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C 2003) Journal of Genetic Counseling, Vol. 12, No. 2, April 2003 (°
Assessment of the Effectiveness of Genetic Counseling by Telephone Compared to a Clinic Visit Karan K. Sangha,1,3 Anita Dircks,2 and Sylvie Langlois2
Maternal serum screening, also known as the triple screen, is used during pregnancy to assess the risk of carrying a fetus with specific chromosome abnormalities or open spina bifida. All women in British Columbia who screen positive are eligible for genetic counseling and are offered amniocentesis. The purpose of this study is to determine what differences (if any) exist in patients’ understanding and/or anxiety when genetic counseling for a positive triple screen is conducted in person versus over the telephone. Each patient who participated was given the choice of having genetic counseling in person or over the telephone, this after a randomized design failed to elicit any participants. Using a written postcounseling questionnaire, each patient was assessed for her understanding of the information presented in the session, and her anxiety regarding her risk. In this small pilot study, no large differences were detected in patients’ understanding or anxiety when genetic counseling was conducted by telephone versus in person. KEY WORDS: genetic counseling; telephone; maternal serum screen; knowledge; anxiety.
INTRODUCTION The triple screen (or maternal serum screen) is a blood test offered to pregnant women between 15 and 20 weeks of gestation to assess the chance of carrying a fetus with specific chromosome abnormalities or an open neural tube defect (Carroll, 1994). The test measures the concentrations of three proteins (human chorionic gonadotropin, alpha-fetoprotein, and unconjugated estriol) in the pregnant woman’s 1 Medical
Genetics Clinic, University of Alberta Hospital, Edmonton, Alberta, Canada.
2 Provincial Medical Genetics Programme, Children’s and Women’s Health Centre of British Columbia
and Department of Medical Genetics, University of British Columbia, Vancouver, British Columbia, Canada. 3 Correspondence should be directed to Karan Sangha, 8-53 Medical Sciences Building, University of Alberta Hospital, 8440 112 Street, Edmonton, Alberta, Canada T6G 2H7; e-mail: [email protected]. 171 C 2003 National Society of Genetic Counselors, Inc. 1059-7700/03/0400-0171/1 °
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serum and compares these with the average concentrations in the general population of pregnant women at the specific time in gestation that the blood is collected. These figures are then used to estimate the chance of the fetus being affected with trisomy 18, trisomy 21, and open spina bifida. A positive screen for Down syndrome is defined as a risk greater than or equal to 1/385. This figure has been chosen as the cutoff because this risk is approximately equal to the risk of trisomy 21 at a maternal age of 35 years. Using the 1/385 cutoff, the triple screen test has the ability to detect 72% of pregnancies with Down syndrome. Of all the women who screen positive for trisomy 21 using the maternal serums screen, only 2.8% are actually carrying a fetus with Down syndrome. The chance of screening negative if the fetus does not have trisomy 21 is approximately 91% (BC Children’s and Women’s Health Centre GEM Program, unpublished data). Currently in British Columbia (BC), every woman who has a positive triple screen is referred to the Provincial Medical Genetics Programme in Vancouver for genetic counseling and is offered amniocentesis. Since the triple screen became available as a clinical test in 1995 in BC, the number of women screened is rising every year, presumably due to increasing awareness of the test among primary care physicians. Approximately 15,000 women were screened using the triple screen test in 2000 in BC. Eight percent of these women screened positive for Down syndrome. Thus, about 1200 women screened positive in that year. At least 25% of these women (300 women per year) live outside of the Greater Vancouver area (Dr. Catherine Halstead, unpublished data) and therefore must travel a significant distance on short notice to follow up on a positive result. Currently, the only choice available to patients who reside outside of the Greater Vancouver area is to travel to Vancouver for a face-to-face appointment with a genetic counselor or to decline genetic counseling. Therefore, there is a need to explore alternative methods of providing genetic counseling such as telephone counseling. Possible advantages of telephone counseling include avoidance of the stress, expense and time commitment of a clinic visit, and reduction in barriers to service by increasing accessibility (Tsu et al., 2002). These issues are compounded for patients traveling from out of town. A telephone genetic counseling service has the ability to increase access to care for patients in geographically isolated areas. In addition, the visual privacy of telephone consultation may allow the patient to feel safe and comfortable expressing her feelings in an open manner, making the interaction more productive (Wilson and Williams, 2000). There are also some potential disadvantages of telephone genetic counseling. Rapport building between the counselor and patient may be hindered by the lack of visual cues available to each party. It is estimated that 65–93% of social meaning between individuals is in the form of nonverbal cues (Soet and Basch, 1997). In addition, one might speculate that the inability of the genetic counselor to use visual aides such as diagrams in providing information may result in a lower level
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of understanding on the part of the patient. Soet and Basch (1997) have also shown that visual learning is preferred over auditory learning. Thus, conveying complex information over the telephone may be more challenging than doing so in a faceto-face context. Telephone contact may also have the effect of an impersonal or anonymous interaction between patient and counselor, impeding the emotional connection. This could result in a higher anxiety level for the patient following telephone counseling compared with counseling in person. In our review of the literature we were able to identify two studies that focused on assessing the effectiveness of telephone genetic counseling. Rayburn et al. (1982) evaluated the use of a toll-free telephone hot line designed to provide prenatal counseling for various indications such as teratogen exposure, obstetric complications, genetic conditions, and other prenatal concerns. Outcome measures were the number of patients using the service, the number of patients undergoing amniocentesis, and feedback from physicians. A steady increase in the number of callers, and the number of amniocenteses performed was found, as well as positive feedback from physicians. Li et al. (1999) assessed a telephone counseling program for parents of children diagnosed with G6PD deficiency through newborn screening in Hong Kong. Success of the program was determined by patients’ satisfaction with counseling and retention of information as ascertained by a questionnaire developed by the investigators. The authors concluded that telephone counseling was the method of choice. However, neither of these studies used a control or comparison group. Although there are few published studies on telephone genetic counseling, a 1993 survey found that 26 out of 26 genetic counselors in the United States reported having done some genetic counseling by telephone (Ormond et al., 2000). The effectiveness of telephone counseling in contexts other than genetic counseling has been assessed by various methods. The delivery of health care by telephone has been studied in general practice (McBride and Rimer, 1999), pediatrics (Hallam, 1989), nursing (Wilson and Williams, 2000), obstetrics (Koren et al., 1993; Rayburn et al., 1982), psychology (Hornblow, 1986), and psychiatry (Lynch et al., 1997). Many of these studies evaluated telephone hotlines set up for use by the general public, or focused on specific patient populations such as smokers, cancer patients, or individuals at high risk for suicide. The telephone counseling was generally combined with other interventions such as face-to-face consultation, physical examination, written material, home visits, or video instruction or education (McBride and Rimer, 1999) and was provided in the context of the introduction of a new service for patients, in addition to their existing health care program. The outcomes measured in these studies included the number of individuals calling a particular help line, the reduction in the number of office visits, the extent of behavior modification (e.g., the rate of smoking cessation within a given time frame), and in one case a user questionnaire to assess the value of the service to the individual in the absence of any alternatives.
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Telephone counseling versus no counseling was evaluated in a study by Lynch et al. (1997) of 29 individuals identified to have minor depression. The treatment group received six weekly 20-min telephone counseling sessions, while the comparison group received no counseling. The outcome measured was the difference in scores on the Hamilton Depression Rating Scale before and after the series of counseling sessions. In this study, the telephone counseled group was found to have a reduction in depression. Generally, prior studies evaluating the usefulness of telephone health care delivery have not taken into consideration the effect of telephone intervention in place of a face-to-face visit. Instead, they attempt to assess the value of adding a telephone-based intervention to the range of medical services available to a particular patient population. Although findings from this research suggest that telephone counseling may be effective, this research is limited by the lack of a comparison group receiving face-to-face counseling. This research may also have limited applicability to a genetic counseling session because of differences in goals. The goals of genetic counseling are not to attract the largest number of patients, nor to necessarily modify patients’ behavior. Success in genetic counseling among workers in the profession is generally thought to be indicated by an increase in the patient’s knowledge of genetics, risks and available options, assistance with decision making, help with psychosocial adjustment, and patient satisfaction (Bernhardt et al., 2000). Outcomes relevant to genetic counseling have not been previously studied comparing face-to-face with telephone genetic counseling. The purpose of this study was to compare telephone counseling with face-toface counseling for pregnant women with a positive triple screen. This study was a limited pilot study to evaluate the feasibility of telephone genetic counseling. For the purposes of this study, we identified two main outcomes for prenatal genetic counseling of women with a positive triple screen. The first was an understanding of the information provided by the genetic counselor regarding the results of the triple screen test, further testing options for the pregnancy, and the meaning of the results of further testing. The second objective was a reduction in the level of the patient’s anxiety about her risk for Down syndrome. Therefore, we assessed patients’ understanding, anxiety, and choices regarding additional testing to determine if there were differences in the two groups receiving the two delivery modes of genetic counseling. MATERIALS AND METHODS Prior to commencement of the study approval was granted by the Behavioral Research Ethics Board of the University of British Columbia and the Research Review Committee of the Children’s and Women’s Health Centre of British Columbia. Between October, 2000, and March, 2001, each patient referred to the Provincial Medical Genetics Programme in BC for a positive triple screen for Down syndrome was assessed for the eligibility criteria for participation in the study. In
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order to be eligible to participate in the study the patient had to be referred for genetic counseling for a positive triple screen for Down syndrome with no other indications for genetic assessment or counseling at the time of the referral, the patient’s first language had to be English, the patient’s residence had to be within the lower mainland of British Columbia, no more than a 2-h drive from the Vancouver clinic where genetic counseling is carried out in the province of BC, and where the amniocentesis procedure is performed. Patients who had been seen previously in the Provincial Medical Genetics Programme for genetic counseling were excluded to avoid the possible effect of this on the results of the assessment of knowledge of genetic information. For patients who met all inclusion criteria, the referring physician was contacted by telephone and asked for permission to contact her patient in order to invite her to participate in a study. If the physician gave permission, the patient was then contacted by telephone. She was given a brief verbal introduction to the study and asked if she would like to participate. The only differences between participation and nonparticipation for the patient were (1) instead of automatically going to the clinic for the genetic counseling appointment, participants would have the choice of being seen in the clinic or to have genetic counseling over the telephone; (2) participants were asked to complete a brief written questionnaire after the genetic counseling session to assess their understanding of the information presented in the session and their level of anxiety regarding their risk. All patients who had genetic counseling by telephone were offered the option of a face-to-face genetic counseling session at the conclusion of the telephone counseling session. The study was originally designed as a randomized trial. However, in the first 2 months of soliciting participants, none of the patients contacted agreed to participate in the study, with many citing anxiety as their main reason for declining. Thus the design was changed to a nonrandomized trial to give patients a choice as to the mode of counseling. Consequently, the number of participants increased. Once the design was altered, the most common reasons for nonparticipation were logistical. Therefore, we believe that the sample is representative of our study population. Once a patient agreed to participate, she received genetic counseling in person or over the telephone. One of the authors, a genetic counselor with greater than 10 years of experience in the field (AD) counseled all patients in both groups in order to minimise any variation in counseling technique. Counseling sessions, whether by phone or face-to-face were typically 45–60 min in length. Patients in both groups were given a brief verbal questionnaire consisting of two questions (see Table I) at the beginning of the session to assess their precounseling anxiety level. After the genetic counseling session, patients in both groups were mailed a written questionnaire to be completed and returned to the investigators. The questionnaire consisted of a demographics section (see Table II), a section to assess knowledge (Table III), a section to assess anxiety (Table IV), and an open-ended section for comments. The knowledge portion consisted of nine questions with the patient
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Table I. Results of the Precounseling Verbal Anxiety Survey. Mean and Standard Deviation of Answers Are Shown for Each Group Mean (SD) Telephone group (n = 12)
Questions On a scale of 1 to 10 (where 1 is none and 10 is the most anxiety you have ever felt), how would you rate the amount of anxiety you have been feeling since learning about your positive triple screen result? On a daily basis, how much do you worry about your positive triple screen result? (Constantly (10), very often (8), sometimes (6), not very often (4), almost never (2), never (0). Total score/20
Clinic visit group (n = 12)
7.3 (1.42)
8.5 (1.51)
7.2 (1.8)
7.3 (1.78)
14.5 (3.1)
15.8 (2.8)
being asked to circle “true” or “false” for each answer. The anxiety portion of the questionnaire was made up of 10 questions with the responses in the form of a 5-point Likert scale. The written questionnaire was developed by adapting questions used in other studies. Questions from these other surveys were combined, modified, and reorganised to suit the purposes of this study and to answer the specific questions we set out to explore. Some of the questionnaires were published as the final product of validation studies of anxiety scales (Doyle-Waters, 1994; Horowitz et al., 1979; Watson et al., 1988). Others were in the context of evaluating patients’ knowledge of the maternal serum screen (Gekas et al., 1999; Goel et al., 1996), patients’ Table II. Demographics
Age (years) [Mean (SD)] Education level Completed high school Some college College graduate University undergraduate University graduate degree(s) Number of pregnancies [Mean (SD)] Ethnic background Caucasian Japanese Indo-Canadian Portuguese Number of days elapsed between counseling and completing questionnaire [Mean (SD)]
Telephone group (n = 12)
Clinic visit group (n = 12)
30.3 (3.58)
31.6 (4.19)
1 2 5 1 3 1.8 (0.94)
2 2 2 1 5 1.9 (1.31)
12 0 0 0 13.5 (6.79)
9 1 1 1 12.2 (11)
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Table III. Results of the Knowledge Portion of the Written Questionnaire Completed After Genetic Counseling. Possible Answers Are True or Falsea Mean (SD) Telephone group (n = 12)
Questions The purpose of the triple screen is to identify pregnancies that are at a higher risk for carrying a fetus with Down syndrome and/or a neural tube defect (spina bifida). If the triple screen result is abnormal, something is usually wrong with the baby. Women who have a normal triple screen result are guaranteed a normal baby. If the triple screen result is positive for Down syndrome, the only way to get a definitive answer prenatally is to have amniocentesis. The amniocentesis test carries a 0.5–1% risk of miscarriage due to the procedure itself. Given my positive triple screen result, the chance that I am carrying a fetus with Down syndrome is greater than the chance that I am carrying a normal fetus. My triple screen result indicated that my risk for carrying a fetus with Down syndrome is now higher than my risk based only on my age. If the results of the amniocentesis test are normal, I am guaranteed a completely normal baby. Even if the amniocentesis test result is normal, there is still a chance that I am carrying a fetus with Down syndrome. Total score/9 a Each
Clinic visit group (n = 12)
12 (0)
12 (0)
11 (0.29)
12 (0)
11 (0.29)
12 (0)
12 (0)
11 (0.29)
12 (0)
12 (0)
10 (0.39)
8 (0.49)
12 (0)
12 (0)
11 (0.29)
12 (0)
8 (0.49)
8.3 (1.2)
9 (0.45)
8.3 (0.50)
correct response was given one point.
anxiety about the test (Abuelo et al., 1991), or both (Quagliarini et al., 1998). Other studies looked at informed consent in genetic counseling for prenatal screening (Marteau, 1995) and women’s opinions about prenatal screening (Kornman et al., 1997). The reading level of the questions was set at a Grade 8 level using Microsoft Word software. The final versions of the questions were reviewed for content and understandability by five genetic counseling students. RESULTS A total of 54 patients were eligible to participate in the study. Of those, 30 patients agreed to participate. From the 30 patients that agreed to participate in the study, 24 questionnaires were returned. Because of the small number of patients in each study group, there was insufficient power to conduct a formal statistical analysis. Therefore, results consist of descriptive statistics. The two groups were similar in terms of their ages, the number of pregnancies, ethnic background,
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Table IV. Results of the Anxiety Portion of the Written Questionnaire Completed After Genetic Counselinga Mean (SD) Questions I was feeling very anxious about my positive triple screen result prior to the genetic counseling session. I have been feeling very anxious about my positive triple screen result since my genetic counseling session. I have difficulty falling asleep or staying asleep due to images or thoughts of having a baby with a genetic abnormality. The genetic counseling session helped to ease some of my fears about my risk of having a baby with Down syndrome. I become very emotional when I think of how high my risk is of having a baby with some kind of birth defect. I feel that the information presented to me in my genetic counseling session made me more worried about my pregnancy than I was before the session. I think that I will be nervous about my pregnancy until my baby is born (or until my amniocentesis test result comes back normal). I often have strong feelings of distress and/or fear related to my pregnancy and its outcome. I wish I hadn’t had the triple screen test because it has made me very anxious about my risk. I feel satisfied with the genetic counseling session that I received, and I feel that all my questions were answered. Total score/40
Telephone group (n = 12)
Clinic visit group (n = 12)
3.4 (0.52)
3.8 (0.45)
1.8 (1.2)
2.4 (0.90)
1 (1.1)
1.5 (1.1)
1.2 (1.0)
1.2 (1.1)
2.1 (1.0)
2.8 (1.2)
0.5 (0.80)
0.5 (0.91)
2.8 (0.84)
2.9 (1.3)
1.8 (0.72)
2.3 (1.4)
2.4 (1.3)
2.4 (1.7)
0.6 (1.2)
0.3 (0.50)
17.7 (3.8)
20 (5.5)
a Possible
answers were strongly agree, agree somewhat, not sure, disagree somewhat, and strongly disagree. Numerical anxiety scores were imposed on these answers for the analysis, with 4 points given for the highest anxiety answer, and 0 points for the lowest anxiety answer.
education level, and the number of days that had lapsed between their genetic counseling session and the time that they completed the written questionnaire (Table II). There was no difference seen between the telephone group and the clinic visit group in the mean triple screen risk for Down syndrome or in the number of patients in each group who chose to have amniocentesis after completing their genetic counseling session (Table V). Table V. Number of Patients in Each Group Who Chose to Undergo Amniocentesis After Genetic Counseling Amniocentesis
Telephone group (n = 12)
Clinic visit group (n = 12)
Yes No
9 3
8 4
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Precounseling anxiety. See Table I for the two questions used to assess the patients’ level of anxiety prior to the genetic counseling session. The answers to the second question were translated into a 10-point scale, with constantly being equal to 10 points (for highest anxiety) and never being equal to 0 points (for no anxiety). The mean and standard deviation of precounseling anxiety scores are also shown in Table I. Based on these two questions the level of anxiety or worry between women choosing telephone over face-to-face counselling appears to be similar. Postcounseling knowledge. Participants were given 1 point for every correct answer and 0 for every incorrect answer in the knowledge portion of the questionnaire for a maximum score of 9 points (Table III). There was no apparent difference between the two groups in the mean score representing understanding of the information presented in the genetic counseling session. Postcounseling anxiety. In order to perform a quantitative analysis of the responses to the anxiety-related questions, a numerical value was imposed on the 5-point Likert scale. For each of the 10 questions, a score of 4 was assigned to the response indicating the highest anxiety answer, and 0 points were assigned to the lowest anxiety answer, with intermediate answers given a proportional number of points. Thus, on this portion of the survey a patient’s maximum score is 40 points indicating the greatest possible anxiety, with a minimum score of 0 indicating no anxiety regarding the pregnancy. The results of this section of the written questionnaire are shown in Table IV. There was an open-ended section after each of the knowledge and anxiety portions of the written questionnaire. Here, participants were given the chance to write any comments regarding the genetic counseling they received, or their anxiety surrounding their pregnancy, or anything else relating to the triple screen test. Generally, patients expressed a high regard for the genetic counselor involved in their care. One patient who received genetic counseling by telephone wrote: “I received counseling over the phone, which was very helpful, informative, listening ear, clear and understandable, professional. As good as going in for an appointment (in my case!)” There was repeated mention (five comments) of the extremely high anxiety present in the time period between learning of their positive result and having their genetic counseling session, and usually some relief after genetic counseling. One patient who had a face-to-face genetic counseling appointment wrote: “I would have preferred the counseling session to be available almost immediately after receiving the positive result. I personally found out Thursday at lunch—counseling session was Tuesday afternoon—the weekend was awful and high anxiety. Thank you.” A few participants (four comments) expressed negative feelings about the maternal serum screen itself. All together 18 patients made comments in the open-ended section of the questionnaire (10 from the Clinic visit group and 8 from the Telephone group). Nine of the comments were positive expressions related to the helpfulness of the genetic counselor. The remaining comments were
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approximately evenly split between (1) negative feelings about the MSS test itself, (2) the distressingly high anxiety level in the time period between learning of the positive screen result and the genetic counseling session, and 3) miscellaneous comments. None of the patients who received genetic counseling by telephone requested a face-to-face genetic counseling session. DISCUSSION In this small pilot study we detected no large differences in understanding or anxiety when genetic counseling is conducted by telephone compared to a clinic visit. Both groups of patients demonstrated a reduction in anxiety after genetic counseling. Both groups of patients indicated a high level of satisfaction with the genetic counseling they received. However, there was an interesting trend toward lower anxiety in the group of patients who chose to have genetic counseling by telephone, both before and after their genetic counseling session. It is possible that less anxious patients are more likely to choose telephone genetic counseling, and to have less anxiety after counseling. Because of the small sample size and limitations related to the study design it is not possible to determine if patients who chose telephone counseling would have experienced an even greater drop in anxiety after counseling in person. In general, the difference between groups in any one measure of knowledge or anxiety was very small. Given that the number of patients that returned the questionnaire in each group was so small, even large differences would be difficult to interpret. The small groups were mainly due to the time frame of the study and the strict inclusion criteria that we set at the beginning in order to avoid as many confounding factors as possible. Since the data in the present study have shown that there is most likely not a significant reduction in quality between telephone and face-to-face genetic counseling, it is safe to initiate a larger study that includes patients throughout the province and occurs over a longer period of time. A randomized trial would be most useful; however, given our experience with attempted randomization, it is questionable that enough patients would agree to participate in the midst of such an anxiety provoking experience to make the study worthwhile. Participation in a study giving patients a choice of groups may be more agreeable as the participant has a sense of control over the situation, whereas being randomized to one group or the other may be perceived as a loss of control in an already stressful circumstance. In a study of 72 individuals or couples receiving genetic counseling in Israel, Shiloh et al. (1997) found that patients with higher perceived personal control reported higher satisfaction with genetic counseling and had better coping strategies. A previous study in the area of prenatal screening, while not specifically testing the provision of genetic counseling by telephone, found that information
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regarding the results of maternal serum screening is particularly misunderstood when relayed by telephone, compared with results given in a face-to-face consultation (Gekas et al., 1999). In the same study, a telephone message was found to be superior to communication by letter for this purpose. In that study, results were given to patients by either an obstetrician or a general physician, not a genetic counselor. Thus, less time may have been taken to explain the results of the test than in a scheduled genetic counseling session lasting 45 min to 1 h, which was the case in the current study. The difference in the health care provider involved may explain the difference in the results of the previous study and the current study. Genetic counselors are specifically trained to provide information in a number of different ways, using simple and easy to understand language, and to verify that the patient has understood the information using a variety of techniques. There are a number of other issues that must be taken into consideration when health counseling by telephone is being studied. One is the perceived legitimacy of telephone work in genetic counseling both on the part of the patient and the counselor (Wilson and Williams, 2000). Skills that are learned and practised in a face-to-face context may not be easily translated to the realm of telephone contact. If the patient senses a counselor’s discomfort with the mode of counseling, she may feel less confident in the information being provided. A counselor’s confidence in the patient’s verbal skills is also a factor. If the counselor is concerned that a patient is having trouble expressing herself or is being untruthful, the ability to elucidate the information and discuss concerns interactively over the telephone may not come as naturally as in a face-to-face counseling situation. It may take specific training and practice for genetic counselors to feel comfortable conducting an entire genetic counseling session over the telephone. The lack of visual cues would also be expected to affect the counseling session. Although being able to “read” a patient’s body language is generally considered an advantage, it can be objectifying of the patient, placing her in a position of less power than the health care provider (Wilson and Williams, 2000). Dependence on visual cues may result in the counselor inadvertently making judgements about the patient based on appearance (although this is clearly opposed to the counselor’s training and purpose), and increases the potential for the counselor to misread or misinterpret a patient’s body language. Telephone counseling may have the advantage of preventing these potential problems. Thus, it could be considered easier to counsel patients over the telephone, as it is impossible to fall into those traps based on visual cues. LIMITATIONS The current study is limited by the self-selection of the two groups. The women who chose telephone genetic counseling may not be similar to those women
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who chose a face-to-face session in terms of general anxiety level in everyday life, in knowledge of screening tests, and/or in comfort level with the use of the telephone for personal communication. However, we have no reason to believe that the patients who selected telephone genetic counseling in the study are any different in this regard than the patients who would select this mode of counseling in a clinical situation. The women in the telephone counseling group were similar in their levels of anxiety and knowledge about the triple screen to the women who choose face-to-face counseling. Thus, while the self-selection is a limitation we believe it is representative of the clinical scenario in which the two modes of genetic counseling would be offered as choices rather than either one being imposed on particular patients. Although there was no apparent difference between the two groups related to education, the participants may not be representative of the general population. The patients who agreed to participate in the study had a higher average education level than the general population, regardless of which group they chose. In Canada, the average number of years of schooling completed by individuals aged 15 years and older in 1996 was 12.3 years (Statistics Canada: see references). In BC in the same year, this number was 12.6 years. There is the possibility that the lack of difference in knowledge between the two groups is not applicable to the rest of the population of BC. This would best be determined with a large randomized trial that includes patients from a variety of geographical areas and socioeconomic backgrounds. Generally, questionnaires are pilot tested and validated for comprehensibility, content validity, test-retest reliability, ambiguity, readability, and length (Goel et al., 1996). Because of the specificity of the counseling situation in this study we modified versions of questions used in other, validated surveys. These revisions may have affected the validity of the items. We also developed the items in the knowledge portion of the survey based upon the primary measurable goals of a genetic counseling session for a positive triple screen for Down syndrome as determined by the authors. Although the lack of standardized validated measures is a study limitation, the items used in this study were either chosen or developed to address this counseling scenario. Thus, correct answers to these questions represent successful provision and understanding of information in the genetic counseling session. CONCLUSION In this, the first study to assess the effectiveness of genetic counseling by telephone compared to a clinic visit, we provide at least preliminary evidence that it is feasible to offer telephone genetic counseling as a service, albeit with caution. It may be possible to expand this service to genetic counseling for more complex indications. Based on these findings additional research is indicated.
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In future studies the role of choice related to precounseling anxiety should be explored. An intriguing finding from this study was that patient participation increased when offered a choice of mode of genetic counseling. Research is also needed to ascertain if the option of telephone counseling provides increased access to genetic counseling, and whether this option increases the likelihood of including a partner in the session. Additionally, the economic costs and benefits to telephone genetic counseling needs to be addressed. Some of the factors that are likely to vary in face-to-face versus telephone counseling are time lost from work, cost of travel, and reimbursement issues. The net savings of a telephone service, both in dollars and in quality of health care, are very difficult to quantify in the context of a complex health care system (Hallam, 1989) and this is reflected in the scarcity of published studies on the subject. These issues, while beyond the scope of this pilot study, are important factors in implementing a new service option in any field. The findings from this preliminary study of telephone genetic counseling are encouraging and will hopefully serve as a staring place for additional research into alternative modes of genetic counseling.
REFERENCES Abuelo DN, Hopmann MR, Barsel-Bowers G, Goldstein A (1991) Anxiety in women with low maternal serum alpha-fetoprotein screening results. Prenat Diagn 11:381–385. Bernhardt BA, Biesecker BB, Mastromarino CL (2000) Goals, benefits and outcomes of genetic counseling: Client and genetic counselor assessment. Am J Med Genet 94:189–197. Carroll JC (1994) Maternal serum screening. Can Fam Physician 40:1756–1764. Doyle-Waters MMR (1994) Anxiety scale for pregnancy: Development and validation. MA Thesis, University of British Columbia, Vancouver. Gekas J, Gondry J, Mazur S, Cesbron P, Thepot F (1999) Informed consent to serum screening for Down syndrome: Are women given adequate information? Prenat Diagn 19:1–7. Goel V, Glazier R, Holzapfel S, Pugh P, Summers A (1996) Evaluating patient’s knowledge of maternal serum screening. Prenat Diagn 16:425–430. Hallam L (1989) You’ve got a lot to answer for, Mr. Bell. A review of the use of the telephone in primary care. Fam Pract 6(1):47–75. Hornblow AR (1986) The evolution and effectiveness of telephone counseling services. Hosp Community Psychiatry 37(7):731–733. Horowitz M, Wilner N, Alvarez W (1979) Impact of Events scale: A measure of subjective stress. Psychosom Med 41(3):209–218. Koren G, Graham K, Feigenbaum A, Einarson T (1993) Evaluation and counseling of teratogenic risk: The Motherisk approach. J Clin Pharmacol 33:405–411. Kornman LH, Wortelboer MJM, Beekhuis JR, Morssink LP, Mantingh A (1997) Women’s opinions and the implications of first- versus second-trimester screening for fetal Down’s syndrome. Prenat Diagn 17(11):1011–1018. Li KC, Lai SS, Lam ST (1999) Adequacy and pitfalls of G6PD deficiency counseling in Hong Kong. Southeast Asian J Trop Med Public Health 30(Suppl 2): 79–83. Lynch DL, Tamburrino MB, Nagel R (1997) Telephone counseling for patients with minor depression: Preliminary findings in a family practice setting. J Fam Pract 44(3):293–298. Marteau TM (1995) Towards informed decisions about prenatal testing: A review. Prenat Diagn 15:1215–1226. McBride CM, Rimer BK (1999) Using the telephone to improve health behavior and health service delivery. Patient Educ Counsel 37:3–18.
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Ormond KE, Haun J, Cook L, Duquette D, Ludowese C, Matthews AL (2000) Recommendations for telephone counseling. J Genet Counsel 9(1):63–71. Quagliarini D, Betti S, Brambati B, Nicolini U (1998) Coping with serum screening for Down syndrome when the result is given as a numeric value. Prenat Diagn 18:816–821. Rayburn W, Wilson G, Schreck J, Louwsma G, Hamman J (1982) Prenatal counseling: A state-wide telephone service. Obstet Gynecol 60(2):243–246. Shiloh S, Berkenstadt M, Meiran N, Bat-Miriam-Katznelson M, Goldman B (1997) Mediating effects of perceived personal control in coping with a health threat: The case of genetic counseling. J Appl Soc Psychol 27:1146–1174. Soet JE, Basch CE (1997) The telephone as a communication medium for health education. Health Educ Behav 24(6):759–772. Statistics Canada Web site: http://www.statcan.ca/english/Pgdb/People/Education/educ44a.htm Tsu RC, Burm ML, Jennifer AG, Sells CW (2002) Telephone vs. face-to-face notification of HIV results in high-risk youth. J Adolesc Health 30:154–160. Watson D, Clark LA, Tellegen A (1988) Development and validation of brief measures of positive and negative affect: The PANAS scales. J Pers Soc Psychol 54(6):1063–1070. Wilson K, Williams A (2000) Visualism in community nursing: Implications for telephone work with service users. Qual Health Res 10(4):507–520.
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Assessment of the Effectiveness of Genetic Counseling by Telephone Compared to a Clinic Visit Karan K. Sangha,1,3 Anita Dircks,2 and Sylvie Langlois2
Maternal serum screening, also known as the triple screen, is used during pregnancy to assess the risk of carrying a fetus with specific chromosome abnormalities or open spina bifida. All women in British Columbia who screen positive are eligible for genetic counseling and are offered amniocentesis. The purpose of this study is to determine what differences (if any) exist in patients’ understanding and/or anxiety when genetic counseling for a positive triple screen is conducted in person versus over the telephone. Each patient who participated was given the choice of having genetic counseling in person or over the telephone, this after a randomized design failed to elicit any participants. Using a written postcounseling questionnaire, each patient was assessed for her understanding of the information presented in the session, and her anxiety regarding her risk. In this small pilot study, no large differences were detected in patients’ understanding or anxiety when genetic counseling was conducted by telephone versus in person. KEY WORDS: genetic counseling; telephone; maternal serum screen; knowledge; anxiety.
INTRODUCTION The triple screen (or maternal serum screen) is a blood test offered to pregnant women between 15 and 20 weeks of gestation to assess the chance of carrying a fetus with specific chromosome abnormalities or an open neural tube defect (Carroll, 1994). The test measures the concentrations of three proteins (human chorionic gonadotropin, alpha-fetoprotein, and unconjugated estriol) in the pregnant woman’s 1 Medical
Genetics Clinic, University of Alberta Hospital, Edmonton, Alberta, Canada.
2 Provincial Medical Genetics Programme, Children’s and Women’s Health Centre of British Columbia
and Department of Medical Genetics, University of British Columbia, Vancouver, British Columbia, Canada. 3 Correspondence should be directed to Karan Sangha, 8-53 Medical Sciences Building, University of Alberta Hospital, 8440 112 Street, Edmonton, Alberta, Canada T6G 2H7; e-mail: [email protected]. 171 C 2003 National Society of Genetic Counselors, Inc. 1059-7700/03/0400-0171/1 °
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serum and compares these with the average concentrations in the general population of pregnant women at the specific time in gestation that the blood is collected. These figures are then used to estimate the chance of the fetus being affected with trisomy 18, trisomy 21, and open spina bifida. A positive screen for Down syndrome is defined as a risk greater than or equal to 1/385. This figure has been chosen as the cutoff because this risk is approximately equal to the risk of trisomy 21 at a maternal age of 35 years. Using the 1/385 cutoff, the triple screen test has the ability to detect 72% of pregnancies with Down syndrome. Of all the women who screen positive for trisomy 21 using the maternal serums screen, only 2.8% are actually carrying a fetus with Down syndrome. The chance of screening negative if the fetus does not have trisomy 21 is approximately 91% (BC Children’s and Women’s Health Centre GEM Program, unpublished data). Currently in British Columbia (BC), every woman who has a positive triple screen is referred to the Provincial Medical Genetics Programme in Vancouver for genetic counseling and is offered amniocentesis. Since the triple screen became available as a clinical test in 1995 in BC, the number of women screened is rising every year, presumably due to increasing awareness of the test among primary care physicians. Approximately 15,000 women were screened using the triple screen test in 2000 in BC. Eight percent of these women screened positive for Down syndrome. Thus, about 1200 women screened positive in that year. At least 25% of these women (300 women per year) live outside of the Greater Vancouver area (Dr. Catherine Halstead, unpublished data) and therefore must travel a significant distance on short notice to follow up on a positive result. Currently, the only choice available to patients who reside outside of the Greater Vancouver area is to travel to Vancouver for a face-to-face appointment with a genetic counselor or to decline genetic counseling. Therefore, there is a need to explore alternative methods of providing genetic counseling such as telephone counseling. Possible advantages of telephone counseling include avoidance of the stress, expense and time commitment of a clinic visit, and reduction in barriers to service by increasing accessibility (Tsu et al., 2002). These issues are compounded for patients traveling from out of town. A telephone genetic counseling service has the ability to increase access to care for patients in geographically isolated areas. In addition, the visual privacy of telephone consultation may allow the patient to feel safe and comfortable expressing her feelings in an open manner, making the interaction more productive (Wilson and Williams, 2000). There are also some potential disadvantages of telephone genetic counseling. Rapport building between the counselor and patient may be hindered by the lack of visual cues available to each party. It is estimated that 65–93% of social meaning between individuals is in the form of nonverbal cues (Soet and Basch, 1997). In addition, one might speculate that the inability of the genetic counselor to use visual aides such as diagrams in providing information may result in a lower level
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of understanding on the part of the patient. Soet and Basch (1997) have also shown that visual learning is preferred over auditory learning. Thus, conveying complex information over the telephone may be more challenging than doing so in a faceto-face context. Telephone contact may also have the effect of an impersonal or anonymous interaction between patient and counselor, impeding the emotional connection. This could result in a higher anxiety level for the patient following telephone counseling compared with counseling in person. In our review of the literature we were able to identify two studies that focused on assessing the effectiveness of telephone genetic counseling. Rayburn et al. (1982) evaluated the use of a toll-free telephone hot line designed to provide prenatal counseling for various indications such as teratogen exposure, obstetric complications, genetic conditions, and other prenatal concerns. Outcome measures were the number of patients using the service, the number of patients undergoing amniocentesis, and feedback from physicians. A steady increase in the number of callers, and the number of amniocenteses performed was found, as well as positive feedback from physicians. Li et al. (1999) assessed a telephone counseling program for parents of children diagnosed with G6PD deficiency through newborn screening in Hong Kong. Success of the program was determined by patients’ satisfaction with counseling and retention of information as ascertained by a questionnaire developed by the investigators. The authors concluded that telephone counseling was the method of choice. However, neither of these studies used a control or comparison group. Although there are few published studies on telephone genetic counseling, a 1993 survey found that 26 out of 26 genetic counselors in the United States reported having done some genetic counseling by telephone (Ormond et al., 2000). The effectiveness of telephone counseling in contexts other than genetic counseling has been assessed by various methods. The delivery of health care by telephone has been studied in general practice (McBride and Rimer, 1999), pediatrics (Hallam, 1989), nursing (Wilson and Williams, 2000), obstetrics (Koren et al., 1993; Rayburn et al., 1982), psychology (Hornblow, 1986), and psychiatry (Lynch et al., 1997). Many of these studies evaluated telephone hotlines set up for use by the general public, or focused on specific patient populations such as smokers, cancer patients, or individuals at high risk for suicide. The telephone counseling was generally combined with other interventions such as face-to-face consultation, physical examination, written material, home visits, or video instruction or education (McBride and Rimer, 1999) and was provided in the context of the introduction of a new service for patients, in addition to their existing health care program. The outcomes measured in these studies included the number of individuals calling a particular help line, the reduction in the number of office visits, the extent of behavior modification (e.g., the rate of smoking cessation within a given time frame), and in one case a user questionnaire to assess the value of the service to the individual in the absence of any alternatives.
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Telephone counseling versus no counseling was evaluated in a study by Lynch et al. (1997) of 29 individuals identified to have minor depression. The treatment group received six weekly 20-min telephone counseling sessions, while the comparison group received no counseling. The outcome measured was the difference in scores on the Hamilton Depression Rating Scale before and after the series of counseling sessions. In this study, the telephone counseled group was found to have a reduction in depression. Generally, prior studies evaluating the usefulness of telephone health care delivery have not taken into consideration the effect of telephone intervention in place of a face-to-face visit. Instead, they attempt to assess the value of adding a telephone-based intervention to the range of medical services available to a particular patient population. Although findings from this research suggest that telephone counseling may be effective, this research is limited by the lack of a comparison group receiving face-to-face counseling. This research may also have limited applicability to a genetic counseling session because of differences in goals. The goals of genetic counseling are not to attract the largest number of patients, nor to necessarily modify patients’ behavior. Success in genetic counseling among workers in the profession is generally thought to be indicated by an increase in the patient’s knowledge of genetics, risks and available options, assistance with decision making, help with psychosocial adjustment, and patient satisfaction (Bernhardt et al., 2000). Outcomes relevant to genetic counseling have not been previously studied comparing face-to-face with telephone genetic counseling. The purpose of this study was to compare telephone counseling with face-toface counseling for pregnant women with a positive triple screen. This study was a limited pilot study to evaluate the feasibility of telephone genetic counseling. For the purposes of this study, we identified two main outcomes for prenatal genetic counseling of women with a positive triple screen. The first was an understanding of the information provided by the genetic counselor regarding the results of the triple screen test, further testing options for the pregnancy, and the meaning of the results of further testing. The second objective was a reduction in the level of the patient’s anxiety about her risk for Down syndrome. Therefore, we assessed patients’ understanding, anxiety, and choices regarding additional testing to determine if there were differences in the two groups receiving the two delivery modes of genetic counseling. MATERIALS AND METHODS Prior to commencement of the study approval was granted by the Behavioral Research Ethics Board of the University of British Columbia and the Research Review Committee of the Children’s and Women’s Health Centre of British Columbia. Between October, 2000, and March, 2001, each patient referred to the Provincial Medical Genetics Programme in BC for a positive triple screen for Down syndrome was assessed for the eligibility criteria for participation in the study. In
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order to be eligible to participate in the study the patient had to be referred for genetic counseling for a positive triple screen for Down syndrome with no other indications for genetic assessment or counseling at the time of the referral, the patient’s first language had to be English, the patient’s residence had to be within the lower mainland of British Columbia, no more than a 2-h drive from the Vancouver clinic where genetic counseling is carried out in the province of BC, and where the amniocentesis procedure is performed. Patients who had been seen previously in the Provincial Medical Genetics Programme for genetic counseling were excluded to avoid the possible effect of this on the results of the assessment of knowledge of genetic information. For patients who met all inclusion criteria, the referring physician was contacted by telephone and asked for permission to contact her patient in order to invite her to participate in a study. If the physician gave permission, the patient was then contacted by telephone. She was given a brief verbal introduction to the study and asked if she would like to participate. The only differences between participation and nonparticipation for the patient were (1) instead of automatically going to the clinic for the genetic counseling appointment, participants would have the choice of being seen in the clinic or to have genetic counseling over the telephone; (2) participants were asked to complete a brief written questionnaire after the genetic counseling session to assess their understanding of the information presented in the session and their level of anxiety regarding their risk. All patients who had genetic counseling by telephone were offered the option of a face-to-face genetic counseling session at the conclusion of the telephone counseling session. The study was originally designed as a randomized trial. However, in the first 2 months of soliciting participants, none of the patients contacted agreed to participate in the study, with many citing anxiety as their main reason for declining. Thus the design was changed to a nonrandomized trial to give patients a choice as to the mode of counseling. Consequently, the number of participants increased. Once the design was altered, the most common reasons for nonparticipation were logistical. Therefore, we believe that the sample is representative of our study population. Once a patient agreed to participate, she received genetic counseling in person or over the telephone. One of the authors, a genetic counselor with greater than 10 years of experience in the field (AD) counseled all patients in both groups in order to minimise any variation in counseling technique. Counseling sessions, whether by phone or face-to-face were typically 45–60 min in length. Patients in both groups were given a brief verbal questionnaire consisting of two questions (see Table I) at the beginning of the session to assess their precounseling anxiety level. After the genetic counseling session, patients in both groups were mailed a written questionnaire to be completed and returned to the investigators. The questionnaire consisted of a demographics section (see Table II), a section to assess knowledge (Table III), a section to assess anxiety (Table IV), and an open-ended section for comments. The knowledge portion consisted of nine questions with the patient
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Table I. Results of the Precounseling Verbal Anxiety Survey. Mean and Standard Deviation of Answers Are Shown for Each Group Mean (SD) Telephone group (n = 12)
Questions On a scale of 1 to 10 (where 1 is none and 10 is the most anxiety you have ever felt), how would you rate the amount of anxiety you have been feeling since learning about your positive triple screen result? On a daily basis, how much do you worry about your positive triple screen result? (Constantly (10), very often (8), sometimes (6), not very often (4), almost never (2), never (0). Total score/20
Clinic visit group (n = 12)
7.3 (1.42)
8.5 (1.51)
7.2 (1.8)
7.3 (1.78)
14.5 (3.1)
15.8 (2.8)
being asked to circle “true” or “false” for each answer. The anxiety portion of the questionnaire was made up of 10 questions with the responses in the form of a 5-point Likert scale. The written questionnaire was developed by adapting questions used in other studies. Questions from these other surveys were combined, modified, and reorganised to suit the purposes of this study and to answer the specific questions we set out to explore. Some of the questionnaires were published as the final product of validation studies of anxiety scales (Doyle-Waters, 1994; Horowitz et al., 1979; Watson et al., 1988). Others were in the context of evaluating patients’ knowledge of the maternal serum screen (Gekas et al., 1999; Goel et al., 1996), patients’ Table II. Demographics
Age (years) [Mean (SD)] Education level Completed high school Some college College graduate University undergraduate University graduate degree(s) Number of pregnancies [Mean (SD)] Ethnic background Caucasian Japanese Indo-Canadian Portuguese Number of days elapsed between counseling and completing questionnaire [Mean (SD)]
Telephone group (n = 12)
Clinic visit group (n = 12)
30.3 (3.58)
31.6 (4.19)
1 2 5 1 3 1.8 (0.94)
2 2 2 1 5 1.9 (1.31)
12 0 0 0 13.5 (6.79)
9 1 1 1 12.2 (11)
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Table III. Results of the Knowledge Portion of the Written Questionnaire Completed After Genetic Counseling. Possible Answers Are True or Falsea Mean (SD) Telephone group (n = 12)
Questions The purpose of the triple screen is to identify pregnancies that are at a higher risk for carrying a fetus with Down syndrome and/or a neural tube defect (spina bifida). If the triple screen result is abnormal, something is usually wrong with the baby. Women who have a normal triple screen result are guaranteed a normal baby. If the triple screen result is positive for Down syndrome, the only way to get a definitive answer prenatally is to have amniocentesis. The amniocentesis test carries a 0.5–1% risk of miscarriage due to the procedure itself. Given my positive triple screen result, the chance that I am carrying a fetus with Down syndrome is greater than the chance that I am carrying a normal fetus. My triple screen result indicated that my risk for carrying a fetus with Down syndrome is now higher than my risk based only on my age. If the results of the amniocentesis test are normal, I am guaranteed a completely normal baby. Even if the amniocentesis test result is normal, there is still a chance that I am carrying a fetus with Down syndrome. Total score/9 a Each
Clinic visit group (n = 12)
12 (0)
12 (0)
11 (0.29)
12 (0)
11 (0.29)
12 (0)
12 (0)
11 (0.29)
12 (0)
12 (0)
10 (0.39)
8 (0.49)
12 (0)
12 (0)
11 (0.29)
12 (0)
8 (0.49)
8.3 (1.2)
9 (0.45)
8.3 (0.50)
correct response was given one point.
anxiety about the test (Abuelo et al., 1991), or both (Quagliarini et al., 1998). Other studies looked at informed consent in genetic counseling for prenatal screening (Marteau, 1995) and women’s opinions about prenatal screening (Kornman et al., 1997). The reading level of the questions was set at a Grade 8 level using Microsoft Word software. The final versions of the questions were reviewed for content and understandability by five genetic counseling students. RESULTS A total of 54 patients were eligible to participate in the study. Of those, 30 patients agreed to participate. From the 30 patients that agreed to participate in the study, 24 questionnaires were returned. Because of the small number of patients in each study group, there was insufficient power to conduct a formal statistical analysis. Therefore, results consist of descriptive statistics. The two groups were similar in terms of their ages, the number of pregnancies, ethnic background,
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Table IV. Results of the Anxiety Portion of the Written Questionnaire Completed After Genetic Counselinga Mean (SD) Questions I was feeling very anxious about my positive triple screen result prior to the genetic counseling session. I have been feeling very anxious about my positive triple screen result since my genetic counseling session. I have difficulty falling asleep or staying asleep due to images or thoughts of having a baby with a genetic abnormality. The genetic counseling session helped to ease some of my fears about my risk of having a baby with Down syndrome. I become very emotional when I think of how high my risk is of having a baby with some kind of birth defect. I feel that the information presented to me in my genetic counseling session made me more worried about my pregnancy than I was before the session. I think that I will be nervous about my pregnancy until my baby is born (or until my amniocentesis test result comes back normal). I often have strong feelings of distress and/or fear related to my pregnancy and its outcome. I wish I hadn’t had the triple screen test because it has made me very anxious about my risk. I feel satisfied with the genetic counseling session that I received, and I feel that all my questions were answered. Total score/40
Telephone group (n = 12)
Clinic visit group (n = 12)
3.4 (0.52)
3.8 (0.45)
1.8 (1.2)
2.4 (0.90)
1 (1.1)
1.5 (1.1)
1.2 (1.0)
1.2 (1.1)
2.1 (1.0)
2.8 (1.2)
0.5 (0.80)
0.5 (0.91)
2.8 (0.84)
2.9 (1.3)
1.8 (0.72)
2.3 (1.4)
2.4 (1.3)
2.4 (1.7)
0.6 (1.2)
0.3 (0.50)
17.7 (3.8)
20 (5.5)
a Possible
answers were strongly agree, agree somewhat, not sure, disagree somewhat, and strongly disagree. Numerical anxiety scores were imposed on these answers for the analysis, with 4 points given for the highest anxiety answer, and 0 points for the lowest anxiety answer.
education level, and the number of days that had lapsed between their genetic counseling session and the time that they completed the written questionnaire (Table II). There was no difference seen between the telephone group and the clinic visit group in the mean triple screen risk for Down syndrome or in the number of patients in each group who chose to have amniocentesis after completing their genetic counseling session (Table V). Table V. Number of Patients in Each Group Who Chose to Undergo Amniocentesis After Genetic Counseling Amniocentesis
Telephone group (n = 12)
Clinic visit group (n = 12)
Yes No
9 3
8 4
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Precounseling anxiety. See Table I for the two questions used to assess the patients’ level of anxiety prior to the genetic counseling session. The answers to the second question were translated into a 10-point scale, with constantly being equal to 10 points (for highest anxiety) and never being equal to 0 points (for no anxiety). The mean and standard deviation of precounseling anxiety scores are also shown in Table I. Based on these two questions the level of anxiety or worry between women choosing telephone over face-to-face counselling appears to be similar. Postcounseling knowledge. Participants were given 1 point for every correct answer and 0 for every incorrect answer in the knowledge portion of the questionnaire for a maximum score of 9 points (Table III). There was no apparent difference between the two groups in the mean score representing understanding of the information presented in the genetic counseling session. Postcounseling anxiety. In order to perform a quantitative analysis of the responses to the anxiety-related questions, a numerical value was imposed on the 5-point Likert scale. For each of the 10 questions, a score of 4 was assigned to the response indicating the highest anxiety answer, and 0 points were assigned to the lowest anxiety answer, with intermediate answers given a proportional number of points. Thus, on this portion of the survey a patient’s maximum score is 40 points indicating the greatest possible anxiety, with a minimum score of 0 indicating no anxiety regarding the pregnancy. The results of this section of the written questionnaire are shown in Table IV. There was an open-ended section after each of the knowledge and anxiety portions of the written questionnaire. Here, participants were given the chance to write any comments regarding the genetic counseling they received, or their anxiety surrounding their pregnancy, or anything else relating to the triple screen test. Generally, patients expressed a high regard for the genetic counselor involved in their care. One patient who received genetic counseling by telephone wrote: “I received counseling over the phone, which was very helpful, informative, listening ear, clear and understandable, professional. As good as going in for an appointment (in my case!)” There was repeated mention (five comments) of the extremely high anxiety present in the time period between learning of their positive result and having their genetic counseling session, and usually some relief after genetic counseling. One patient who had a face-to-face genetic counseling appointment wrote: “I would have preferred the counseling session to be available almost immediately after receiving the positive result. I personally found out Thursday at lunch—counseling session was Tuesday afternoon—the weekend was awful and high anxiety. Thank you.” A few participants (four comments) expressed negative feelings about the maternal serum screen itself. All together 18 patients made comments in the open-ended section of the questionnaire (10 from the Clinic visit group and 8 from the Telephone group). Nine of the comments were positive expressions related to the helpfulness of the genetic counselor. The remaining comments were
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approximately evenly split between (1) negative feelings about the MSS test itself, (2) the distressingly high anxiety level in the time period between learning of the positive screen result and the genetic counseling session, and 3) miscellaneous comments. None of the patients who received genetic counseling by telephone requested a face-to-face genetic counseling session. DISCUSSION In this small pilot study we detected no large differences in understanding or anxiety when genetic counseling is conducted by telephone compared to a clinic visit. Both groups of patients demonstrated a reduction in anxiety after genetic counseling. Both groups of patients indicated a high level of satisfaction with the genetic counseling they received. However, there was an interesting trend toward lower anxiety in the group of patients who chose to have genetic counseling by telephone, both before and after their genetic counseling session. It is possible that less anxious patients are more likely to choose telephone genetic counseling, and to have less anxiety after counseling. Because of the small sample size and limitations related to the study design it is not possible to determine if patients who chose telephone counseling would have experienced an even greater drop in anxiety after counseling in person. In general, the difference between groups in any one measure of knowledge or anxiety was very small. Given that the number of patients that returned the questionnaire in each group was so small, even large differences would be difficult to interpret. The small groups were mainly due to the time frame of the study and the strict inclusion criteria that we set at the beginning in order to avoid as many confounding factors as possible. Since the data in the present study have shown that there is most likely not a significant reduction in quality between telephone and face-to-face genetic counseling, it is safe to initiate a larger study that includes patients throughout the province and occurs over a longer period of time. A randomized trial would be most useful; however, given our experience with attempted randomization, it is questionable that enough patients would agree to participate in the midst of such an anxiety provoking experience to make the study worthwhile. Participation in a study giving patients a choice of groups may be more agreeable as the participant has a sense of control over the situation, whereas being randomized to one group or the other may be perceived as a loss of control in an already stressful circumstance. In a study of 72 individuals or couples receiving genetic counseling in Israel, Shiloh et al. (1997) found that patients with higher perceived personal control reported higher satisfaction with genetic counseling and had better coping strategies. A previous study in the area of prenatal screening, while not specifically testing the provision of genetic counseling by telephone, found that information
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regarding the results of maternal serum screening is particularly misunderstood when relayed by telephone, compared with results given in a face-to-face consultation (Gekas et al., 1999). In the same study, a telephone message was found to be superior to communication by letter for this purpose. In that study, results were given to patients by either an obstetrician or a general physician, not a genetic counselor. Thus, less time may have been taken to explain the results of the test than in a scheduled genetic counseling session lasting 45 min to 1 h, which was the case in the current study. The difference in the health care provider involved may explain the difference in the results of the previous study and the current study. Genetic counselors are specifically trained to provide information in a number of different ways, using simple and easy to understand language, and to verify that the patient has understood the information using a variety of techniques. There are a number of other issues that must be taken into consideration when health counseling by telephone is being studied. One is the perceived legitimacy of telephone work in genetic counseling both on the part of the patient and the counselor (Wilson and Williams, 2000). Skills that are learned and practised in a face-to-face context may not be easily translated to the realm of telephone contact. If the patient senses a counselor’s discomfort with the mode of counseling, she may feel less confident in the information being provided. A counselor’s confidence in the patient’s verbal skills is also a factor. If the counselor is concerned that a patient is having trouble expressing herself or is being untruthful, the ability to elucidate the information and discuss concerns interactively over the telephone may not come as naturally as in a face-to-face counseling situation. It may take specific training and practice for genetic counselors to feel comfortable conducting an entire genetic counseling session over the telephone. The lack of visual cues would also be expected to affect the counseling session. Although being able to “read” a patient’s body language is generally considered an advantage, it can be objectifying of the patient, placing her in a position of less power than the health care provider (Wilson and Williams, 2000). Dependence on visual cues may result in the counselor inadvertently making judgements about the patient based on appearance (although this is clearly opposed to the counselor’s training and purpose), and increases the potential for the counselor to misread or misinterpret a patient’s body language. Telephone counseling may have the advantage of preventing these potential problems. Thus, it could be considered easier to counsel patients over the telephone, as it is impossible to fall into those traps based on visual cues. LIMITATIONS The current study is limited by the self-selection of the two groups. The women who chose telephone genetic counseling may not be similar to those women
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who chose a face-to-face session in terms of general anxiety level in everyday life, in knowledge of screening tests, and/or in comfort level with the use of the telephone for personal communication. However, we have no reason to believe that the patients who selected telephone genetic counseling in the study are any different in this regard than the patients who would select this mode of counseling in a clinical situation. The women in the telephone counseling group were similar in their levels of anxiety and knowledge about the triple screen to the women who choose face-to-face counseling. Thus, while the self-selection is a limitation we believe it is representative of the clinical scenario in which the two modes of genetic counseling would be offered as choices rather than either one being imposed on particular patients. Although there was no apparent difference between the two groups related to education, the participants may not be representative of the general population. The patients who agreed to participate in the study had a higher average education level than the general population, regardless of which group they chose. In Canada, the average number of years of schooling completed by individuals aged 15 years and older in 1996 was 12.3 years (Statistics Canada: see references). In BC in the same year, this number was 12.6 years. There is the possibility that the lack of difference in knowledge between the two groups is not applicable to the rest of the population of BC. This would best be determined with a large randomized trial that includes patients from a variety of geographical areas and socioeconomic backgrounds. Generally, questionnaires are pilot tested and validated for comprehensibility, content validity, test-retest reliability, ambiguity, readability, and length (Goel et al., 1996). Because of the specificity of the counseling situation in this study we modified versions of questions used in other, validated surveys. These revisions may have affected the validity of the items. We also developed the items in the knowledge portion of the survey based upon the primary measurable goals of a genetic counseling session for a positive triple screen for Down syndrome as determined by the authors. Although the lack of standardized validated measures is a study limitation, the items used in this study were either chosen or developed to address this counseling scenario. Thus, correct answers to these questions represent successful provision and understanding of information in the genetic counseling session. CONCLUSION In this, the first study to assess the effectiveness of genetic counseling by telephone compared to a clinic visit, we provide at least preliminary evidence that it is feasible to offer telephone genetic counseling as a service, albeit with caution. It may be possible to expand this service to genetic counseling for more complex indications. Based on these findings additional research is indicated.
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In future studies the role of choice related to precounseling anxiety should be explored. An intriguing finding from this study was that patient participation increased when offered a choice of mode of genetic counseling. Research is also needed to ascertain if the option of telephone counseling provides increased access to genetic counseling, and whether this option increases the likelihood of including a partner in the session. Additionally, the economic costs and benefits to telephone genetic counseling needs to be addressed. Some of the factors that are likely to vary in face-to-face versus telephone counseling are time lost from work, cost of travel, and reimbursement issues. The net savings of a telephone service, both in dollars and in quality of health care, are very difficult to quantify in the context of a complex health care system (Hallam, 1989) and this is reflected in the scarcity of published studies on the subject. These issues, while beyond the scope of this pilot study, are important factors in implementing a new service option in any field. The findings from this preliminary study of telephone genetic counseling are encouraging and will hopefully serve as a staring place for additional research into alternative modes of genetic counseling.
REFERENCES Abuelo DN, Hopmann MR, Barsel-Bowers G, Goldstein A (1991) Anxiety in women with low maternal serum alpha-fetoprotein screening results. Prenat Diagn 11:381–385. Bernhardt BA, Biesecker BB, Mastromarino CL (2000) Goals, benefits and outcomes of genetic counseling: Client and genetic counselor assessment. Am J Med Genet 94:189–197. Carroll JC (1994) Maternal serum screening. Can Fam Physician 40:1756–1764. Doyle-Waters MMR (1994) Anxiety scale for pregnancy: Development and validation. MA Thesis, University of British Columbia, Vancouver. Gekas J, Gondry J, Mazur S, Cesbron P, Thepot F (1999) Informed consent to serum screening for Down syndrome: Are women given adequate information? Prenat Diagn 19:1–7. Goel V, Glazier R, Holzapfel S, Pugh P, Summers A (1996) Evaluating patient’s knowledge of maternal serum screening. Prenat Diagn 16:425–430. Hallam L (1989) You’ve got a lot to answer for, Mr. Bell. A review of the use of the telephone in primary care. Fam Pract 6(1):47–75. Hornblow AR (1986) The evolution and effectiveness of telephone counseling services. Hosp Community Psychiatry 37(7):731–733. Horowitz M, Wilner N, Alvarez W (1979) Impact of Events scale: A measure of subjective stress. Psychosom Med 41(3):209–218. Koren G, Graham K, Feigenbaum A, Einarson T (1993) Evaluation and counseling of teratogenic risk: The Motherisk approach. J Clin Pharmacol 33:405–411. Kornman LH, Wortelboer MJM, Beekhuis JR, Morssink LP, Mantingh A (1997) Women’s opinions and the implications of first- versus second-trimester screening for fetal Down’s syndrome. Prenat Diagn 17(11):1011–1018. Li KC, Lai SS, Lam ST (1999) Adequacy and pitfalls of G6PD deficiency counseling in Hong Kong. Southeast Asian J Trop Med Public Health 30(Suppl 2): 79–83. Lynch DL, Tamburrino MB, Nagel R (1997) Telephone counseling for patients with minor depression: Preliminary findings in a family practice setting. J Fam Pract 44(3):293–298. Marteau TM (1995) Towards informed decisions about prenatal testing: A review. Prenat Diagn 15:1215–1226. McBride CM, Rimer BK (1999) Using the telephone to improve health behavior and health service delivery. Patient Educ Counsel 37:3–18.
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Ormond KE, Haun J, Cook L, Duquette D, Ludowese C, Matthews AL (2000) Recommendations for telephone counseling. J Genet Counsel 9(1):63–71. Quagliarini D, Betti S, Brambati B, Nicolini U (1998) Coping with serum screening for Down syndrome when the result is given as a numeric value. Prenat Diagn 18:816–821. Rayburn W, Wilson G, Schreck J, Louwsma G, Hamman J (1982) Prenatal counseling: A state-wide telephone service. Obstet Gynecol 60(2):243–246. Shiloh S, Berkenstadt M, Meiran N, Bat-Miriam-Katznelson M, Goldman B (1997) Mediating effects of perceived personal control in coping with a health threat: The case of genetic counseling. J Appl Soc Psychol 27:1146–1174. Soet JE, Basch CE (1997) The telephone as a communication medium for health education. Health Educ Behav 24(6):759–772. Statistics Canada Web site: http://www.statcan.ca/english/Pgdb/People/Education/educ44a.htm Tsu RC, Burm ML, Jennifer AG, Sells CW (2002) Telephone vs. face-to-face notification of HIV results in high-risk youth. J Adolesc Health 30:154–160. Watson D, Clark LA, Tellegen A (1988) Development and validation of brief measures of positive and negative affect: The PANAS scales. J Pers Soc Psychol 54(6):1063–1070. Wilson K, Williams A (2000) Visualism in community nursing: Implications for telephone work with service users. Qual Health Res 10(4):507–520.
