PMCCPMCCPMCC

Search tips
Search criteria 

Advanced

 
Logo of diacareAmerican Diabetes AssociationSubscribeSearchDiabetes Care Journal
 
Diabetes Care. 2009 December; 32(12): 2181–2183.
Published online 2009 September 14. doi:  10.2337/dc09-0423
PMCID: PMC2782973

From Genetic Risk Awareness to Overt Type 1 Diabetes

Parental stress in a placebo-controlled prevention trial
Eszter Goldstein, MD,1,2 Robert Hermann, MD, PHD,1,3 Timo J. Renfors, PHD,4,5 Kirsti M. Näntö-Salonen, MD, PHD,1 Tapio Korhonen, PHD,6 Maarit Kärkkäinen, MHSCI,1,7 Riitta K. Veijola, MD, PHD,8 Mikael Knip, MD, PHD,9,10 Tuula T. Simell, PHD,1 and Olli G. Simell, MD, PHD1

Abstract

OBJECTIVE

To evaluate the psychological burden of parents facing increasing risk of type 1 diabetes in their children.

RESEARCH DESIGN AND METHODS

In the population-based Type 1 Diabetes Prediction and Prevention (DIPP) Study, newborn infants with HLA-DQB1–conferred diabetes risk were enrolled in sequential analyses of diabetes-associated autoantibodies. Those persistently positive for at least two autoantibodies were recruited to a randomized double-blinded intervention trial. The experience of stress in parents of 664 children was measured using Parenting Stress Index self-report inventory.

RESULTS

While diagnosis of diabetes increased parental stress, the appearance of autoantibodies or participation in the intervention trial did not. Mothers had higher stress levels than fathers. Single parenthood and chronically ill family members increased parental stress.

CONCLUSIONS

Parental stress was not increased by notification of autoantibody positivity or by participation in an intervention trial. Other demanding family conditions contributed to the experience of stress.

Natural history and prevention studies screening genetic risk for type 1 diabetes have raised concerns of the burden of risk awareness in asymptomatic individuals, most of whom will never develop the disease (1,2).

RESEARCH DESIGN AND METHODS

Study subjects

The population-based Type 1 Diabetes Prediction and Prevention (DIPP) Study screened neonates for HLA-DQB1–conferred diabetes risk, enrolling children at risk in sequential monitoring for diabetes-associated autoantibodies. Children permanently positive for multiple autoantibodies were invited to a randomized double-blinded prevention trial comparing intranasal insulin with placebo (3).

Parents of 1,125 participants received a self-administered questionnaire (see below). There were 1,204 questionnaires (59%) returned by parents of 664 children. A total of 457 children showed genetic predisposition only, whereas 188 had diabetes-associated autoantibodies and 19 had progressed to diabetes. The time from notification of autoantibody positivity ranged from 0.5 to 6.7 years (mean 2.9).

A total of 35 parents had a child diagnosed with diabetes, and 326 had a child with diabetes-associated autoantibodies only. There were 18 parents of a child with diabetes (51.4%) and 84 parents of an autoantibody-positive child (25.8%) who enrolled their child in the prevention trial. There were 69 parents who had a child eligible for the trial but had chosen not to participate, whereas the children of 173 parents had tested only transiently positive for one autoantibody species. A total of 843 control parents, whose child had not developed autoantibodies, were matched with the parents of children with diabetes (n = 197) or autoantibodies (n = 646) for parental age, child's age, and study site. Age, employment, marital status, place of living, and chronic illness in the family were recorded.

Measurement of parenting stress

Eleven questions focusing on parenting stress were selected from the 34-item Swedish version (4) of the Parenting Stress Index self-report inventory (5) and modified to this scale with a four-factor construction. An index describing “parental stress” was calculated from the mean of the scores. Four additional factors (relationship with spouse, sense of competence of parenthood, social life, and privacy) were assessed (scale 1–7 from worst to best).

Statistical analyses

Scores were compared using independent samples' t test. The associations between parental stress, time from notification of the autoantibody result, and duration of the study were examined using linear regression analysis. Association between group and categorical variables in the epidemiological data were tested with χ2 statistics. The effect of epidemiological variables was analyzed with regression and univariate ANOVA. The SPSS for Windows release 11.0 software (SPSS, Chicago, IL) was used.

RESULTS

Sociodemographic characteristics

The parent groups were closely similar. Most lived in couples; however, more parents in the autoantibody-positive group than the control group lived alone (7.7 vs. 4.2%, P = 0.03). The proportion of chronically ill adults was higher in the autoantibody-positive group than in the control group (23.9 vs. 16.5%, P = 0.007); also, the unemployment rate tended to be higher. Similar trends were seen in the diabetic group.

Parenting stress

Stress indexes were similar in parents of antibody-positive children and control parents. Fathers experienced less stress than mothers (Table 1), considered parenthood easier (P = 0.008), and had more time for private life than mothers (P < 0.0001; data not shown). Control parents showed similar sex difference. Transient autoantibody positivity or the presence of multiple permanent autoantibodies in the child did not alter parental stress level. Of note, parental stress was similar whether or not the child participated in the prevention trial. There was no difference between parents of trial participants and parents who chose not to enroll an eligible child (Table 1).

Table 1
Parental stress expressed as parental stress index, with lower scores indicating higher stress levels

Parents whose child had developed diabetes showed higher stress than control subjects (Table 1). They also considered child care and parenthood more difficult (P = 0.032; data not shown) and regarded parents' responsibilities more demanding and social relations more difficult. They had more marital problems and more distant relation with their spouse than the control subjects (P = 0.013). The answers of the mothers and fathers were similar.

Parental stress decreased with duration of the follow-up (r = 0.142, P = 0.01). Single parents had higher stress than couples. Urban environment, unemployment, and chronic illness in the family were associated with higher stress. Parental stress increased with maternal age (r = −0.115, P = 0.039) but not with paternal age.

CONCLUSIONS

Parental stress was not increased when the family learned that their child had progressed to autoantibody positivity, or during the prevention trial. At enrollment, the implication was that although the 2–8% genetic risk was greater than the 0.7% in the background population, the odds were still strongly against a particular child to develop diabetes. Multiple autoantibodies increased the risk to >50%. The prevention trial presented a choice of taking an action with potentially beneficial consequences, or leaving the child without this option. The urge to do something to prevent diabetes is strong (6), and parents may see an intervention trial either as an opportunity to actively interfere in the course of events, or a daily reminder of the risk.

Parental anxiety is not significantly elevated in screening programs for type 1 diabetes risk and further dissipates over time (710). We did not observe the temporary increase in anxiety after notification of positive autoantibody results reported in some other studies (11). In agreement with the experiences in the Diabetes Prevention Trial–Type 1 (DPT-1) Study (11,12), even the long-term randomized prevention trial did not increase parental stress. In the ethnically homogeneous and well-educated Finnish population, the problems involved are probably smaller than in many other countries.

Mothers had higher stress than fathers, regarded parenthood as more demanding, and needed more social support. This may reflect traditional parental roles or a differential effect of risk awareness. Unrelated life experiences like single parenthood and chronic illness in the family increased the stress and may call for special attention.

Stress and early negative life events may associate with increased risk of chronic diseases, including type 1 diabetes (1315). The association between the development of autoimmunity and potentially stressful life circumstances (Table 1) supports this theory.

In conclusion, in a large population-based cohort of children at increased genetic risk for type 1 diabetes, parental stress was not increased by notification of autoantibody positivity or participation in the double-blinded prevention trial. The burden of risk awareness can be minimized by proper study setup.

Acknowledgments

This work was supported by grants from the Juvenile Diabetes Research Foundation International (4-1998-274, 4-1999-731, 4-2001-435), European Union (grant BMH4-CT98-3314); Novo Nordisk Foundation; Academy of Finland (grant 68292); Special Research Funds for University Hospitals in Finland; Finnish Office for Health Technology Assessment, Finland; Diabetes Research Foundation, Finland; Foundation for Pediatric Research, Finland; and Emil Aaltonen Foundation, Jalmari and Rauha Ahokas Foundation, Signe and Ane Gyllenberg Foundation, Yrjö Jahnsson Foundation, Sigrid Juselius Foundation, Päivikki and Sakari Sohlberg Foundation, and the Research Foundation of Orion Corporation, Finland.

No potential conflicts of interest relevant to this article were reported.

We thank Lauri Sillanmäki, Stud. Soc. Sci., University of Turku, Turku, Finland, for help in planning the statistical analyses of the data. We also thank the dedicated personnel of the DIPP study in Turku, Oulu, and Tampere. The essential contribution of the study children and their families is cordially acknowledged.

Footnotes

Clinical trial reg. no. NCT00223613, clinicaltrials.gov.

The costs of publication of this article were defrayed in part by the payment of page charges. This article must therefore be hereby marked “advertisement” in accordance with 18 U.S.C. Section 1734 solely to indicate this fact.

References

1. Ross LF. Minimizing risks: the ethics of predictive diabetes mellitus screening research in newborns. Arch Pediatr Adolesc Med 2003; 157: 89– 95 [PubMed]
2. Roth R. Psychological and ethical aspects of prevention trials. J Pediatr Endocrinol Metab 2001; 14 ( Suppl. 1): 669– 674 [PubMed]
3. Nanto-Salonen K, Kupila A, Simell S, Siljander H, Salonsaari T, Hekkala A, Korhonen S, Erkkola R, Sipila JI, Haavisto L, Siltala M, Tuominen J, Hakalax J, Hyoty H, Ilonen J, Veijola R, Simell T, Knip M, Simell O. Nasal insulin to prevent type 1 diabetes in children with HLA genotypes and autoantibodies conferring increased risk of disease: a double-blind, randomised controlled trial. Lancet 2008; 372: 1746– 1755 [PubMed]
4. Ostberg M. Parental stress, psychosocial problems and responsiveness in help-seeking parents with small (2–45 months old) children. Acta Paediatr 1998; 87: 69– 76 [PubMed]
5. Abidin RR. Parenting Stress Index: Professional Manual Odessa, FL, Psychological Assessment Resources, 1995
6. Baughcum AE, Johnson SB, Carmichael SK, Lewin AB, She JX, Schatz DA. Maternal efforts to prevent type 1 diabetes in at-risk children. Diabetes Care 2005; 28: 916– 921 [PubMed]
7. Bennett Johnson S, Baughcum AE, Carmichael SK, She JX, Schatz DA. Maternal anxiety associated with newborn genetic screening for type 1 diabetes. Diabetes Care 2004; 27: 392– 397 [PubMed]
8. Simonen P, Korhonen T, Simell T, Keskinen P, Karkkainen M, Knip M, Ilonen J, Simell O. Parental reactions to information about increased genetic risk of type 1 diabetes mellitus in infants. Arch Pediatr Adolesc Med 2006; 160: 1131– 1136 [PubMed]
9. Bennett Johnson S, Tercyak KP., Jr Psychological impact of islet cell antibody screening for IDDM on children, adults, and their family members. Diabetes Care 1995; 18: 1370– 1372 [PubMed]
10. Hummel M, Ziegler AG, Roth R. Psychological impact of childhood islet autoantibody testing in families participating in the BABYDIAB study. Diabet Med 2004; 21: 324– 328 [PubMed]
11. Johnson SB, Baughcum AE, Hood K, Rafkin-Mervis LE, Schatz DA. the DPT-1 Study Group. Participant and parent experiences in the parenteral insulin arm of the Diabetes Prevention Trial for Type 1 Diabetes. Diabetes Care 2007; 30: 2193– 2198 [PubMed]
12. Johnson SB, Baughcum AE, Rafkin-Mervis LE, Schatz DA. DPT-1 Study Group: Participant and parent experiences in the oral insulin study of the Diabetes Prevention Trial for Type 1 Diabetes. Pediatr Diabetes 2009; 10: 177– 183 [PubMed]
13. Russek LG, Schwartz GE, Bell IR, Baldwin CM. Positive perceptions of parental caring are associated with reduced psychiatric and somatic symptoms. Psychosom Med 1998; 60: 654– 657 [PubMed]
14. Karavanaki K, Tsoka E, Liacopoulou M, Karayianni C, Petrou V, Pippidou E, Brisimitzi M, Mavrikiou M, Kakleas K, Dacou-Voutetakis C. Psychological stress as a factor potentially contributing to the pathogenesis of type 1 diabetes mellitus. J Endocrinol Invest 2008; 31: 406– 415 [PubMed]
15. Sepa A, Frodi A, Ludvigsson J. Could parenting stress and lack of support/confidence function as mediating mechanisms between certain environmental factors and the development of autoimmunity in children? A study within ABIS. Ann N Y Acad Sci 2002; 958: 431– 435 [PubMed]

Articles from Diabetes Care are provided here courtesy of American Diabetes Association