- Email: [email protected]
Impact of extreme prematurity on families of adolescent children
Impact of extreme prematurity on families of adolescent children Saroj Saigal, MD, FRCP, Elizabeth Burrows, MBA, Barbara L. Stoskopf, RN, MHSc, Peter L. Rosenbaum, MD, FRCP, and David Streiner, PhD Objectives: To compare the impact of burden of illness on families of teenaged children who were extremely low birth weight (ELBW) with that of members of a term control group (C) and to determine whether the attitudes toward active treatment of very immature infants differ between the 2 cohorts. Design: In a cross-sectional survey, parents of 145 (86%) of 169 members of an ELBW cohort and 123 (85%) of 145 members of a control cohort completed a 23-item self-completed questionnaire encompassing occupational, marital, and family-related issues and attitudes toward treatment of infants of borderline viability. Results: Both positive (P = .0003) and negative (P < .005) effects on marriage were higher in parents of the ELBW group; although more parents in the ELBW group felt that their child had brought their families closer together (P = .0001), their child’s health had adversely affected their emotional health (P = .02) and that of other children in the family (P = .003). Despite this result, a significant proportion of parents from both cohorts supported saving all infants (ELBW 68%; C 58%) and favored the role of parents in decision making (ELBW 98%; C 97%). Conclusions: In the long term, it appears that parents of ELBW children have adjusted fairly well to their work and family life. Although some negative effects were identified, there was still considerable support for active treatment of infants of borderline viability. (J Pediatr 2000;137:701-6)
The extensive literature on the outcome of very low birth weight infants suggests significantly higher rates of neurodevelopmental morbidity, ill
health, and recurrent hospitalizations in infancy compared with children born at term.1,2 With longer-term follow-up to school age, other problems
From the Department of Pediatrics, McMaster University, Hamilton, Ontario, Canada; and Department of Psychology, University of Toronto, Toronto, Ontario, Canada.
Supported by grant 6606-5316-301 from the National Health Research and Development Programme, Ottawa, Ontario, Canada, and in part by HS - 08385 from the Agency for Health Care Policy Research, Rockville, Maryland. Submitted for publication Dec 22, 1999; revisions received Mar 15, 2000, and May 1, 2000; accepted May 17, 2000. Reprint requests: Saroj Saigal, MD, Hamilton Health Sciences Centre, McMaster Division, Room 4G40, 1200 Main St West, Hamilton, Ontario, L8S 4J9, Canada. Copyright © 2000 by Mosby, Inc. 0022-3476/2000/$12.00 + 0 9/21/109001 doi:10.1067/mpd.2000.109001
such as behavioral difficulties, cognitive deficits, and underachievement in the classroom become apparent, contributing to an additional burden and stress on the parents.3-6 However, little information is available in the literature regarding the psychologic impact on families with the birth of VLBW infants and their subsequent adaptation. These studies suggest that families of VLBW infants have a significant negative impact that is greater when the children have disabilities,7-13 although one study reports otherwise.14 ELBW Extremely low birth weight VLBW Very low birth weight
In this study we compared the impact of burden of illness in the teenaged years on the families of infants who were extremely low birth weight with those whose children were born at term to determine whether the impact is long-lasting. We also investigated whether there was a significantly greater negative impact on parents of children with impairments, and last, we compared the attitudes of parents of children in the premature and control groups towards active treatment of infants of borderline viability. We hypothesized that parents of ELBW children would continue to experience a significant negative impact that would be more pronounced in families of children with disabilities, and that there would be no differences in the attitudes towards active treatment of infants of borderline viability between parents of children in the ELBW and control groups. 701
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Table I. Sociodemographic data: parents of teens in ELBW and control groups
children. The questionnaire in this form was not validated before its use.
Parents
Respondents, mothers (%) Maternal age, mean (SD) Born in Canada (%) Two-parent families (%) Have children with impairments (%) Socioeconomic status16 (%) I, II III IV, V Maternal education (%) Secondary school or less Some postsecondary education Completed university Religious participation (%) Almost every week/>just holidays Only on holidays/almost never or never
METHODS Parent Respondents Parents of ELBW survivors born between 1977 and 1982 in a geographically defined region in central west Ontario and monitored longitudinally from birth were approached to participate in the study.1 Parents of children born at term were recruited when the children were 8 years old from a random list of children obtained through the Directors of the Hamilton Public and Roman Catholic Separate School Boards and matched for age, sex, and social class to the index children.3 At the time of the study both cohorts of children ranged in age between 12 and 16 years.
Measures The parent respondents completed the following self-administered questionnaires. SOCIODEMOGRAPHIC INFORMATION ON RESPONDENTS. This questionnaire inquired about age, marital status, composition of the household (single 702
ELBW group (n = 145)
Control group (n = 123)
82 41 (6) 76 89 26
94 43 (5) 64 85 2
39 30 31
44 31 25
59 24 17
47 33 20
44 56
50 50
parent, 2-parent families), maternal education, employment status, and participation in religious activities.15 Social class was classified according to the Hollingshead 4-factor social class index.16 Information on the neurodevelopmental status of the children of the respondents was obtained from the ongoing longitudinal follow-up studies of these children.4 IMPACT ON THE FAMILY. This 17-item questionnaire encompassing work-, marital-, and family-related issues was derived from items from the following sources: 2 questions from The Child General Health Survey, 199117; 4 questions from the National Centre for Health Statistics, National Health Interview Survey, 198118; 8 questions from the Survey of Disabled Children, Great Britain, 198919; and 3 questions from the study on the impact of VLBW on the family by Lee et al.14 Multiple sources were used because no single questionnaire was available that addressed the family impact in both disabled and nondisabled teenaged
ATTITUDES TOWARD SAVING VLBW INFANTS. This 6-item self-completed questionnaire was developed and validated by Lee et al14 with a Likert scale format with a 5-point scale ranging from “strongly agree” to “strongly disagree.” It assessed perceptions of the risks of handicap in VLBW infants, attitudes towards active treatment for VLBW infants, and designation of who should be involved in decision making. The questionnaires were completed by the parents in a private room at the Hamilton Health Sciences Corporation. A number coding system was used to maintain anonymity of respondents. The questionnaires for this study were administered to the respondents after they completed direct interviews for a study on preferences of parents for neonatal outcomes.20
Consent and Ethics Approval Written informed consent was obtained from all respondents who agreed to participate in the study. The study was approved by the Ethics Review Board of the Hamilton Health Sciences Corporation, Hamilton, Ontario.
Statistical Analysis Statistical analysis included t tests for continuous variables and χ2 test for categoric ones; a P value of <.05 was considered to be significant.
RESULTS Demographic Characteristics of Respondents Parents of 145 (86%) of 169 ELBW survivors and 123 (85%) of 145 parents of term children participated in the study (Table I). Most of the respondents were mothers. No differences were noted between the groups in socioeconomic status, maternal education, proportion of 2-parent families, or participation of parents in religious activities. The proportion of parents
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THE JOURNAL OF PEDIATRICS VOLUME 137, NUMBER 5 who had children with neurosensory impairments was significantly higher among the ELBW group (ELBW 26%; control 2%, P < .0001) and included 1 or more of the following conditions: cerebral palsy (n = 19), hydrocephalus (n = 6), significant cognitive impairments (n = 14), autism (n = 5), unilateral blindness (n = 5), bilateral blindness (n = 9), and sensorineural deafness (n = 2).4 Of parents of ELBW children with neurosensory impairments, 75% completed the impact questionnaire. No differences were noted in sociodemographic variables between respondents and nonrespondents. The mean birth weight of the ELBW infants was 837 g (SD 124) with a mean gestational age of 27 weeks (SD 2.0). Infants in the control group were born at term, and the mean birth weight was 3391 g (SD 486).
Impact on the Family In the 6 months before the survey, no differences were noted in the proportion of parents of the ELBW and control groups who had emotional suffering or worry because of their child’s physical or emotional health; nor were there any differences in the proportion who felt they had limited time available for their personal needs (Table II). Although very few parents of both cohorts felt that their own physical health was currently affected, a significantly higher proportion of ELBW parents felt that their child’s health had an impact on their own emotional health (P < .05), and that there were other negative effects on the family (P < .005). There were mixed findings in terms of the effects of the ELBW children on marriage. A significantly higher proportion of parents of ELBW children reported that their child’s health status had caused stresses and strains (P < .005), had brought the partners closer together (P < .001), and was a major factor in separation and divorce. The impact also negatively affected other siblings in the families of ELBW children (P < .005), primarily because of
Table II. Impact on family caused by child’s health problems
Parents ELBW group (n = 145) (%) Emotional & physical health of parent Emotional suffering: past 6 months: Due to child’s physical health None at all 62.1 A little bit/some 32.4 Quite a bit/a lot 5.5 Due to child’s emotional health None at all 34.7 A little bit/some 48.6 Quite a bit/a lot 16.7 Limited time for personal needs due to child’s physical health None at all 77.9 A little bit/some 20.0 Quite a bit/a lot 2.1 Limited time for personal needs due to child’s emotional health None at all 53.5 A little bit/some 38.9 Quite a bit/a lot 7.6 Own emotional and physical health: now Own physical health affected 7.7 Own emotional health affected 21.0 Other negative effects on family 15.4 Marriage, children, and friendship: ever Effects on marriage: No effect 55.2 Brought closer together 24.6 Caused stresses/strains 14.0 Major factor in separation/divorce 4.2 Other 2.1 Effects on other children: Negative effects 21.6 Making friends/keeping friends 2.4 Inviting friends home/visiting friends 4.0 Less parental attention 14.4 Subsequent children: No subsequent children 58.6 Decision influenced by study child 58.3 Effects on family, friends, and self: Brought family closer together 38.7 Friends and relatives are more understanding 53.5 Difficulties in making friends 6.2 Improved feelings about self 56.3 Job-related effects Unable to take job, ever 7.6 Had to quit/change jobs, ever 4.8 Ability to work, in past 7.6 Ability to work, now 4.8 Finances: ever Major financial problems 4.8 Residence: ever Moved because of child’s health 0.7 To be near special services 0.7 Needed different type of house 0
Control group (n = 123) (%)
65.9 31.7 2.4 43.1 39.8 17.1 80.5 17.9 1.6 63.4 28.5 8.1 4.1 9.8* 3.3†
85.4 7.3‡§ 5.7† 0 1.6 7.1† 0 1.8 3.6* 39.3* 6.3‡ 13.2‡ 25.2‡ 2.5 42.5* 0.8* 0.8 4.9 0.8 1.6 0 0 0
*P < .05. †P < .005. ‡P < .001. §Versus no effect. Versus no effect.
703
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Figure. Response to question about “who should make the ultimate decision on whether an extremely premature infant should be treated or allowed to die.”
less parental attention (14% vs 4%, P < .05). Parents of the ELBW cohort were more likely not to have subsequent children (P < .05), and this decision was directly influenced by the health of the child in the study (P < .001). Overall, a higher proportion of families of ELBW parents felt that the experience of the birth of their child had brought the families closer together (P < .001), and that relatives and friends were more understanding and helpful (P < .001). The experience had also improved their feelings about themselves (P < .05). A significantly higher proportion of ELBW parents reported that they were unable to take a job because of their child’s health (P < .05), but no significant differences were reported between the groups in experiencing any major financial difficulties or problems with housing.
Impact on Families of ELBW Children With and Without Impairments Further comparisons of the impact among families after exclusion of children with impairments revealed that the differences between the ELBW and control groups were now dimin704
ished in most variables listed in Table II. Negative effects on the other children in the family and parents’ own physical health were no longer significantly different. The experience of having an ELBW child, even though the child was nonimpaired, remained as a significant impact on other aspects of the family: marriage relationships (positive effects 21% vs 7%, P < .001; negative effects 14% vs 6%, P < .05), positive interactions with family (37% vs 13%, P < .001) and friends (51% vs 25%, P < .001), and the decision not to have further children (54% vs 39%, P < .05). Last, a subanalysis was conducted within families of ELBW children with (n = 35) and without (n = 110) impairments to determine whether the magnitude of the impact was greater among parents of ELBW children with disabilities. Data are presented only on those variables for which the differences were statistically significant. A higher proportion of parents of ELBW children with disabilities reported that their own emotional health was affected (34% vs 17%, P < .05), that they had limited time for personal needs because of their child’s emotional health (34% vs 16%, P < .05), and that their
child’s impairment had negatively affected other children in the family (45% vs 14%, P < .001), primarily because of less parental attention to the siblings (24% vs 6%, P < .05). Parents of children with disabilities had considerably more difficulties in their ability to take on a job (17% vs 5%, P < .05). Yet more parents of disabled children than nondisabled children reported that the experience had improved their feelings about themselves (74% vs 51%, P < .05).
Attitudes of Parents Toward Active Treatment of Infants of Borderline Viability More parents of infants in the ELBW group than in the control group believed that premature infants have an increased chance of being disabled (66% vs 39%, P < .001). The proportion of parents who believed that an attempt should be made to save all infants, even if the infant will most likely be severely disabled, was similar for both groups (ELBW 68%, control 58%). Among those who agreed with the previous statement, the reasons cited were a willingness to take a chance, no matter how small, that the
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THE JOURNAL OF PEDIATRICS VOLUME 137, NUMBER 5 infant will be normal (ELBW 92%; control 93%), willingness to accept any outcome because this was their child (ELBW 94%; control 93%), belief in the sanctity of life (ELBW 93%; control 99%), and religious reasons (ELBW 77%; control 80%). Of those who disagreed with the statement that doctors should always try to save all infants regardless of outcome, most parents felt that the decision should be made on an individual basis (ELBW 100%; control 96%) and not on the basis of a standard policy (ELBW 80%; control 84%). Parental responses regarding who should make the final decision on whether to actively treat an immature infant were also similar for both groups (Figure). Overwhelmingly, both cohorts of parents believed that the final decision should be made by parents (ELBW 98%; control 97%). A significant proportion supported the role of doctors (ELBW 66%; control 77%), but not as many were in favor of nurses making the decision (ELBW 31%, control 31%). There was much less support (< 36%) by both groups for hospital ethics committees, law courts, and other professional bodies in decision making. Both cohorts felt strongly that costs should not enter into the decision regarding life-saving measures (ELBW 91%, control 86%).
DISCUSSION Our study adds to the growing literature on the impact of prematurity on the family and on the attitudes of parents toward active treatment of infants at high risk. This large-scale controlled study includes the impact on the family at adolescence. Furthermore the parents of the ELBW cohort were a population-based sample with a high response rate, thus eliminating the sampling bias inherent in some studies. It is possible that because several years have elapsed, parents may have underestimated the significant negative impact of the earli-
er years. Nevertheless, they have also had a longer time to reflect and may be making a more informed judgement of the overall impact on their family. The results of this longitudinal study to adolescence indicate that in the long term, parents of ELBW children appear to have adjusted fairly well to most aspects of their work and family life. However, this is not to minimize the difficulties experienced earlier by parents of ELBW children, many of whom still feel that it has taken a significant toll on their emotional health. The negative impact on the family diminished when children with impairments were excluded. Yet it was interesting that a higher proportion of parents of ELBW children with impairments than parents of nonimpaired children reported that the experience had improved their feelings about themselves. A similar theme of parenting satisfaction and positive family adaptation has been reported in several other studies,7,10,12,14 attesting to the remarkable resilience of parents. In this homogeneous, relatively advantaged socioeconomic population, no significant financial burdens were reported by most of the ELBW cohort as a result of their child’s health. No doubt the Canadian socialized health and welfare system may have decreased some of the financial anxieties. The impact could be significantly more devastating to socioeconomically disadvantaged or single-parent families.7,9,10,12,14 It is also likely that the impact may be the same or greater for the families of the current survivors of neonatal intensive care, because morbidity rates have not decreased. Unlike the findings by Lee et al,14 parents of ELBW children in this study appear to be better informed about the possibility of impairments in preterm infants and were somewhat less enthusiastic about saving all infants. Nevertheless, a significant proportion of parents from both cohorts supported life-sustaining medical technology. In contrast, and clearly of great
concern, are the perspectives of some parents who have written moving accounts of their personal tragedies resulting from overtreatment of their newborns.21,22 Our study also suggests that both cohorts of parents are overwhelmingly supportive of their personal involvement in decisions regarding life-sustaining treatment. These findings are in accord with those reported by Lee et al14 and are shared by parent advocates.23 We are struck with the remarkable similarities in the attitudes of parents of premature and term infants in their views toward management decisions in the perinatal period. Others have explored parental perceptions 4 years after the child’s discharge from the neonatal intensive care unit, and the key issues that emerged were the lack of involvement in ethical decision making, insufficient knowledge to participate in such processes, and a clear mandate for change.24 Silverman,25 a pioneering neonatologist, has long argued for less aggressive treatment and for more discussions with families regarding their wishes. Several other investigators have also written passionately on this subject.26-28 Despite technologic advances, there continues to be scientific uncertainty regarding the outcomes of critically ill newborns. Therefore in making evidence-based decisions about imperiled newborns, the benefits and burdens of treatment alternatives and the values and preferences of the family must be considered.29 The literature suggests conflicting perspectives27,28 and marked heterogeneity in the preferences of patients, parents, and health care providers.30 Given this, we concur with the recommendation by Fost31 that the best approach for assisting parents in the decision regarding instituting neonatal intensive care is for a candid dialogue between parents and health professionals. In these deliberations, which should occur from the earliest possible moment, all relevant facts should be provided, and the interests of the infant and the family 705
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should be taken into consideration at an individual level. Some believe that in this process “the stake of parents who must provide lifelong care is much larger than that of the physicians whose involvement is transitory” (W. Silverman, personal communication). We are most grateful to the parents of teenagers in the premature and control groups for taking the time and effort in completing the questionnaires. Dr. Shoo Lee and Dr. M. Cox were generous in sharing the questionnaires and providing advice. We thank Lorraine Hoult for her participation in the research activities and Jennifer Isaacs for secretarial assistance. We acknowledge the support of the Department of Pediatrics, McMaster University.
REFERENCES 1. Saigal S, Rosenbaum P, Hattersley B, Milner R. Decreased disability rate among 3-year-old survivors weighing 501 to 1000 grams at birth and born to residents of a geographically defined region from 1981 to 1984 compared with 1977 to 1980. J Pediatr 1989;114: 839-46. 2. Lorenz JM, Wooliever DE, Jetton JR, Paneth N. A quantitative review of mortality and developmental disability in extremely premature newborns. Arch Pediatr Adolesc Med 1998;152:425-35. 3. Saigal S, Szatmari P, Rosenbaum P, Campbell D, King S. Cognitive abilities and school performance of extremely low birth weight children and matched term control children at age 8 years: a regional study. J Pediatr 1991; 118:751-60. 4. Saigal S, Hoult LA, Streiner DL, Stoskopf BL, Rosenbaum PL. School difficulties at adolescence in a regional cohort of children who were extremely low birthweight. Pediatrics 2000;105: 325-31. 5. Szatmari P, Saigal S, Rosenbaum P, Campbell D. Psychopathology and adaptive functioning among extremely low birthweight children at eight years of age. Dev Psychopathol 1993;5:345-57. 6. Hack M, Taylor HG, Klein N, Eiben R, Schatschneider C, Mercuri-Minich N. School-age outcomes in children
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with birthweights under 750g. N Engl J Med 1994;331:753-9. Cronin C, Shapiro C, Casiro O, Cheang M. The impact of very low birth weight infants on the family is long lasting. Arch Pediatr Adolesc Med 1995;149:151-8. Singer L, Davillier M, Bruening P, Hawkins S, Yamashita T. Social support, psychological distress, and parenting strains in mothers of VLBW infants. Fam Relations 1996;45:343-50. Rivers A, Caron B, Hack M. Experience of families with very low birthweight children with neurologic sequelae. Clin Pediatr 1987;26:223-30. McCormick MC, Stemmler MM, Bernbaum JC, Farran AC. The very low birth weight transport goes home: impact on the family. J Dev Behav Pediatr 1986;7:217-23. Trause MA, Kramer LI. The effects of premature birth on parents and their relationship. Dev Med Child Neurol 1983;25:459-65. Singer L, Salvator A, Guo S, Collin M, Lilien L, Baley J. Maternal psychological distress and parenting stress after the birth of a very low birth weight infant. JAMA 1999;281:799-805. Warfield ME, Krauss MW, HauserCram P, Upshur CC, Shonkoff JP. Adaptation during early childhood among mothers of children with disabilities. J Dev Behav Pediatr 1999;20:9-16. Lee SK, Penner PL, Cox M. Impact of very low birth weight infants on the family and its relationship to parental attitudes. Pediatrics 1991;88:105-9. Boyle MH, Offord DR, Hoffmann HG, Catlin GP, Byles JA, Cadman DT, et al. Ontario child health study 1: methodology. Arch Gen Psychiatry 1987;44:826-31. Hollingshead AB. Four Factor Index of Social Status, unpublished working paper, 1975, Department of Sociology, Yale University, P.O. Box 1965, New Haven (CT). Landgraf JM, Ware JE, Schor E, Rossi-Roh K. Child General Health Survey. Child Health Project/The Health Institute, New England Medical Center Hospital, 1991. National Centre for Health Statistics. National Health Interview Survey, 1981.
19. Survey of Disabled Children. Office of Population Censuses and Surveys, Great Britain, 1989. 20. Saigal S, Rosenbaum PL, Feeny D, Burrows E, Furlong F, Stoskopf BL, et al. Parental perspectives of the health status and health-related quality of life of teenaged children who were extremely low birthweight and term controls. Pediatrics 2000;105:569-74. 21. Stinson R, Stinson P. The long dying of baby Andrew. Boston (MA): Little, Brown & Co; 1992. 22. Montalvo N, Vila BP. Parent’s grand rounds speech on neonatal intensive care unit experience. J Perinatol 1999; 19:525-7. 23. Harrison H. The principles for familycentered neonatal care. Pediatrics 1993;92:643-50. 24. Pinch WJE, Spielman ML. Ethics in the neonatal intensive care unit: parental perceptions at four years post discharge. ANS Adv Nurs Sci 1996; 19:72-85. 25. Silverman WA. Overtreatment of neonates? A personal retrospective. Pediatrics 1992;90:971-6. 26. Stahlman MT. Ethical issues in the nursery: priorities versus limits. J Pediatr 1990;116:167-70. 27. Lantos JD, Tyson JE, Allen A, Frader J, Hack M, Korones S, et al. Withholding and withdrawing life sustaining treatment in neonatal intensive care: issues for the 1990s. Arch Dis Child 1994;71:F218-23. 28. Pinkerton JV, Finnerty JJ, Lombardo PA, Rorty MV, Chapple H, Boyle RJ. Parental rights at the birth of a near-viable infant: conflicting perspectives. Am J Obstet Gynecol 1997;177:283-90. 29. Tyson J. Evidence-based ethics and the care of premature infants. In: Behrman RE, editor. Low birth weight. The future of children. Los Altos (CA): Center for the Future of Children; 1995;5:197-213. 30. Saigal S, Stoskopf BL, Feeny D, Furlong W, Burrows E, Rosenbaum PL, et al. Differences in preferences for neonatal outcomes among health care professionals, parents, and adolescents. JAMA 1999;281:1991-7. 31. Fost N. Decisions regarding treatment of seriously ill newborns. JAMA 1999; 281:2041-3.
The extensive literature on the outcome of very low birth weight infants suggests significantly higher rates of neurodevelopmental morbidity, ill
health, and recurrent hospitalizations in infancy compared with children born at term.1,2 With longer-term follow-up to school age, other problems
From the Department of Pediatrics, McMaster University, Hamilton, Ontario, Canada; and Department of Psychology, University of Toronto, Toronto, Ontario, Canada.
Supported by grant 6606-5316-301 from the National Health Research and Development Programme, Ottawa, Ontario, Canada, and in part by HS - 08385 from the Agency for Health Care Policy Research, Rockville, Maryland. Submitted for publication Dec 22, 1999; revisions received Mar 15, 2000, and May 1, 2000; accepted May 17, 2000. Reprint requests: Saroj Saigal, MD, Hamilton Health Sciences Centre, McMaster Division, Room 4G40, 1200 Main St West, Hamilton, Ontario, L8S 4J9, Canada. Copyright © 2000 by Mosby, Inc. 0022-3476/2000/$12.00 + 0 9/21/109001 doi:10.1067/mpd.2000.109001
such as behavioral difficulties, cognitive deficits, and underachievement in the classroom become apparent, contributing to an additional burden and stress on the parents.3-6 However, little information is available in the literature regarding the psychologic impact on families with the birth of VLBW infants and their subsequent adaptation. These studies suggest that families of VLBW infants have a significant negative impact that is greater when the children have disabilities,7-13 although one study reports otherwise.14 ELBW Extremely low birth weight VLBW Very low birth weight
In this study we compared the impact of burden of illness in the teenaged years on the families of infants who were extremely low birth weight with those whose children were born at term to determine whether the impact is long-lasting. We also investigated whether there was a significantly greater negative impact on parents of children with impairments, and last, we compared the attitudes of parents of children in the premature and control groups towards active treatment of infants of borderline viability. We hypothesized that parents of ELBW children would continue to experience a significant negative impact that would be more pronounced in families of children with disabilities, and that there would be no differences in the attitudes towards active treatment of infants of borderline viability between parents of children in the ELBW and control groups. 701
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THE JOURNAL OF PEDIATRICS NOVEMBER 2000
Table I. Sociodemographic data: parents of teens in ELBW and control groups
children. The questionnaire in this form was not validated before its use.
Parents
Respondents, mothers (%) Maternal age, mean (SD) Born in Canada (%) Two-parent families (%) Have children with impairments (%) Socioeconomic status16 (%) I, II III IV, V Maternal education (%) Secondary school or less Some postsecondary education Completed university Religious participation (%) Almost every week/>just holidays Only on holidays/almost never or never
METHODS Parent Respondents Parents of ELBW survivors born between 1977 and 1982 in a geographically defined region in central west Ontario and monitored longitudinally from birth were approached to participate in the study.1 Parents of children born at term were recruited when the children were 8 years old from a random list of children obtained through the Directors of the Hamilton Public and Roman Catholic Separate School Boards and matched for age, sex, and social class to the index children.3 At the time of the study both cohorts of children ranged in age between 12 and 16 years.
Measures The parent respondents completed the following self-administered questionnaires. SOCIODEMOGRAPHIC INFORMATION ON RESPONDENTS. This questionnaire inquired about age, marital status, composition of the household (single 702
ELBW group (n = 145)
Control group (n = 123)
82 41 (6) 76 89 26
94 43 (5) 64 85 2
39 30 31
44 31 25
59 24 17
47 33 20
44 56
50 50
parent, 2-parent families), maternal education, employment status, and participation in religious activities.15 Social class was classified according to the Hollingshead 4-factor social class index.16 Information on the neurodevelopmental status of the children of the respondents was obtained from the ongoing longitudinal follow-up studies of these children.4 IMPACT ON THE FAMILY. This 17-item questionnaire encompassing work-, marital-, and family-related issues was derived from items from the following sources: 2 questions from The Child General Health Survey, 199117; 4 questions from the National Centre for Health Statistics, National Health Interview Survey, 198118; 8 questions from the Survey of Disabled Children, Great Britain, 198919; and 3 questions from the study on the impact of VLBW on the family by Lee et al.14 Multiple sources were used because no single questionnaire was available that addressed the family impact in both disabled and nondisabled teenaged
ATTITUDES TOWARD SAVING VLBW INFANTS. This 6-item self-completed questionnaire was developed and validated by Lee et al14 with a Likert scale format with a 5-point scale ranging from “strongly agree” to “strongly disagree.” It assessed perceptions of the risks of handicap in VLBW infants, attitudes towards active treatment for VLBW infants, and designation of who should be involved in decision making. The questionnaires were completed by the parents in a private room at the Hamilton Health Sciences Corporation. A number coding system was used to maintain anonymity of respondents. The questionnaires for this study were administered to the respondents after they completed direct interviews for a study on preferences of parents for neonatal outcomes.20
Consent and Ethics Approval Written informed consent was obtained from all respondents who agreed to participate in the study. The study was approved by the Ethics Review Board of the Hamilton Health Sciences Corporation, Hamilton, Ontario.
Statistical Analysis Statistical analysis included t tests for continuous variables and χ2 test for categoric ones; a P value of <.05 was considered to be significant.
RESULTS Demographic Characteristics of Respondents Parents of 145 (86%) of 169 ELBW survivors and 123 (85%) of 145 parents of term children participated in the study (Table I). Most of the respondents were mothers. No differences were noted between the groups in socioeconomic status, maternal education, proportion of 2-parent families, or participation of parents in religious activities. The proportion of parents
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THE JOURNAL OF PEDIATRICS VOLUME 137, NUMBER 5 who had children with neurosensory impairments was significantly higher among the ELBW group (ELBW 26%; control 2%, P < .0001) and included 1 or more of the following conditions: cerebral palsy (n = 19), hydrocephalus (n = 6), significant cognitive impairments (n = 14), autism (n = 5), unilateral blindness (n = 5), bilateral blindness (n = 9), and sensorineural deafness (n = 2).4 Of parents of ELBW children with neurosensory impairments, 75% completed the impact questionnaire. No differences were noted in sociodemographic variables between respondents and nonrespondents. The mean birth weight of the ELBW infants was 837 g (SD 124) with a mean gestational age of 27 weeks (SD 2.0). Infants in the control group were born at term, and the mean birth weight was 3391 g (SD 486).
Impact on the Family In the 6 months before the survey, no differences were noted in the proportion of parents of the ELBW and control groups who had emotional suffering or worry because of their child’s physical or emotional health; nor were there any differences in the proportion who felt they had limited time available for their personal needs (Table II). Although very few parents of both cohorts felt that their own physical health was currently affected, a significantly higher proportion of ELBW parents felt that their child’s health had an impact on their own emotional health (P < .05), and that there were other negative effects on the family (P < .005). There were mixed findings in terms of the effects of the ELBW children on marriage. A significantly higher proportion of parents of ELBW children reported that their child’s health status had caused stresses and strains (P < .005), had brought the partners closer together (P < .001), and was a major factor in separation and divorce. The impact also negatively affected other siblings in the families of ELBW children (P < .005), primarily because of
Table II. Impact on family caused by child’s health problems
Parents ELBW group (n = 145) (%) Emotional & physical health of parent Emotional suffering: past 6 months: Due to child’s physical health None at all 62.1 A little bit/some 32.4 Quite a bit/a lot 5.5 Due to child’s emotional health None at all 34.7 A little bit/some 48.6 Quite a bit/a lot 16.7 Limited time for personal needs due to child’s physical health None at all 77.9 A little bit/some 20.0 Quite a bit/a lot 2.1 Limited time for personal needs due to child’s emotional health None at all 53.5 A little bit/some 38.9 Quite a bit/a lot 7.6 Own emotional and physical health: now Own physical health affected 7.7 Own emotional health affected 21.0 Other negative effects on family 15.4 Marriage, children, and friendship: ever Effects on marriage: No effect 55.2 Brought closer together 24.6 Caused stresses/strains 14.0 Major factor in separation/divorce 4.2 Other 2.1 Effects on other children: Negative effects 21.6 Making friends/keeping friends 2.4 Inviting friends home/visiting friends 4.0 Less parental attention 14.4 Subsequent children: No subsequent children 58.6 Decision influenced by study child 58.3 Effects on family, friends, and self: Brought family closer together 38.7 Friends and relatives are more understanding 53.5 Difficulties in making friends 6.2 Improved feelings about self 56.3 Job-related effects Unable to take job, ever 7.6 Had to quit/change jobs, ever 4.8 Ability to work, in past 7.6 Ability to work, now 4.8 Finances: ever Major financial problems 4.8 Residence: ever Moved because of child’s health 0.7 To be near special services 0.7 Needed different type of house 0
Control group (n = 123) (%)
65.9 31.7 2.4 43.1 39.8 17.1 80.5 17.9 1.6 63.4 28.5 8.1 4.1 9.8* 3.3†
85.4 7.3‡§ 5.7† 0 1.6 7.1† 0 1.8 3.6* 39.3* 6.3‡ 13.2‡ 25.2‡ 2.5 42.5* 0.8* 0.8 4.9 0.8 1.6 0 0 0
*P < .05. †P < .005. ‡P < .001. §Versus no effect. Versus no effect.
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Figure. Response to question about “who should make the ultimate decision on whether an extremely premature infant should be treated or allowed to die.”
less parental attention (14% vs 4%, P < .05). Parents of the ELBW cohort were more likely not to have subsequent children (P < .05), and this decision was directly influenced by the health of the child in the study (P < .001). Overall, a higher proportion of families of ELBW parents felt that the experience of the birth of their child had brought the families closer together (P < .001), and that relatives and friends were more understanding and helpful (P < .001). The experience had also improved their feelings about themselves (P < .05). A significantly higher proportion of ELBW parents reported that they were unable to take a job because of their child’s health (P < .05), but no significant differences were reported between the groups in experiencing any major financial difficulties or problems with housing.
Impact on Families of ELBW Children With and Without Impairments Further comparisons of the impact among families after exclusion of children with impairments revealed that the differences between the ELBW and control groups were now dimin704
ished in most variables listed in Table II. Negative effects on the other children in the family and parents’ own physical health were no longer significantly different. The experience of having an ELBW child, even though the child was nonimpaired, remained as a significant impact on other aspects of the family: marriage relationships (positive effects 21% vs 7%, P < .001; negative effects 14% vs 6%, P < .05), positive interactions with family (37% vs 13%, P < .001) and friends (51% vs 25%, P < .001), and the decision not to have further children (54% vs 39%, P < .05). Last, a subanalysis was conducted within families of ELBW children with (n = 35) and without (n = 110) impairments to determine whether the magnitude of the impact was greater among parents of ELBW children with disabilities. Data are presented only on those variables for which the differences were statistically significant. A higher proportion of parents of ELBW children with disabilities reported that their own emotional health was affected (34% vs 17%, P < .05), that they had limited time for personal needs because of their child’s emotional health (34% vs 16%, P < .05), and that their
child’s impairment had negatively affected other children in the family (45% vs 14%, P < .001), primarily because of less parental attention to the siblings (24% vs 6%, P < .05). Parents of children with disabilities had considerably more difficulties in their ability to take on a job (17% vs 5%, P < .05). Yet more parents of disabled children than nondisabled children reported that the experience had improved their feelings about themselves (74% vs 51%, P < .05).
Attitudes of Parents Toward Active Treatment of Infants of Borderline Viability More parents of infants in the ELBW group than in the control group believed that premature infants have an increased chance of being disabled (66% vs 39%, P < .001). The proportion of parents who believed that an attempt should be made to save all infants, even if the infant will most likely be severely disabled, was similar for both groups (ELBW 68%, control 58%). Among those who agreed with the previous statement, the reasons cited were a willingness to take a chance, no matter how small, that the
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THE JOURNAL OF PEDIATRICS VOLUME 137, NUMBER 5 infant will be normal (ELBW 92%; control 93%), willingness to accept any outcome because this was their child (ELBW 94%; control 93%), belief in the sanctity of life (ELBW 93%; control 99%), and religious reasons (ELBW 77%; control 80%). Of those who disagreed with the statement that doctors should always try to save all infants regardless of outcome, most parents felt that the decision should be made on an individual basis (ELBW 100%; control 96%) and not on the basis of a standard policy (ELBW 80%; control 84%). Parental responses regarding who should make the final decision on whether to actively treat an immature infant were also similar for both groups (Figure). Overwhelmingly, both cohorts of parents believed that the final decision should be made by parents (ELBW 98%; control 97%). A significant proportion supported the role of doctors (ELBW 66%; control 77%), but not as many were in favor of nurses making the decision (ELBW 31%, control 31%). There was much less support (< 36%) by both groups for hospital ethics committees, law courts, and other professional bodies in decision making. Both cohorts felt strongly that costs should not enter into the decision regarding life-saving measures (ELBW 91%, control 86%).
DISCUSSION Our study adds to the growing literature on the impact of prematurity on the family and on the attitudes of parents toward active treatment of infants at high risk. This large-scale controlled study includes the impact on the family at adolescence. Furthermore the parents of the ELBW cohort were a population-based sample with a high response rate, thus eliminating the sampling bias inherent in some studies. It is possible that because several years have elapsed, parents may have underestimated the significant negative impact of the earli-
er years. Nevertheless, they have also had a longer time to reflect and may be making a more informed judgement of the overall impact on their family. The results of this longitudinal study to adolescence indicate that in the long term, parents of ELBW children appear to have adjusted fairly well to most aspects of their work and family life. However, this is not to minimize the difficulties experienced earlier by parents of ELBW children, many of whom still feel that it has taken a significant toll on their emotional health. The negative impact on the family diminished when children with impairments were excluded. Yet it was interesting that a higher proportion of parents of ELBW children with impairments than parents of nonimpaired children reported that the experience had improved their feelings about themselves. A similar theme of parenting satisfaction and positive family adaptation has been reported in several other studies,7,10,12,14 attesting to the remarkable resilience of parents. In this homogeneous, relatively advantaged socioeconomic population, no significant financial burdens were reported by most of the ELBW cohort as a result of their child’s health. No doubt the Canadian socialized health and welfare system may have decreased some of the financial anxieties. The impact could be significantly more devastating to socioeconomically disadvantaged or single-parent families.7,9,10,12,14 It is also likely that the impact may be the same or greater for the families of the current survivors of neonatal intensive care, because morbidity rates have not decreased. Unlike the findings by Lee et al,14 parents of ELBW children in this study appear to be better informed about the possibility of impairments in preterm infants and were somewhat less enthusiastic about saving all infants. Nevertheless, a significant proportion of parents from both cohorts supported life-sustaining medical technology. In contrast, and clearly of great
concern, are the perspectives of some parents who have written moving accounts of their personal tragedies resulting from overtreatment of their newborns.21,22 Our study also suggests that both cohorts of parents are overwhelmingly supportive of their personal involvement in decisions regarding life-sustaining treatment. These findings are in accord with those reported by Lee et al14 and are shared by parent advocates.23 We are struck with the remarkable similarities in the attitudes of parents of premature and term infants in their views toward management decisions in the perinatal period. Others have explored parental perceptions 4 years after the child’s discharge from the neonatal intensive care unit, and the key issues that emerged were the lack of involvement in ethical decision making, insufficient knowledge to participate in such processes, and a clear mandate for change.24 Silverman,25 a pioneering neonatologist, has long argued for less aggressive treatment and for more discussions with families regarding their wishes. Several other investigators have also written passionately on this subject.26-28 Despite technologic advances, there continues to be scientific uncertainty regarding the outcomes of critically ill newborns. Therefore in making evidence-based decisions about imperiled newborns, the benefits and burdens of treatment alternatives and the values and preferences of the family must be considered.29 The literature suggests conflicting perspectives27,28 and marked heterogeneity in the preferences of patients, parents, and health care providers.30 Given this, we concur with the recommendation by Fost31 that the best approach for assisting parents in the decision regarding instituting neonatal intensive care is for a candid dialogue between parents and health professionals. In these deliberations, which should occur from the earliest possible moment, all relevant facts should be provided, and the interests of the infant and the family 705
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should be taken into consideration at an individual level. Some believe that in this process “the stake of parents who must provide lifelong care is much larger than that of the physicians whose involvement is transitory” (W. Silverman, personal communication). We are most grateful to the parents of teenagers in the premature and control groups for taking the time and effort in completing the questionnaires. Dr. Shoo Lee and Dr. M. Cox were generous in sharing the questionnaires and providing advice. We thank Lorraine Hoult for her participation in the research activities and Jennifer Isaacs for secretarial assistance. We acknowledge the support of the Department of Pediatrics, McMaster University.
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19. Survey of Disabled Children. Office of Population Censuses and Surveys, Great Britain, 1989. 20. Saigal S, Rosenbaum PL, Feeny D, Burrows E, Furlong F, Stoskopf BL, et al. Parental perspectives of the health status and health-related quality of life of teenaged children who were extremely low birthweight and term controls. Pediatrics 2000;105:569-74. 21. Stinson R, Stinson P. The long dying of baby Andrew. Boston (MA): Little, Brown & Co; 1992. 22. Montalvo N, Vila BP. Parent’s grand rounds speech on neonatal intensive care unit experience. J Perinatol 1999; 19:525-7. 23. Harrison H. The principles for familycentered neonatal care. Pediatrics 1993;92:643-50. 24. Pinch WJE, Spielman ML. Ethics in the neonatal intensive care unit: parental perceptions at four years post discharge. ANS Adv Nurs Sci 1996; 19:72-85. 25. Silverman WA. Overtreatment of neonates? A personal retrospective. Pediatrics 1992;90:971-6. 26. Stahlman MT. Ethical issues in the nursery: priorities versus limits. J Pediatr 1990;116:167-70. 27. Lantos JD, Tyson JE, Allen A, Frader J, Hack M, Korones S, et al. Withholding and withdrawing life sustaining treatment in neonatal intensive care: issues for the 1990s. Arch Dis Child 1994;71:F218-23. 28. Pinkerton JV, Finnerty JJ, Lombardo PA, Rorty MV, Chapple H, Boyle RJ. Parental rights at the birth of a near-viable infant: conflicting perspectives. Am J Obstet Gynecol 1997;177:283-90. 29. Tyson J. Evidence-based ethics and the care of premature infants. In: Behrman RE, editor. Low birth weight. The future of children. Los Altos (CA): Center for the Future of Children; 1995;5:197-213. 30. Saigal S, Stoskopf BL, Feeny D, Furlong W, Burrows E, Rosenbaum PL, et al. Differences in preferences for neonatal outcomes among health care professionals, parents, and adolescents. JAMA 1999;281:1991-7. 31. Fost N. Decisions regarding treatment of seriously ill newborns. JAMA 1999; 281:2041-3.