Original Article

Journal of Perinatology (2008) 28, 12–19; doi:10.1038/sj.jp.7211860; published online 25 October 2007

The health-care experiences of families given the prenatal diagnosis of trisomy 18

L V Walker1,4, V J Miller2 and V K Dalton3

  1. 1Department of Human Genetics, University of Michigan, Ann Arbor, MI, USA
  2. 2Trisomy 18 Foundation Inc, Dale City, VA, USA
  3. 3Department of Obstetrics and Gynecology, University of Michigan, Ann Arbor, MI, USA

Correspondence: Dr VK Dalton, Department of Obstetrics and Gynecology, L4000 Women's Hospital, University of Michigan, 1500 E. Medical Center Drive, Ann Arbor, MI 48109, USA. E-mail: daltonvk@med.umich.edu

4Current address: Reproductive Genetics Institute, Chicago, IL, USA

Received 9 April 2007; Revised 13 September 2007; Accepted 5 October 2007; Published online 25 October 2007.

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Abstract

Objective:

 

The objective of this study was to examine parent-reported experiences in the health care system after receiving the prenatal diagnosis of trisomy 18 and to identify factors that contribute to satisfaction with care.

Study Design:

 

Nineteen families who received the diagnosis between 2002 and 2005 were given semistructured telephone interviews. Of the 19 families, 11 continued the pregnancy while the remaining 8 chose induced abortion. Classical content analysis was utilized to identify themes among subject responses.

Result:

 

We identified several specific aspects of care as key in either being highly satisfied or dissatisfied: expressions of empathy from provider, continuity of care, communication, valuing the fetus and participation in medical decision-making.

Conclusions:

 

Aspects of care that were identified as reasons for dissatisfaction are potentially modifiable by training, education or team-based approaches. Further studies are necessary to determine how we can improve the quality of services during prenatal diagnosis.

Keywords:

trisomy 18, prenatal screening, patient satisfaction

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Introduction

Trisomy 18 is a chromosomal disorder of aneuploidy associated with a high rate of spontaneous loss and early infant deaths, mainly due to central apnea in the first week of life.1, 2, 3, 4 The condition leads to many different developmental problems such as growth deficiency, feeding and breathing difficulties, developmental delays, and psychomotor mental retardation.5 Recent studies document that 50% of liveborn infants with trisomy 18 survive beyond the first week of life,1 with 5 to 10% surviving beyond the first year.3, 6

Recent studies show that maternal serum and ultrasound screening procedures can detect over 80% of pregnancies affected with trisomy 18.4 Prenatal ultrasound is widely perceived by expectant families to be a social event and a pleasant opportunity to see and meet their baby. Oftentimes, families are not prepared for the idea that abnormalities may be detected.7 Once a concern is detected or a diagnosis is made, families commonly respond with deep shock at the initial loss of a normal pregnancy.8 Losing the expectation of a healthy baby is also associated with a grief response characterized by stages of shock and disbelief, denial, anger, adaptation and adjustment similar to the phases identified in grief literature.9

Not only do families need to process the information conveyed at the time of a prenatal diagnosis, but they also must make decisions in a limited amount of time, with the most immediate one being whether to continue the pregnancy or to induce abortion. Families choosing to continue the pregnancy face decisions regarding delivery and treatment of the baby after birth.8 Those opting for an induced abortion often need to decide how and where the procedure will be carried out and must comply with the national and state abortion regulations. The care team is often called upon to assist patients in their decision-making.

Satisfaction is an important measure of quality of care because it explores how health-care professionals meet a patient's values and expectations of treatment.10, 11 For instance, the more a doctor's performance meets a patient's expectations, the more satisfied the patient will be with the services.12 A summary of 17 reports published between 1984 and 2001 integrated the findings of qualitative studies of expectant families who were prenatally diagnosed with a fetal anomaly or other problem. The review found that while some couples perceived their health-care workers to be both supportive and informative during the decision-making and treatment process, other couples reported their providers as judgmental and misinformed.13 It is neither known how a parent's prior attitude impacts their response to prenatal diagnosis, nor how that response influences subsequent emotions and behaviors.14 In addition, the relationship a health-care provider has with a couple, his/her ability to use a couple's agenda to guide the conversation and the provision of support and empathy may greatly affect a couple's perception and satisfaction with their provider.15 Ineffective communication between health-care professionals and families, coupled with discomfort among professionals on how to deal with these situations, can negatively influence families' experiences and their perceived quality of care.14 It has been suggested that medical professionals should attempt to understand families' needs and experiences and provide explicit support for the decisions that are made in the aftermath of the diagnosis.16

As yet, there have been no studies dealing exclusively with families who have received the prenatal diagnosis of trisomy 18 and their satisfaction with care. This study explores families' overall experiences in the health care system after receiving a diagnosis of trisomy 18. The objective of our study was to examine the quality of their interaction with health care providers and to identify aspects of care associated with satisfaction.

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Methods

This study consisted of a non-random survey of families who experienced a pregnancy complicated by trisomy 18 within the last 4 years. A total of 26 families participated in this study. Families were recruited through the Trisomy 18 Foundation online parent support program (www.trisomy18.org) and from the University of Michigan Perinatal Assessment Center. Approval for this study was obtained through the Institutional Review Board of the University of Michigan Medical School (No. 1647).

Potential participants learned of the study from a message board post on the Trisomy 18 Foundation website, reaching over 4000 registered users, which described the study and listed the contact information for the principal investigator. Five additional families at the University of Michigan were contacted over the telephone by a genetic counselor not affiliated with this study and given the principal investigator's contact information. Participants must have received the diagnosis of trisomy 18 prenatally since 2000 and had to be over the age of 18 years to be eligible. Thirty-three families contacted the principal investigator and were sent the study information, including the informed consent document, a list of grief support resources and a pre-enrollment questionnaire. Participants were given the interview questions prior to study consent. Of the 33 families requesting information, 19 completed the written informed consent process. This comprised of 7 couples and 12 mothers.

Data collection

Our survey questions were modeled on previous studies on prenatal diagnosis of fetal anomalies.14, 16, 17, 18. Interviews consisted of a series of open- and close-ended questions regarding reproductive history, the events leading up to the diagnosis, the delivery of the diagnosis, decision-making and overall satisfaction with care. Other questions addressed perceived quality of information and emotional support from health-care providers.

Satisfaction with care was measured on a 10-point scale four times during the interview. Participants evaluated their satisfaction with care during the following stages:prenatal screening, diagnosis and decision-making; prenatal care after diagnosis; and at the time of delivery or induced abortion. (For instance: ‘on a scale between 1 and 10, with 1 being least satisfied with your care and 10 being the most satisfied, how would you rate the health care that you received during the screening process?’) Following the quantitative questions, participants were asked to explain what features of their care were key in their level of satisfaction (or dissatisfaction). We further examined satisfaction by identifying themes among these explanations. Two pilot telephonic interviews were conducted with volunteer mothers from the Trisomy 18 Foundation in December 2005 to assess clarity, length and sensitivity. Amendments were made to the protocol based on these interviews.

The primary means of collecting data was through semistructured telephone interviews. All interviews were conducted by L.V.W. and were audio-taped. On average, the interviews lasted for an hour . When both partners participated, they were interviewed separately but not always in immediate succession. Data collection took place from December 2005 to March 2006.

Analysis

Our primary unit of analysis was the individual parent. We considered examining each parent separately but found a high degree of consistency within couples; therefore, couples were analyzed as a pair. The interviews were transcribed in their entirety. Based on these transcripts, we prepared a case report for each individual participant. Using classical content analysis,19 two researchers concurrently coded the interviews to identify major themes and reached a consensus on all differences. Transcripts and field notes were perused as necessary.

As a result of the coding, we identified themes in the following categories: patient–provider relationship, importance of communication, valuing the pregnancy/fetus by provider and family control. We created matrices to examine similarities and differences between those who chose to continue the pregnancy and those who opted for an induced abortion. Descriptive statistics were used to describe our sample population. Mean satisfaction was compared using t-tests. We defined high satisfaction as a score of 8 or higher, and themes associated with high satisfaction were examined using χ2.

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Results

Sample characteristics

A total of 19 families participated in this study. The socio-demographics of the individuals participating in the study are given in Table 1. Overall, participants were white, non-Hispanic and had a college degree or higher. All families received the diagnosis between 2002 and 2005. Eight families chose an induced abortion, while the remaining 11 continued their pregnancies until fetal demise or spontaneous labor. Several themes emerged as important in determining satisfaction with care: patient–provider relationship, importance of communication, valuing the pregnancy/fetus by providers and family control.


Patient–provider relationship
 

The families' relationship with their health care providers had a strong impact on numerous elements of satisfaction with their care. Several themes were associated with positive experiences.

Expressing empathy
 

Fourteen of 19 families reported that they felt that members of their health-care team expressed compassion and empathy: It wasn't just ‘here's your diagnosis, bye, now I have to go meet another patient.’ Rather it was ‘this is what it is. I'm going to go leave the room now because I know you two are a little upset and then I'm going to come back in.’ She was just very good about the whole thing. (Mother 13, induced abortion) My genetic counselor was available to talk to me whenever I called her. She indulged my scenarios, my million scenarios. I think I asked her the same questions a lot of times, and she was very patient about that. (Mother 33, induced abortion) The nurse always spent time with me, and she said, ‘You know you can call me anytime if you just want to talk.’ (Mother 2, continued pregnancy)

In many instances, the support was important after delivery or induced abortion during the family's continued grieving period: Our main doctor for our pregnancy… let me know which days she was available after clinic, and invited me to come in any time I wanted to ask questions and to review what had happened at the delivery. Besides the 6-week appointment, I met with her 2 other times. (Mother 10, continued the pregnancy)

Expressing empathy was identified as a key factor in satisfaction levels and was positively associated with both higher overall mean satisfaction scores (Table 2) and being highly satisfied (P<0.02).


Continuity of care
 

Discontinuity of care was a common complaint among our study population. Families were often referred to a larger medical center or a high-risk obstetric clinic for further care. Maintaining communication with the referring provider was important for many families. The continued involvement of professionals and office staff with whom they had already established trust provided families with an open environment where they felt supported: The Ob, she kept in constant touch with me. She would call me after perinatalogy appointments or would call and ask how things were going. She was very helpful in my post-care. (Mother 32, induced abortion)

However, seven families reported losing contact with their regular obstetrician or health-care provider after receiving the diagnosis, and this had a negative impact on the satisfaction with care. The remainder of their care continued with providers with whom they were not familiar: I lost contact with my regular health care provider, so we got shuttled into a little sub-system. It's like ‘something's wrong, so you don't go to us anymore, you go to these new people.’ It would've been nice if my doctor would've called and said, ‘you know I understand that there's a problem, let me know if there's anything I can help you with.’ (Mother 29, induced abortion) Our initial doctor was amazing. He was very personable, very in tune to everything. Then we had to switch to another doctor. We had to switch to a whole new hospital after that. (Mother 11, continued the pregnancy)

Continuity of care was specifically mentioned as a reason for lower overall satisfaction and was associated with lower satisfaction in the patient-provider relationship category (P<0.004).

Importance of communication
 

Communication between families and their providers was often cited as a negative aspect of care, and complaints were directed at several points in the care process.

Education during the screening process
 

Eleven of 19 families reported that they were poorly informed about the screening process. Some families felt their health care providers overly reassured once they received their abnormal serum screening results. Other families thought they were being screened for Down's syndrome and did not realize that they were being evaluated for trisomy 18 as well. It was never really mentioned to me, like some sentence saying cysts on the brain were a strong indication of Down's syndrome but I don't even think they said trisomy 18. I think that made it that much harder when we actually got the diagnosis. (Mother 20, continued the pregnancy)

Ultrasound
 

Many families were unhappy with the way in which they were informed of their ultrasound results. Families often reported that they could tell that their ultrasound technician noticed a problem with the ultrasound, but that since the technician could not disclose findings without a doctor present, they had to wait to review the findings. In some instances, families had to wait until the next day for their results. It was pretty evident from the woman who did our ultrasound that something was wrong. She was very happy and cheery when we started and she became more serious and subdued. She said, ‘You need to call one of your doctors right away tomorrow.’ I had had a baby before and I knew that they don't normally say that. (Mother 10, continued the pregnancy)

Other families described that they had no idea that there was a problem seen on ultrasound until a doctor called them the following day. In these instances, the ultrasound technician gave little or no indication that there was a problem and a doctor was not present at that time to explain what was being observed: The ultrasound technician had a poker face like you would not believe. I had no clue that anything was wrong. I think that I would have preferred for her to have found a doctor and talked to me right there…because we learned the sex of our baby and I had practically redecorated the nursery the night I found out, and we had called the whole family. (Mother 31, induced abortion)

Diagnosis and prognosis
 

Families talked extensively about how they were given the diagnosis of trisomy 18 and its prognosis. All families spoke specifically about the words and phrases used to explain trisomy 18. All families were told there was no hope for survival and most indicated that they were advised that an induced abortion was probably the best option. Seven families reported that the prognoses they were given were overly negative and six of these families opted to continue the pregnancy. Among families that opted to continue the pregnancy, the dominant impression was that providers did not disclose the options fully. Six families reported that the information they received was inaccurately presented and cited ‘evidence’ of survival and successful treatment options they identified during their own research on trisomy 18. To these families, this meant that providers were either biased in their presentation of information or uninformed. Specifically, they felt that the term ‘incompatible with life’ was not entirely true. I think the ‘not compatible with life’ is not only terrifying to hear, but it's not true. It's incompatible with living to 40 years, but it's not incompatible to life in general. It's such a black and white statement that it gives you no hope at all…it's a false statement. (Mother 10, continued the pregnancy) I think they need to put something together that has more factual information. I mean, I've heard about children living with trisomy 18 for a short amount of time, and…I think that part's not clear. They should say, ‘Your child may have a chance to live.’ (Mother 29, induced abortion)

This sentiment continued during descriptions of families' experience with options-counseling. Many families that continued with their pregnancies used the phrase ‘worst case scenario’ to describe the emphasis of the information they were given. Many felt this illustrated their doctor's preference toward induced abortion and was seen as offensive: I think many families won't have heard of trisomy 18 either, so to portray it in such a terrible and frightening light would probably be to persuade people to terminate. If they had a more balanced view, then maybe people wouldn't feel they had to terminate… I thought it was a really one-sided view we were given. (Mother 10, continued the pregnancy)

Out of the total of 11 families that continued with their pregnancies, only 1 parent, a mother, mentioned that she did not want ‘false expectations or hopes’ when learning about trisomy 18. In comparison, six of the eight families that terminated their pregnancies said that they wanted the information delivered in a straightforward manner with no false hope: She said that trisomy 18 was ‘not compatible with life.’ Those words carry a lot of weight. Children are born who don't need extensive medical care for a number of years. It's nice to hear some of that, but then you don't want too much false hope. It keeps you in denial. Personally, I want the reality of it. (Father 13, induced abortion)

For some families, the counseling helped them justify their decision to induce abortion: She used the term a ‘lethal diagnosis’…When you have to make a decision to terminate a pregnancy, that is ,I guess, at least helpful in trying to make that decision. I would say they really didn't sugarcoat it but that was probably good. Because if they had left me with a glimmer of hope, I would not have terminated my pregnancy. (Mother 31, induced abortion)

Lack of written information
 

Eleven families reported that there was a lack of written information and no support group information provided to them at the time of the diagnosis: They wrote ‘trisomy 18’ on the back of a business card and we were told to look it up on the internet. That was something that was very upsetting to us (Mother 1, continued the pregnancy).

One father spoke of the difficulty of finding accurate information when health-care professionals do not provide those written resources: I looked on the Internet and found some support groups. They get people posting who have a spiritual view or whose babies live a long time. There is the tendency to think that that is the typical experience, which is a shortcoming of leaving the patient to find information on their own. (Father 18, continued the pregnancy)

Valuing of pregnancy/fetus by providers
 

In this sample, expressing value for the fetus or treating the pregnancy like any other, as much as possible, was important to families. Examples were given from experiences with prenatal care and from decisions regarding mode of delivery (vaginal versus cesarean section). Families that felt that their health-care team valued their fetus during their pregnancy reported higher mean satisfaction (Table 2) and were more likely to be highly satisfied than those that did not. (100 versus 56%, P=0.03).

Seven families expressed appreciation that health-care professionals treated them with special consideration and care because of their unique situation. Families were also appreciative when their doctors attempted to normalize their prenatal care during the remainder of the pregnancy: We'd go into the doctors and they'd let us know that things were going well with the pregnancy. They tried to normalize it as much as possible. They understood the fact that we were going through a lot and may need some extra attention. (Mother 1, continued the pregnancy)
My ob would bring the ultrasound in the room whenever we visited so we could see her on the screen moving around. (Mother 2, continued the pregnancy)

Conversely, six families believed that their providers did not share their outcome expectations and felt that this led to substandard care. When we got the amnio results, my ob said, ‘Now I am working with one patient instead of two.’ I left the office that day thinking, ‘He doesn't get me.’ (Mother 17, continued the pregnancy) Before the amnio, the doctor said that they would want to do an echocardiogram to look at the heart. But once we got the trisomy 18 diagnosis, they canceled the appointment for the echocardiogram thinking it wasn't necessary. We called back and were quite adamant that we still wanted to have it done. (Mother 10, continued the pregnancy) They told me that my regular doctor probably wouldn't be seeing me anymore, there wouldn't be any more ultrasounds, it would basically be my health screening done but not the baby's. (Mother 20, continued the pregnancy)

Three families that continued the pregnancy expressed dissatisfaction with doctors who declined to perform a cesarean section at the time of the delivery, even if the baby showed signs of distress during labor. These families felt that they were offered fewer treatment options as compared to families with normal pregnancies and this was a form of discrimination by physicians who were not committed to increasing the baby's chance for survival: He said that he would not give me a C-section for a baby with trisomy 18…that if I decided to carry her to term, and during labor and delivery the baby went into distress, he would not do a C-section on me and he would just let the baby die. Because the risks were greater to me than the chance of the baby making it through delivery. I wouldn't have gone 9 months for her to go during that last hour. (Mother 25, continued the pregnancy)

The remaining families did not report this conflict regarding delivery options. Some stated that they had the option of a cesarean section delivery and never mentioned that their doctors dissuaded them from that option, while others appreciated it when doctors recommended against a cesarean section because of the risks to the mother.

Family control
 

Participating in medical decision-making was important to the families in this study, while feeling that they had no input to the care plan was an explanation for low satisfaction. This lack of control was evident at several points along the care continuum, including the decision to continue the pregnancy and delivery options.

Several families described having to ‘fight’ on behalf of their fetus. Families desired autonomy over the decision-making process and many chose not to seek counsel from providers who seemed biased toward certain options. Some families reported that they feared their fetus would receive substandard care from providers not interested in supporting their hopes and preferences for the remainder of the pregnancy and delivery. Families who reported struggling for control over prenatal and delivery options had lower satisfaction levels than those families that did not perceive these conflicts (Table 2, P=0.007).

Induced abortion versus continuing the pregnancy
 

Overall, there was no difference in mean satisfaction scores or the likelihood of being highly satisfied between those families that chose to continue the pregnancy and those that chose to undergo an induced abortion. Both groups reported similar experiences with regard to informed consent for the screening process, delivery of diagnosis and options counseling.

We, however, identify some interesting differences. First, with regard to options counseling, those families who opted for induced abortions consistently reported having the support of their providers. This was not always the case for those who opted to continue the pregnancy. In fact, 6 of the 11 families that continued the pregnancy indicated feeling that they were going against the advice of their providers. Second, differences were seen among families' preferences of how trisomy 18 is explained. Overall, families that continued the pregnancy wished that medical information had been more positive. These families were offended by the phrase ‘incompatible with life’, which often created an environment of distrust with their health-care providers. For those who opted for induced abortion, the portrayal of the severe medical consequences of the trisomy 18 diagnosis justified their decision to end the pregnancy. These families wanted their education of trisomy 18 to be straightforward but not idealistic.

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Discussion

Satisfaction with care is an important measure of quality and is independent of morbidity or treatment success in many clinical settings.20, 21, 22 A few studies have examined satisfaction among families undergoing prenatal diagnosis, but none have linked satisfaction or dissatisfaction with specific aspects of care. This study adds to our knowledge by identifying aspects of care that are key in determining satisfaction and could be targeted to optimize family satisfaction with care: expressions of empathy from provider, continuity of care, communication, valuing the fetus and developing care plans.

Expressions of empathy are valued by families after a prenatal diagnosis.23, 24, 25, 26 Leithner et al.24 reported that 42% of women given an abnormal prenatal diagnosis were dissatisfied with their physician's attitude. Further, our study found that a perceived lack of health-care provider's empathy was associated with overall dissatisfaction with care and was specifically cited as a contributing factor. Often, improving provider education is suggested to improve our skills in helping families through loss. Alternatively, multidisciplinary teams with specialized skills that focus on providing psychosocial support along with information have been shown to improve satisfaction in other care settings27 and could be adapted to perinatal care.

The quality of communication between families and care providers affects satisfaction with care in many clinical settings.10, 24, 27 Education about screening, the delivery of bad news and discussions regarding prognosis and treatment were all cited as reasons for dissatisfaction by our study sample. Leithner et al.24 also found a high degree of dissatisfaction with ‘the information provided by physicians’ among prenatal diagnosis patients. Our study and others23 have suggested that families desire both written and oral information, which could be easily adopted in perinatal settings. We were particularly struck by the frequent reports of being overly reassured by providers after initial abnormal serum screening results and of not having prompt access to the ultrasound results even when they suspected something was wrong. Others have also reported similar findings.7 Both these examples provide clear opportunities to improve our quality of care.

Families repeatedly talked about the language used during counseling about trisomy 18 and its prognosis. They reported that the use of specific terms, such as ‘incompatible with life,’ contributed to their dissatisfaction with their care. These families believed that they were given an overly pessimistic prognosis, citing the survivors they have located in their own research. (Although ultimately fatal, some reports estimate that 5 to 25% of infants with trisomy 18 survive the first year.5, 6, 28) Specifically, several families felt that terminology was fatalistic to encourage them to choose an induced abortion rather than to continue the pregnancy, a theme echoed in a similar study of families receiving a prenatal diagnosis of holoprosencephaly.16 The possibility of any life, however brief, was important to many of the families in our study. For some, the possibility of seeing and holding their baby was seen as an opportunity to grieve and heal. For others, an induced abortion was not a personal option. It appeared that providers and families interpreted prognostic estimates quite differently, despite having access to the same statistics. However, this study could not determine whether families were really reacting to the specific language used or whether these reactions were really an expression of loss. In other words, changing the language we use might not improve satisfaction. Still, providers need to be aware that these different perspectives can later be a source of dissatisfaction for families. It is important for providers to be sensitive to families' perspectives while being accurate. Choice of words may be important during these discussions.

Many families reported that they did not believe their provider shared their expectations for the remainder of the pregnancy. Similarly, families that opted to continue the pregnancy sometimes felt as though they had little input in medical decision-making. For instance, several families expressed frustration because they felt that some care options were unavailable, such as fetal monitoring or cesarean section. Participation in medical decision-making in other clinical situations has been shown to improve satisfaction29 and could possibly improve the experience of families faced with abnormal prenatal diagnosis. Care plans should be developed with as much family input as possible, balancing safety, resource use and family preferences.

This study has several limitations. Since this study was largely hypothesis generating, many of our questions were open-ended so that the respondent could talk freely and would not feel forced to answer questions arousing emotional discomfort. As a result, themes that arose outside the prompts included in our instrument could not be quantified. We also acknowledge that our sample is unique and may not represent the sentiments of other families experiencing similar loss. Further, our unit of analysis includes both individuals and couples which may have affected our findings, particularly the quantitative results. Additionally, we group families that experienced stillbirths with those that had live births, which did not allow us to examine differences between these groups. Even accounting for these limitations, our findings add to the growing literature on patients' experiences with the health-care system after perinatal loss or abnormal prenatal screening. Our study and others suggest that there are specific clinical areas that could be targeted to improve our performance in caring for families experiencing loss.

This examination of families' experiences with receiving the prenatal diagnosis of trisomy 18 leaves room for future studies continuing to explore patient satisfaction after receiving prenatal diagnoses. Further characterization of patient preferences with regard to education and communication during screening, diagnosis and treatment plan making is needed. Guidance from families is also needed to train providers how to deliver bad news and express empathy for families. In the meantime, providers should be aware of the growing information regarding patient preferences in care after an abnormal prenatal diagnosis.

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References

  1. Baty BJ, Blackburn BL, Carey JC. Natural history of trisomy 18 and trisomy 13: I. Growth, physical assessment, medical histories, survival, and recurrence risk. Am J Med Genet 1994; 49(2): 175–188. | Article | PubMed | ChemPort |
  2. Carey JC. Trisomy 18 and trisomy 13 syndromes In: Cassidy SB, Allanson JE (eds). Management of Genetic Syndromes 2005. 2nd edn. John Wiley & Sons Inc.: Hoboken, 555–568.
  3. Carey JC. Introductory comments special section: trisomy 18. Am J Med Genet A 2006; 140A(9): 935–936.
  4. Parker MJ, Budd JL, Draper ES, Young ID. Trisomy 13 and trisomy 18 in a defined population: epidemiological, genetic and prenatal observations. Prenat Diagn 2003; 23(10): 856–860. | Article | PubMed | ChemPort |
  5. Kosho T, Nakamura T, Kawame H, Baba A, Tamura M, Fukushima Y. Neonatal management of trisomy 18: clinical details of 24 patients receiving intensive treatment. Am J Med Genet A 2006; 140(9): 937–944. | PubMed |
  6. Rasmussen SA, Wong LY, Yang Q, May KM, Friedman JM. Population-based analyses of mortality in trisomy 13 and trisomy 18. Pediatrics 2003; 111(4): 777–784. | Article | PubMed |
  7. Mitchell LM. Women's experiences of unexpected ultrasound findings. J Midwifery Womens Health 2004; 49(3): 228–234. | Article | PubMed | ISI |
  8. Statham H, Solomou W, Chitty L. Prenatal diagnosis of fetal abnormality: psychological effects on women in low-risk pregnancies. Baillieres Best Pract Res Clin Obstet Gynaecol 2000; 14(4): 731–747. | Article | PubMed | ChemPort |
  9. Badenhorst W, Riches S, Turton P, Hughes P. The psychological effects of stillbirth and neonatal death on fathers: systematic review. J Psychosom Obstet Gynaecol 2006; 27(4): 245–256. | Article | PubMed |
  10. Scholle SH, Weisman CS, Anderson R, Weitz T, Freund KM, Binko J. Women's satisfaction with primary care: a new measurement effort from the PHS National Centers of Excellence in Women's Health. Womens Health Issues 2000; 10(1): 1–9. | Article | PubMed | ChemPort |
  11. Williams B. Patient satisfaction: a valid concept? Soc Sci Med 1994; 38(4): 509–516. | Article | PubMed | ISI | ChemPort |
  12. Sitzia J, Wood N. Patient satisfaction: a review of issues and concepts. Soc Sci Med 1997; 45(12): 1829–1843. | Article | PubMed | ChemPort |
  13. Sandelowski M, Barroso J. The travesty of choosing after positive prenatal diagnosis. J Obstet Gynecol Neonatal Nurs 2005; 34(3): 307–318. | Article | PubMed |
  14. Chaplin J, Schweitzer R, Perkoulidis S. Experiences of prenatal diagnosis of spina bifida or hydrocephalus in parents who decide to continue with their pregnancy. J Genet Couns 2005; 14(2): 151–162. | Article | PubMed |
  15. Street Jr RL, Krupat E, Bell RA, Kravitz RL, Haidet P. Beliefs about control in the physician–patient relationship: effect on communication in medical encounters. J Gen Intern Med 2003; 18(8): 609–616. | Article | PubMed |
  16. Redlinger-Grosse K, Bernhardt BA, Berg K, Muenke M, Biesecker BB. The decision to continue: the experiences and needs of parents who receive a prenatal diagnosis of holoprosencephaly. Am J Med Genet 2002; 112(4): 369–378. | Article | PubMed |
  17. Skotko B. Mothers of children with Down syndrome reflect on their postnatal support. Pediatrics 2005; 115(1): 64–77. | PubMed |
  18. Skotko BG. Prenatally diagnosed Down syndrome: mothers who continued their pregnancies evaluate their health care providers. Am J Obstet Gynecol 2005; 192(3): 670–677. | Article | PubMed |
  19. Ryan GW, Bernard HR. Data management and analysis methods In: Denzin N, Lincoln YS (eds). Collecting and Interpreting Qualitative Materials. 2nd edn. Sage: London, 2003; 259–309.
  20. Avery KN, Metcalfe C, Nicklin J, Barham, Alderson D, Donovan JL et al. Satisfaction with care: an independent outcome measure in surgical oncology. Ann Surg Oncol 2006; 13(6): 817–822. | Article | PubMed |
  21. Hirsh AT, Atchison JW, Berger JJ, Waxenberg LB, Lafayette-Lucey A, Bulcourf BB et al. Patient satisfaction with treatment for chronic pain: predictors and relationship to compliance. Clin J Pain 2005; 21(4): 302–310. | Article | PubMed |
  22. Seecharan GA, Andresen EM, Norris K, Toce SS. Parents' assessment of quality of care and grief following a child's death. Arch Pediatr Adolesc Med 2004; 158(6): 515–520. | Article | PubMed | ISI |
  23. Alkazaleh F, Thomas M, Grebenyuk J, Glaude L, Savage D, Johannesen J et al. What women want: women's preferences of caregiver behavior when prenatal sonography findings are abnormal. Ultrasound Obstet Gynecol 2004; 23(1): 56–62. | Article | PubMed | ChemPort |
  24. Leithner K, Assem-Hilger E, Fischer-Kern M, Loffler-Stastka H, Thien R, Ponocny-Seliger E. Prenatal care: the patient's perspective. A qualitative study. Prenat Diagn 2006; 26(10): 931–937. | Article | PubMed |
  25. Verjaal M, Leschot NJ, Treffers PE. Women's experiences with second trimester prenatal diagnosis. Prenat Diagn 1982; 2(3): 195–209. | Article | PubMed | ChemPort |
  26. White-van Mourik MC, Connor JM, Ferguson-Smith MA. The psychosocial sequelae of a second-trimester termination of pregnancy for fetal abnormality. Prenat Diagn 1992; 12(3): 189–204. | Article | PubMed | ChemPort |
  27. Frost MH, Arvizu RD, Jayakumar S, Schoonover A, Novotny P, Zahasky K. A multidisciplinary healthcare delivery model for women with breast cancer: patient satisfaction and physical and psychosocial adjustment. Oncol Nurs Forum 1999; 26(10): 1673–1680. | PubMed | ChemPort |
  28. Brewer CM, Holloway SH, Stone DH, Carothers AD, FitzPatrick DR. Survival in trisomy 13 and trisomy 18 cases ascertained from population based registers. J Med Genet 2002; 39(9): e54. | Article | PubMed | ChemPort |
  29. Gattellari M, Butow PN, Tattersall MH. Sharing decisions in cancer care. Soc Sci Med 2001; 52(12): 1865–1878. | Article | PubMed | ISI | ChemPort |
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Acknowledgments

No financial support was provided to the authors for this research.

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