproductive technologies. Massachusetts has the highest concentration of higher order multiples in the world and the stats are going up because of the flagrant use of ovulation enhancing drugs. Let’s be blunt. They prescribe them like candy. Some doctors give them and are not following up with ultrasounds. (Shiela, mother of twins) I have a girl in my triplet moms support group who didn’t know she was carrying triplets for 20 weeks. Because Clomid is supposed to have a very low risk of multiples they didn’t do an ultrasound. (Connie, mother of triplets)
As participants often recounted, “Clinicians give you those statistics and you think that your chance is small.” Thus, both patients’ and physicians’ desires for a live birth may inadvertently contribute to the continuing rise of iatrogenic multiple births. I had in vitro. I didn’t have to do selective reduction. I had four implanted and two had taken. But when I was in the operating room for the implantation, they said, “You’ve got two “B⫹’s” and these look like two “B’s.” I’m like, “Okay, so we’ll take those two.” He said, “You decide, you tell us
FERTILITY & STERILITY威
which ones you want. Do you want to do two? Do you want to do three? Do you want to try four?” Of course, I said, “I don’t know.” What a time to make the decision. (Jillian, mother of twins) Look where you are, though. You’re in the operating room. You’re vulnerable. (Nancy, mother of triplets)
Many respondents also felt there was a lack of congruency between their own and their physician’s goals. Some participants were willing to risk a multiple pregnancy by having more than one embryo transferred, but others felt that physicians were more likely to play the odds to increase clinic success rates. My husband and I had in vitro. Because my sister-in-law had already had twins, I told the doctor, “I only want one egg at a time.” When they implant they give you Valium to knock you out. He said, “All right, we’re going to put two in, because the odds are that you’re not going to get pregnant.” He talked my husband into doing it. We’d already discussed it. I only wanted one at a time; I didn’t want to take the chance. I conceived twins. Now we’re doing in vitro again. I told them, “Don’t you dare. Only one at a time or I will make you personally responsible. I’m not taking any chances.” (Susan, mother of twins)
The “physician–patient disconnect” that some participants described may be exacerbated by the fact that many fertility experts are men. Thus, there was concern that male physicians may be unaware of the long-term ramifications of their actions on family life, particularly for multiple-birth families or those whose children’s health and well-being are compromised by chronic illness or disabilities. I had triplets but I didn’t work once they were born, and only work part-time now. When my sister went through IVF and her doctor said, “We have three eggs. Do you want two or three?” She said, “I’ll take three. My sister has triplets. I can do it.” She and her husband work full-time. It’s been very difficult for her. I think it’s wrong to ask if they want two or three; they need to be counseled about the ramifications of having so many babies at once. The people that are doing the actual implantation have no idea what it’s like on the outside. (Nancy, mother of triplets) Most of them are men. (Jillian, mother of twins) They have no idea what life is like. They’re, “Oh, my god, I did it. I got the three in. She has three babies. She’s going to deliver.” Boom. Get discharged from the hospital. There’s no back-up services. Nothing. (Nancy)
However, one of the most critical issues in patients’ evaluations of their treatment experience and its outcome was the timing of the patient–physician dialogue about selective reduction. I walked out of the ultrasound where I was told I was having triplets, directly into the doctor’s office, where almost the first thing said to me was, “Should we reduce?” That hadn’t crossed my mind. I didn’t need to hear it the second after I learned I was having triplets. Who could make a rational decision at that point? I hadn’t even accepted the fact that I was having them. I
411
was a little offended. Give me some time to digest the information, and then I could deal with making the decision about reduction. (Alice, mother of triplets)
In contrast, another participant, who was referred to a perinatologist, felt she and her husband were given ample time and information to reach an informed decision congruent with their moral beliefs. No matter how I or anybody else feels morally about that subject, academically and emotionally it was great information to be given. Every bit of information possible was presented in an unemotional and very informational way. My husband and I did not share those conversations with anyone among our family and friends because we wanted to make sure our decision was totally ours. We didn’t need anyone else’s opinions or comments. (Shelly, mother of triplets)
DISCUSSION The focus group data indicated key quality-of-life domains that are particularly relevant to multiple-birth families. This qualitative research strategy revealed areas of concern that have not been focal points in the literature to date: social stigma, perinatal losses, potential positive marital outcomes, and the enduring impacts of the infertility experience. Each of these novel findings demonstrate the strengths of qualitative research strategies in eliciting the point of view and core concerns of an identified population. This is particularly useful when there are insufficient data to generate meaningful hypothesis, or to further illustrate and elucidate quantified outcomes. This study’s most significant limitation is its size, a common limitation of qualitative research. However, this small nonrepresentative sample revealed novel findings. While qualitative research strategies may not yield statistically significant data, they may provide theoretical significance and extend our understanding of a particular phenomenon. Another limitation of this study is that the sample was skewed toward triplets and higher order multiples. This overrepresentation may be advantageous, as birth rates for iatrogenic triplets and higher order multiples have increased the most dramatically over the past 30 years (2). A final limitation is that focus groups have a tendency toward consensus (24), an effect that was diminished by purposively eliciting contradictory responses during the group sessions. The social stigma that multiple-birth mothers experience was the most novel finding of this study. This suggests that in the United States multiple births have become equated with infertility and assisted reproduction. Therefore, regardless of the etiology of their conception, multiple-birth mothers become vulnerable to public evaluations of their fertility status and medical histories. The social interactions the participants described, of feeling devalued, different, or morally judged, underscore the need to prepare patients to effectively cope with the social 412
Ellison and Hall
Quality-of-Life: Multiple birth families
stigma they may experience. These interactions also illuminate the lack of public understanding about the medical condition of infertility and its psychosocial consequences. Public information campaigns could increase public awareness of infertility and its consequences, demystify assisted reproduction, and clarify that treatment may help people achieve what most of the fertile population takes for granted, a family. The compounded pregnancy losses this study identified were particularly salient given that participants felt these experiences were frequently minimized or overlooked; also important are the potential long-term psychosocial implications these losses may have on families. Early IVF pregnancy losses are associated with the greater likelihood of a subsequent full-term pregnancy (32), but the psychological toll associated with pregnancy losses has been documented (33, 34). Thus, involuntary losses, such as miscarriages, stillbirths, or neonatal deaths, increase women’s psychosocial risks, including increased vulnerability to future major depression (35, 36). Selective reduction may also increase women’s psychosocial vulnerability. Younger women, who are more likely to conceive a multiple gestation if two or more embryos are transferred (37), report poorer psychosocial outcomes following selective reduction (38). Other factors that influence poor outcomes, and thus could be assessed during treatment, are high levels of religiosity and growing up in or desiring a large family (38). Yet for most women the long-term impacts of selective reduction, similar to those associated with elective abortion, appear to be transient (38). This may be due to the degree of volition involved in both procedures. Another unexpected finding was the heterogeneity of the impact of multiple births on marital satisfaction and stability. Anecdotal reports suggest that multiple births result in higher rates of divorce and marital dissatisfaction (39). In this current study, participants described the impact of multiple births on their marriage with the following words: “boom,” “screeching,” “bombarded,” “havoc,” and “kabam.” This conveys the implosion of marriage and family life in the initial wake of a multiple birth. It also vividly describes the aftermath that couples face as they struggle to maintain their marital ties amid the inordinate demands of raising multiple-birth infants. However, the extremity and unremitting nature of these stressors may lead couples to renegotiate their relationship and roles, which can result in increased marital satisfaction and stability. This study suggests that a husband’s or partner’s willingness to share in household and childcare activities may be a significant factor in stabilizing marriages after the birth of multiple children. This finding is at odds with a recent study that found women’s marital satisfaction was more dependent on their spouse’s emotional support (40). Thus, the increased need for paternal participation in houseVol. 80, No. 2, August 2003
hold and childcare activities may indicate a unique need for multiple-birth mothers.
decision that they could live with and incorporate into their own religious and moral beliefs, as well as their personal histories (46).
Multiple births also present dilemmas for physicians in the United States, who face the double jeopardy of providing fertility treatment in a market-driven medical environment that is marked by competition for success rates and a lack of universal health care (41). Even with the refinement of assisted reproductive techniques (42) and physicians’ willingness to reduce the number of embryos transferred (43), the rate of twin births in the United States is still double (28 per 1000) that of countries with nationalized coverage for fertility treatment such as Sweden, England, and Australia (14 per 1000).
Patients who are well informed about multiple births and selective reduction before treatment begins may be better able to take the time they need to make their decisions, and feel that they can do so in partnership with their physicians. This may assuage the feeling some participants described of being pressured to reduce, or being pushed to make a decision prematurely. In addition, the continued controversy among treatment providers (41, 47–51) regarding practice guidelines for ovulation induction continues to put families at risk for multiple births.
This suggests that U.S. clinics and physicians can still further reduce iatrogenic multiple births. Given the inadequate information patients often have regarding medical treatment and its outcomes (44, 45), the timing of patient– physician dialogues and decision making appears to be critical. The candid stories that participants shared indicate that well-timed physician–patient dialogues, that increase a sense of partnership, might further decrease multiple births while improving patient evaluations of treatment outcomes.
In conclusion, this study’s qualitative approach minimized a priori assumptions about the impact of multiple births on family quality of life. This strategy resulted in novel findings. Its results extend our understanding of the ramifications of a common treatment outcome. The key findings were the social stigma and compounded pregnancy losses associated with multiple births. Another unexpected finding was the potential increase in marital stability as parents cope with the stresses of multiple births.
Participants reported that their sense of invincibility and desire for a child made it easy for them to discount statistics about their potential outcomes. This tendency might be countered through frank discussions about the maternal– child health risks and the social risks of multiple births, the realities of selective reduction, and the need to limit the number of embryos transferred or cancel cycles. These discussions may enhance patients’ informed decision making and decrease the perception of a patient–physician disconnect.
As anticipated, children’s health, unmet family needs, maternal depression, and parental stress were also key areas of concern, which supports previous studies on multiplebirth families. Finally, the infertility experience had a lasting impact. Participants’ evaluations of their fertility treatment identified several means of improving patient–physician interactions. This study provides critical data on quality-of-life concerns for multiple-birth families and lays the groundwork for further quantitative and qualitative study of this unique subpopulation.
Moreover, while physicians may try to avoid paternalistic, unilateral decision making, the time and place of shared and informed decision making appears to be critical. For example, the participants felt it was inappropriate that they were asked to make decisions about the number of embryos to be transferred during the transfer itself, while they were particularly vulnerable. In addition, the participants indicated that they needed adequate time to process information. For instance, this study’s findings indicate that the diagnosis of a multiple gestation and the possibility of selective reduction represent separate issues for patients. The participants needed time to process their diagnosis before discussing the realities, as opposed to the theoretical possibilities, of selective reduction. Furthermore, within the cultural context of the protracted abortion controversy in the United States, selective reduction also increases a couple’s vulnerability to social stigma. Thus, selective reduction may create an even more poignant moral dilemma for patients. This was evident in participants’ avid assertions that they needed time and information to make a FERTILITY & STERILITY威
Acknowledgments: The authors thank the women who participated in the focus groups for their candid stories and insights. The authors would also like to thank Triplets Moms and More, and RESOLVE for making their group members aware of this study. In addition, the authors thank Janet Rich-Edwards, Ph.D., Harvard Pilgrim Health Care, Harvard Medical School, for her work in some of the initial coding of the focus group data. Finally, the authors thank the Harvard Medical School’s Center of Excellence in Women’s Health for the grant from the Fund for Women’s Health that funded this study, and the NIH for Dr. Ellison’s postdoctoral fellowship grant (T32HD 07396).
References 1. Martin JA, Hamilton BE, Ventura SJ, Menacker F, Park MM. Births: final data for 2000. Natl Vital Stat Rep 2000;50:1–24. 2. Ventura SJ, Martin JA, Curtin SC, Mathews TJ, Park MM. Births: final data for 1998. Natl Vital Stat Rep 2000;48:1–17. 3. Callahan TL, Hall JE, Ettner SL, Christiansen CL, Greene MF, Crowley WF Jr. The economic impact of multiple-gestation pregnancies and the contribution of assisted reproductive techniques to their incidence. N Engl J Med 1994;331:244 –9.
413
4. Schieve LA, Jeng G, Wilcox L. Use of assisted reproductive technology—United States, 1996 and 1998. Morb Mortal Wkly Rep 2002;51: 97–101. 5. Schieve LA, Meikle SF, Ferre C, Peterson HB, Jeng G, Wilcox LS. Low and very low birth weight in infants conceived with use of assisted reproductive technology. N Engl J Med 2002;346:731–7. 6. Hansen M, Kurinczuk JJ, Bower C, Webb S. The risk of major birth defects after intracytoplasmic sperm injection and in vitro fertilization. N Engl J Med 2002;346:725–30. 7. Stro¨ mberg B, Dahlquist G, Ericson A, Finnstro¨ m O, Ko¨ ster M, Stjernqvist K. Neurological sequelae in children born after in-vitro fertilisation: a population-based study. Lancet 2002;359:461–5. 8. Tarlatzis BC, Grimbizis G. Pregnancy and child outcomes after assisted reproduction techniques. Hum Reprod 1999;14S:231–42. 9. Sattar N, Greer I. Pregnancy complications and maternal cardiovascular risk: opportunities for intervention and screening? BMJ 2002;325:157– 60. 10. Lynch A, McDuffie R, Murphy J, Faber K, Orleans M. Preeclampsia in multiple gestations: role of assisted reproductive technologies. Obstet Gynecol 2002;99:445–51. 11. Golombok S, Cook R, Bish A, Murray C. Families created by the new reproductive technologies: quality of parenting and social and emotional development of the children. Child Dev 1995;66:285–8. 12. Golombok S, Brewaeys A, Cook R, Giavazzi MT, Guerra D, Dexeus S, et al. European study of assisted reproduction families: family functioning and child development. Hum Reprod 1996;11:2324 –31. 13. Golombok S, MacCallum F, Goodman E. The “test-tube” generation: parent-child relationships and the psychological well-being of in vitro fertilization children at adolescence. Child Dev 2001;72:599 –608. 14. Golombok S, Brewaeys A, Giavazzi MT, Guerra D, MacCallum F, Rust J. The European study of assisted reproduction families: the transition to adolescence. Hum Reprod 2002;17:830 –40. 15. McMahon CA, Ungerer JA, Tennant C, Saunders D. Psychosocial adjustment and the quality of the mother-child relationship at four months postpartum after conception by in vitro fertilization. Fertil Steril 1997;68:492–500. 16. Gibson FL, Ungerer JA, Tennant CC, Saunders DM. Parental adjustment and attitudes to parenting after in vitro fertilization. Fertil Steril 2000;73:565–74. 17. Cook R, Bradley S, Golombok S. A preliminary study of parental stress and child behavior in families with twins conceived by in-vitro fertilization. Hum Reprod 1998;13:3244 –6. 18. Colpin HL, Vandemeulebroecke L. Parenting stress and psychosocial well-being among parents with twins conceived naturally or by reproductive technology. Hum Reprod 1999;14:3133–7. 19. Thorpe K, Golding J, MacGillivray I, Greenwood R. Comparison of prevalence of depression in mothers of twins and mothers of singletons. BMJ 1991;302:875–7. 20. Robin M, Bydlowski M, Cahen F, Josse D. Maternal reactions to the birth of triplets. Acta Genet Med Gemellol (Roma) 1991;40:41–51. 21. Garel M, Salobir C, Blondel B. Psychological consequences of having triplets: a 4-year follow-up study. Fertil Steril 1997;67:1162–65. 22. Bartel DA, Engler AJ, Natale JE, Misra V, Lewin A, Joseph JG. Working with families of suddenly and critically ill children: physician perspectives. Arch Pediatr Adolesc Med 2000;154:1127–33. 23. Anderson RT, Barbara AM, Weisman C, Hudson Scholle S, Binko J, Gwinner V, et al. A qualitative analysis of women’s satisfaction with primary care from a panel of focus groups in the National Centers of Excellence in Women’s Health. J Womens Health Gend Based Med 2001;10:637–47. 24. Ellison M. Authoritative knowledge and single women’s unintentional pregnancies, abortion, adoption and single motherhood: social stigma and structural violence. Med Anthropol Q 2003;17:in press. 25. Krueger RA. Focus groups: a practical guide for applied research, 2nd ed. Thousand Oaks, CA: Sage, 1994. 26. Strauss AL, Corbin J. Basics of qualitative research: techniques and procedures for developing grounded theory. Thousand Oaks, CA: Sage, 1998.
414
Ellison and Hall
Quality-of-Life: Multiple birth families
27. Cohen BB, Friedman DJ, Zhang Z, Trudeau EB, Walker DK, McKenna PA, et al. Impact of multiple births on low birthweight: Massachusetts, 1989 –1996. MMWR Morb Mortal Wkly Rep 1999;48:289 –92. 28. Stephen EH, Chandra A. Use of infertility services in the United States: 1995. Fam Plann Perspect 2000;32:132–7. 29. Goffman E. Stigma: notes on the management of spoiled identity. New York: Simon & Schuster, 1963. 30. Ellison MA. Disclosing selves/exposing culture: the gender work of single women’s crisis pregnancies—abortion, birthmothers, and single mothers. Ph.D. disseration, University of California, Los Angeles, 2000. 31. Centers for Disease Control. Live birth rates for ART cycles using fresh, nondonor eggs or embryos, by women’s age and history of miscarriage, among women with no previous births, 1999. Available online at http://www.cdc.gov/nccdphp/drh/ART99/images. 32. Bates J, Ginsburg ES. Early pregnancy loss in in vitro fertilization (IVF) is a positive predictor of subsequent IVF success. Fertil Steril 2002;77:337–41. 33. Shapiro CH. Infertility and pregnancy loss. San Francisco: Jossey-Bass, 1988. 34. Berson A. Quality-of-life issues for patients with reproductive loss. Clin Consult Obstet Gynecol 1994;6:100 –8. 35. Franche RL, Mikail SF. The impact of perinatal loss on adjustment to subsequent pregnancy. Soc Sci Med 1999;48:1613–23. 36. Bernazzani O, Bifulco A. Motherhood as a vulnerability factor in major depression: the role of negative pregnancy experiences. Soc Sci Med 2003;56:1249 – 60. 37. Schieve L, Peterson HB, Meidle SF, Jeng G, Danel I, Wilcox LS. Live-birth rates and multiple-birth risk using in vitro fertilization. JAMA 1999;282:1832–8. 38. Schreiner-Engel P, Walther VN, Mindes J, Lynch L, Berkowitz RL. First-trimester multifetal pregnancy reduction: acute and persistent psychologic reactions. Am J Obstet Gynecol 1995;172:541–7. 39. Malmstrom PM, Biale R. An agenda for meeting the special needs of multiple birth families. Acta Genet Med Gemellol (Roma) 1990;39: 507–14. 40. Erickson RJ. Reconceptualizing family work: the effect of emotion work on perceptions of marital quality. J Marriage Fam 1993;55:888 – 900. 41. Jones HW. Multiple births: how are we doing? Fertil Steril 2003;79: 17–21. 42. Cramer DW, Liberman RF, Powers D, Hornstein MD, McShane P, Barbieri RL. Recent trends in assisted reproductive techniques and associated outcomes. Obstet Gynecol 2000;95:61–6. 43. Hock DL, Seifer DB, Kontopoulos E, Ananth CV. Practice patterns among board-certified reproductive endocrinologists regarding highorder multiple gestations: a United States national survey. Obstet Gynecol 2002;99:763–70. 44. Deber RB, Kraetschmer N, Irvine J. What role do patients wish to play in treatment decision making? Arch Intern Med 1996;156:1414 –20. 45. Gleicher N, Campbell DP, Chan CL, Karande V, Rao R, Balin M, et al. The desire for multiple births in couples with infertility problems contradicts present practice patterns. Hum Reprod 1995;10:1079 –84. 46. Ginsburg FD. Contested lives: the abortion debate in an American community. Berkeley, CA: University of California Press, 1989. 47. Dickey RP. A year of inaction on high-order multiple pregnancies due to ovulation induction. Fertil Steril 2003;79:14 –6. 48. Fritz MA, Ory SJ. Practice guidelines cannot be justified in the absence of sufficient evidence: inaction is far more appropriate than indefensible action. Fertil Steril 2003;79:22–4. 49. Rosenwaks Z, Chung PH. High-order multiple pregnancy: is it a matter of inaction or a consequence of practice patterns? Fertil Steril 2003; 79:25–6. 50. Daya S. Inappropriate use of cohort studies to formulate treatment guidelines. Fertil Steril 2003;79:27. 51. Dickey RP. It has really been 15 years of inaction on high-order multiple pregnancies due to ovulation induction. Fertil Steril 2003;79: 28 –9.
Vol. 80, No. 2, August 2003