Disability and Rehabilitation
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Parenting stress in mothers with cystic fibrosis Gerald Ullrich, Ingrid Bobis & Burkhard Bewig To cite this article: Gerald Ullrich, Ingrid Bobis & Burkhard Bewig (2015): Parenting stress in mothers with cystic fibrosis, Disability and Rehabilitation To link to this article: http://dx.doi.org/10.3109/09638288.2015.1031290
Published online: 10 Apr 2015.
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RESEARCH PAPER
Parenting stress in mothers with cystic fibrosis* Gerald Ullrich1, Ingrid Bobis2, and Burkhard Bewig2 Private Practice, Schwerin, Germany and 2Adult CF Centre, University Hospital Schleswig-Holstein, Kiel, Germany
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Abstract
Keywords
Aim: To assess the parenting experience of mothers with cystic fibrosis (CF) and to compare with normative data. Methods: Cross-sectional study with a validated generic parental stress questionnaire (PSQ). This PSQ differentiates four components of parental stress: main factor ‘‘parental stress’’, compounding factor ‘‘role restrictions’’, protective factors ‘‘support from spouse’’, and ‘‘social support’’. Cut-off scores categorise results as ‘‘normal’’, ‘‘borderline’’ or ‘‘concerning’’. Sample: Seventy-three women were informed by their local CF centre. Of these, 36 enrolled and had a first-born child aged 1–12 years (consistent with reference values of the PSQ). Of these, 31 (86%) returned the PSQ. Mean age of mothers was 32.6 years ± 6.9 years, mean age of first-born child was 5.2 years ± 3.4 years. Most of the mothers had one biological child, five women had two children and one had three children. Results: Parental stress scores were normally distributed, the same applies for contributing factors and for the two protective factors. Favourable scores were twice as frequent as concerning scores. Mothers of younger children scored slightly better than mothers of school-aged children. Conclusion: In line with the only comparable study, mothers with CF seem to be a remarkably resilient group who mostly cope well with parental stress even in the face of a progressive, chronic disease requiring time-consuming treatment.
Adults, cystic fibrosis, parenthood, stress History Received 28 September 2014 Revised 6 March 2015 Accepted 16 March 2015 Published online 10 April 2015
ä Implications for Rehabilitation
Today, motherhood is increasingly becoming an option in fertile women with cystic fibrosis. The additional burden of parenting seems to be rewarded by fulfilling essential personal goals. CF clinics should routinely address a possible wish for a child and to discuss it, openly.
Introduction Cystic fibrosis (CF) is one of the most common, life-shortening genetic diseases affecting young people of Caucasian background. It is a complex, multi-system disorder, with a heterogeneous presentation, a variable rate of progression and peak adverse effects occurring at varying times, mostly starting in childhood [1]. The primary impairments in CF affect respiratory and digestive structures and functions, but also fertility. In male, infertility is almost certain [2], while female are considered fertile, and women in stable health status and wishing to become pregnant, today are encouraged to do so [2–5]. The majority of mortality and morbidity is related to respiratory impairment, and the expected lifespan has increased into the fourth decade. Today, people with CF are however healthier than ever before when they enter adult life [6]. Not surprisingly then, parenthood is becoming an issue [5,7]. One of the workshops at the 2013 European Cystic Fibrosis Conference in Lisbon, Portugal, was even entitled ‘‘The pregnancy epidemic’’
*Portions of this work were presented at the 37th ECFC in Gothenburg, 11–14 June 2014. Address for correspondence: Dr Gerald Ullrich, Reutzstr. 1, 19055 Schwerin, Germany. E-mail: [email protected]
[8]. However, CF research almost completely missed the topic of parenthood. So far, there is only one systematic study from Sweden [9], though not yet published as a full paper. A mixed sample of 35 mothers and fathers with CF was assessed regarding parental stress and further aspects. Other studies [10–13], all of them with a qualitative approach, focus on the life experience of parents with CF, in general, but so far are unpublished, either. The lack of empirical data, on the life experience of parents with CF and specifically on the parenting experience, is a drawback for health caregivers since they are expected to counsel beyond the medical pros and cons of pregnancy in CF but also to include aspects of parenthood [2,14,15]. As long as respective data are lacking, this extended counselling will likely be omitted. Therefore, we aimed to systematically assess (a) the life experience of mothers with CF by means of semi-structured interviews and a questionnaire developed for the purpose of this study and (b) parenting stress by means of a standardised, validated questionnaire. The focus of the present article is on parenting stress in mothers with CF.
Methods This is a cross-sectional, mixed-methods study (qualitative and quantitative measures). The present part of the study is based on
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quantitative measures. Data were mainly assessed with a validated generic parental stress questionnaire (PSQ).
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Measures We used the ‘‘Elternstressfragebogen’’ [16], which is similar to established PSQs, such as the Parenting Stress Index [17]. The present PSQ refers to a transactional concept of stress which implies that stress is not qualified in itself but as a result of the individual’s perception of options to cope with the respective challenge. The instrument differentiates four subscales (components) of parenting stress: a main factor ‘‘parental stress’’ (17 items), a compounding factor ‘‘role restrictions’’ (7 items), and two buffering factors, namely ‘‘support from spouse’’ (7 items) and ‘‘social support’’ (7 items). Items are summed up to scores and cut-off scores classify results either as ‘‘normal’’, ‘‘borderline’’ or ‘‘concerning’’. The latter is interpreted as requiring further assessment. Categories refer to Stanine values and are equivalent to the following frequencies: 77% (‘‘normal’’), 12% (‘‘borderline’’), and 11% (‘‘concerning’’). High scores on ‘‘parental stress’’/‘‘role restriction’’ and low scores on ‘‘support from spouse’’/‘‘social support’’ indicate negative results. There are reference values both pooled for parents and exclusively for mothers and for fathers. Results may further be differentiated according to age of the child (infants and pre-schoolers versus school-aged children). Reference values are available for parents of children aged 1–12 years. An additional item of the PSQ refers to life events during the last year (occurrence; impact), and a final item asks about the general stressfulness of looking after the respective child (compared to siblings). Both of these items are descriptive (no normative data). This PSQ yielded satisfactory psychometric properties with internal consistencies ranging from a 0.76 to 0.92 and retest reliability ranging from rtt 0.76 to 0.91 [16]. An additional questionnaire was designed specifically for the purpose of the study. It explored the life experience of mothers with CF and assessed demographic and health-related data. This article only refers to data concerning the status of the former pregnancy (planned, unplanned) as well as to socio-demographic and health-related items. Health-related data were assessed by recall of respondents but not by medical records. Among others, respondents were asked about their FEV1 prior to pregnancy, 12 months after birth and at the time of the study. Respondents were to tell the respective FEV1 value or to indicate that they ‘‘don’t know/remember’’.
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We announced our study to all German CF centres by means of a nationwide mailing list. CF centres that were willing to cooperate disclosed the number of eligible women. Inclusion criteria were: (1) the mother had to be capable to understand and respond to the questionnaires, (2) her first-born child was at least 1 year and younger than 13 years (due to the reference values of the PSQ). We then sent invitation letters for each mother to the respective centre where staff forwarded our letter to the mother’s address. If the respective mother was willing to participate, she filled in the informed consent and a short demographic sheet and returned it in a prepaid envelope. Afterwards, we mailed the set of study questionnaires to each participant together with a postagepaid return envelope.
Sample Local CF centres reported on 73 ‘‘eligible’’ mothers. Of these, 44 signed informed consent. However, it turned out that centre staff had forwarded our invitation letters also to mothers not meeting the second inclusion criteria (i.e. their first-born child being younger than 1 year or older than 12 years), so the true number of participants eligible for PSQ assessment was 36 only. Of these, 31 filled in and returned the PSQ. Demographic details of the sample are summarised in Table 1. The majority of participants had given birth to one child and most of the mothers were currently in their thirties (Figure 1). Age at birth was 27 years ± 5.8 years with four mothers being younger than 20 years (youngest mother 16 years). Pregnancy had been planned in 25/31 women. Planning of pregnancy was unrelated to age. At the time of birth, most of the mothers were living with their partner/husband, while 23% were living with their parents. At the time of the study, 87% of women were still in partnership with the child’s father, while 13% either had separated meanwhile or had no contact to him right from the beginning. Almost half of the women (48%) currently earned money out of part-time employment. The mother’s CF was diagnosed most often during the first 2 years of life (74%), however 16% of participants were diagnosed late, as adolescents or as adults. Prior to becoming a mother, 19% of respondents denied taking enzymes as part of their routine CF therapy, and 13% neither used enzymes nor inhaled antibiotics, at that time. The average time devoted to daily CF treatment (including physiotherapy and sports) before pregnancy was 87 min (± 63 min), and was almost the same at the time of the study (83 min ± 53 min).
Results Statistical analysis As the main outcome, we used frequencies of the respective score categories (normal, borderline, concerning) in the sample of mothers with CF compared to the expected frequencies in a given (unselected) sample. Statistical significance was intended to be tested by 2-tests. Comparisons of the four subscale means were performed as a second-line outcome. Statistical significance was tested by t-tests. All statistical analyses were performed with GraphPad Prism 6.01 (GraphPad Software, Inc., La Jolla, CA). Further analyses, such as concerning life events, the mothers’ overall judgement about the stressfulness of parenting the firstborn child, or associations of scores with demographic variables were analysed, descriptively only. Procedure The study protocol was approved by the Institutional Review Board at the University Hospital Kiel, Germany.
As described, we will first report about the categorical results (i.e. number of clinically significant scores), next about comparison of means (which – even if elevated – may or may not be clinically meaningful), and finally about associations of PSQ scores to various variables (from an exploratory point of view). Frequency distributions Figures 2–5 show the results of descriptive frequency comparisons regarding the four PSQ scales in terms of either normal, borderline, or concerning scores. Over the four scales, there were only nine ‘‘concerning’’ scores (stemming from nine different subjects, i.e. no subject yielded two ‘‘concerning’’ scores) and 12 ‘‘borderline’’ scores. There were two women who scored high (i.e. concerning or borderline scores) on three of the four PSQ scales, indicating profound problems. One participant had a family background of migration and the other one made a narrative comment indicating a ‘‘chaotic’’ family life and that family therapy was already in operation.
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Table 1. Demographic and biomedical characteristics of the sample (n ¼ 31).
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Demographic aspects Age of mother (years; mean ± SD) Age of first-born child (years; mean ± SD) Age category of first-born child Infant (53 year) Pre-schooler (3–5 years) Primary school-age (6–9 year) Secondary school-age (49 years) Sex of first-born child (F/M%) Number of biological children One Two Three Married/living in partnership At least part-time employment (i.e. mother with own salary at the time of the study) Health-related aspects Mother’s age at CF diagnosis At birth/within first 2 years Up to 12 years During adolescence As an adult Current body mass index [BMI, kg/m2] (mean ± SD; range) BMI518 kg/m2 Current FEV1 in Perc. pred. (mean ± SD) FEV1550% CF-related diabetes Treatment with pancreatic enzymes prior to pregnancy Chronic infection with Pseudomonas aeruginosa Amount of time (minutes) devoted every day to CF therapies (i.e. including physiotherapy and sports) Prior to first pregnancy (mean ± SD; median) Currently (mean ± SD; median)
32.6 ± 6.9 5.2 ± 3.4 10/31 10/31 5/31 6/31 17:14
(32%) (32%) (16%) (19%) (55%/45%)
25/31 5/31 1/31 27/31 15/31
(81%) (16%) (3%) (87%) (48%)
23/31 (74%) 3/31 (10%) 2/31 (6%) 3/31 (10%) 20.8 ± 2.6; 16.8–28.3 4/31 (13%) 67 ± 17 (n ¼ 23)a 1/23 (4%) 12/31 (39%) 25/31 (81%) 27/31 (87%) 87 ± 63; 85 (n ¼ 28) 83 ± 53; 60 (n ¼ 29)
a
Thirteen mothers were unaware of their FEV1 or missing data.
Figure 2. Parenting stress in mothers of school-aged children (CF2) and of younger children (CF1) compared to the expected frequencies of ‘‘normal’’, ‘‘borderline’’ and ‘‘concerning’’ scores. Figure 1. Age distribution of participants and respective age of mother at giving birth to their first child.
Of note, favourable scores (i.e. high protective impact, low parenting stress or low contributing impact) were twice as frequent as concerning scores. Due to the low frequencies of scores in the ‘‘borderline’’ or ‘‘concerning’’ range, statistical tests regarding frequency distributions could not be computed.
stress in CF mothers of school-aged children compared to healthy mothers of primary school children. Also, there was a trend (p ¼ 0.08) to more social support in CF mothers of school-aged children compared to healthy mothers of children beyond primary school (Table 2). Remarkably, even though time pressure (due to the burden of CF treatment) was objectively higher for mothers with CF, their perception of role restrictions was almost identical to normative data.
Comparison of means Comparison of statistical means showed no statistically significant difference between CF mothers and reference values. However, there was a trend (p ¼ 0.09) to higher parenting
Exploratory data analysis If the child was a boy, ‘‘parental stress’’ tend to be higher (p ¼ 0.09), while the other three PSQ subscales showed no difference.
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trend to more favourable results in the group of working mothers (Table 4). Global rating and life events
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Figure 3. Compounding factor role restriction (RR) in CF mothers of school-aged children (CF2) and of younger children (CF1) compared to the expected frequencies of ‘‘normal’’, ‘‘borderline’’ and ‘‘concerning’’ scores.
Only the subscale on parenting stress highly correlated with mother’s overall judgement of the stressfulness of parenting the first-born child (r ¼ 0.78; p50.001), while the other scales were unrelated. Eight of the 10 mothers of school-aged children and 7/14 mothers of younger children considered parenting the child ‘‘somewhat’’ stressful, while only two mothers said that parenting the child was ‘‘very’’ stressful. Additional burden due to life events in the preceding year occurred in a number of families: divorce (3%), death of a relative (10%), serious illness or accident (23%), financial constraints (19%), or substantial alteration of the setting at home (e.g. moving house; 23%). If death of a relative, a serious illness, or financial constraints occurred, these were considered ‘‘somewhat’’ of ‘‘very’’ stressful, the other events were considered ‘‘hardly’’ stressful.
Discussion
Figure 4. Protective factor Support from Spouse (Spou) in CF mothers of school-aged children (CF2) and of younger children (CF1) compared to the expected frequencies of ‘‘normal’’, ‘‘borderline’’ and ‘‘concerning’’ scores.
Figure 5. Protective factor Social Support (Supp) in CF mothers of school-aged children (CF2) and of younger children (CF1) compared to the expected frequencies of ‘‘normal’’, ‘‘borderline’’ and ‘‘concerning’’ scores.
None of the subscales showed a difference according to whether or not pregnancy had been planned. Also, PSQ results were unrelated to mother’s age, FEV1, or amount of time spent for CF therapies. In order to describe the experience of child rearing from these mothers’ point of view, we report on the five most- and leastfrequently endorsed items of the PSQ (Table 3). Mothers who were at least part-time employed (6/11 mothers of school-aged children and 9/20 mothers of younger children) also responded similar to non-working mothers, or even showed a
The experience of parenting of mothers with CF so far did not attract much attention of the scientific community. This is the first study to report on the parenting experience of 31 mothers aged 21 years to 45 years, who mostly gave birth to one child. The parenting experience was assessed from a clinical point of view, i.e. focussing on parenting stress, and by means of an established, validated PSQ. The primary aim was to ascertain whether or not mothers with CF more often scored in the range categorised as ‘‘borderline’’ or even as ‘‘concerning’’ compared to the respective frequencies in an unselected sample. Remarkably, mothers with CF showed similar or even more favourable results for most of the PSQ scales. However, there was a trend to elevated ‘‘parenting stress’’ in mothers of school-aged children. Of note, while support from spouse was remarkably high in CF mothers of infants and pre-schoolers, this was less than average (though not statistically significant) for CF mothers of school-aged children. The vulnerable condition of young mothers with CF might trigger higher social support and more support from spouse, which might fade away as the child grows. The only other study, though so far only presented at a conference, also assessed the parenting experience by means of a PSQ in a sample of (n ¼ 35) Swedish mothers or fathers with CF [9]. This study, too, reported results comparable to normative data. Both studies, then, describe mothers with CF as a remarkably resilient group. To our knowledge, there are no studies on parenting that focus on chronically ill parents of healthy children. More often, the reverse is under study, namely how healthy parents experience rearing a chronically ill child. Typically, these parents show elevated parenting stress (e.g. [18,19]). Why are CF mothers of healthy children so normal? First, there will of course be kind of a positive bias due to a ‘‘need to be normal’’, which is well-known in the CF literature (e.g. [20]). Second, there will also be a positive bias due to response shift effects, namely that people with CF have another personal scale of stressors and strains. As one mother puts it in a telephone interview performed with a subset of mothers: ‘‘And for me, it is actually an incentive because I can see how Sarah is growing up, what she is doing, because I want to be part of it. And when I look at ‘normal’ mothers at the kindergarten (or elsewhere), who whine about things, I say then: ‘What sort of worries do you have?!’’
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Table 2. Comparison of means (PSQ subscales) between mothers with CF and normative values of three groups of healthy mothers (according to Domsch & Lohaus [16]). Infants and pre-schoolers
School-aged children Healthy
PSQ Subscale (M[SD])
CF n ¼ 20
Parental stressa Role restrictiona Support from spouseb Social supportb
13.04 9.45 16.00 12.95
(8.67) (4.82) (5.77) (4.66)
Healthy n ¼ 523 16.22 10.18 14.05 12.17
(8.92) (5.02) (4.75) (4.62)
c
CF n ¼ 11 22.64 8.36 12.05 12.18
Forms 1–4 n ¼ 694
(8.37) (4.23) (4.37) (4.35)
17.33 8.99 13.80 11.01
(10.27) (4.75) (4.64) (4.91)
Forms 5–6 n ¼ 668c 17.79 8.37 13.91 9.68
(10.08) (4.82) (5.04) (4.76)
Forms 1 to 4 and Forms 5 to 6 are synonymous to Grade level 1 to 4 (primary school) and grades 5 to 6 (beyond primary school), respectively. Lower scores more favourable. b Higher scores more favourable. c Reduced sample size regarding ‘‘Support from spouse’’: n ¼ 625 and n ¼ 583, respectively.
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a
Table 3. The five most- and least-frequently endorsed PSQ-items. Item title
Percentage a
Highest approval 1. There are people around who look after my child 2. With respect to child rearing, my partner and I fully agree on all aspects 3. My partner understands my concerns 4. In difficult situations with my child; I cope well 5. If I have questions about my child, there are friends or other people I can refer to Lowest approvala 1. I repeatedly feel embarrassed about my child’s behaviour 2. As a mother, there are some tasks that I have difficulty coping with 3. At times I feel helpless about my child’s behaviour 4. My child is demanding 5. (a) There are days when I feel uncertain about rearing my childb (b) Supervising my child’s homework is a challenging task for meb
87 87 83 78 77 9 13 16 22 26
a
Responses to the PSQ, both agreement and disagreement, were consolidated from two options to one option. b Different wording according to age of the child: (a) infants and pre-schoolers, (b) school-aged children. Table 4. Employment status and parenting stress in mothers with CF. Scales and groups PSQ scale: Parenting stress (main factor)* Mothers of infants and pre-schoolers (9 working, 11 non-working)a Mothers school-aged children (6 working, 5 non-working) PSQ scale: Role restriction (compounding factor)* Mothers of infants and pre-schoolers (9 working, 11 non-working) Mothers school-aged children (6 working, 5 non-working) PSQ scale: Support from spouse (buffering factor)** Mothers of infants and pre-schoolers (9 working, 11 non-working)b Mothers school-aged children (6 working, 5 non-working) PSQ scale: Social support (buffering factor)** Mothers of infants and pre-schoolers (9 working, 11 non-working)c Mothers school-aged children (6 working, 5 non-working)
Working M (SD)
Not working M (SD)
9.4 (7.0) 22.7 (7.7)
16.6 (8.9) 22.6 (10.1)
8.2 (5.6) 7.3 (2.9)
10.5 (3.9) 9.6 (5.6)
18.8 (6.0) 12.8 (5.3)
13.7 (4.5) 11.2 (3.2)
14.9 (3.4) 13.04 (8.67)
11.4 (5.1) 10.02 (4.7)
*Lower scores more favourable. **Higher scores more favourable. a p ¼ 0.06. b p ¼ 0.04. c p ¼ 0.09.
Third, a response shift effect may also result from the fact, that motherhood had mostly not been self-evident for these young women with CF, since many of them were raised with the grim outlook of dying early and not being able to lead a full adult life [12,21]. During our interviews, as well as in narrative comments to the purpose-designed questionnaire, a number of respondents referred to the ‘‘right’’ of the woman with CF to become a mother (similarly [22]). If this ‘‘right’’ is questioned, either by virtue of
the disease or by critical comments of parents or healthcare professionals, finally becoming a mother will then likely be described in even more positive terms. Last but not least, positive (or rather ‘‘normal’’) PSQ results will probably be a consequence of raising healthy (i.e. ‘‘normal’’) children. Therefore, the initial question, why CF mothers are so normal, should perhaps be reworded: Why should they be different?!
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Limitations and future directions This study has several limitations. First, we relied on normative data provided by the validated instrument, but did not include a control group. This may be misleading [23]. However, reference scores of this PSQ are rather up-to-date, since this measure was published only recently. Second, we used a convenience sample and women with discouraging experience of motherhood might have avoided enrolment. However, we clearly advised cooperating CF centres not to select candidates but to forward our invitation letter to all eligible mothers. Third, although our study is the largest on mothers with CF, the sample size still was small, at least regarding in-between group comparisons (working versus not working CF mothers). Despite these limitations, our study indicated that motherhood is a viable option in woman with CF. Future studies will show whether the trend to less favourable results for mothers of older children may be replicated.
Acknowledgements We appreciate the support from following colleagues: CF nurse specialist Kerscher and PD Dr Fischer (Munich), Dipl. Soz.-Pa¨d. Becker (Essen), Dr Sauer-Heilborn (Hanover), Prof. Bargon (Frankfurt/Main), PD Dr Kappler (Munich), Dr Dieninghoff (Cologne), Dr Hammermann (Dresden), Dr Heyder (Stuttgart), Dr Mainz (Jena), PD Dr Mellies (Essen), Dr Na¨hrlich (Gießen), Dr Ko¨ster (Oldenburg), Dr Claßen (Bremen), Dr Kinder (Neubrandenburg), Prof. Skopnik (Worms), Prof. Hebestreit (Wu¨rzburg), Dr Breuel (Rostock), Dr Heuer (Hamburg), Dr Smaczny (Frankfurt/Main), Dr Vogl-Vosswinkel (Munich), Dr Rietschel (Cologne), Dr Wiebel/Dr Po¨mpeler (Heidelberg), Dr Bu¨sing (Osnabru¨ck), Prof. Ballmann (Bochum), PD Dr Gru¨ber (Frankfurt/Oder). Finally, we are grateful to Prof. Gratiana Steinkamp for helpful comments to a prior draft of this article.
Declaration of interest This study was supported in total by Novartis Pharma GmbH, Germany. Dr Bobis and Prof. Bewig wish to confirm that there are no known conflicts of interest associated with this publication and there has been no significant financial support for this work that could have influenced its outcome. Dr Ullrich declares to have been paid for substantial parts of the research the results of which are described in this article. He also received a professional fee for preparation of the article. Research funding and source of payment were identical, namely Novartis Pharma GmbH, Germany. The authors declare no conflicts of interests. The authors alone are responsible for the content and writing of this article.
References 1. Hodson ME, Geddes D, Bush A. Cystic fibrosis. London, United Kingdom: Arnold Publ; 2007.
Disabil Rehabil, Early Online: 1–6
2. Sawyer SM. Sexual and reproductive health. In: Hodson M, Geddes D, Bush A, eds. Cystic fibrosis. London, United Kingdom: Arnold Publ; 2007:279–90. 3. Edenborough FP. Women with cystic fibrosis and their potential for reproduction. Thorax 2001;56:649–55. 4. Wexler ID, Johannesson M, Edenborough FP, et al. Pregnancy and chronic progressive pulmonary disease. Am J Respir Crit Care Med 2007;175:300–5. 5. Edenborough FP, Borgo G, Knoop C, et al. Guidelines for the management of pregnancy in women with cystic fibrosis. J Cyst Fibros 2008;7:s2–32. 6. Hardt Hvd, Schwarz C, Ullrich G. [Adults with cystic fibrosis. It’s not just about longevity] Erwachsene mit Mukoviszidose. Es geht um mehr als die Lebensdauer. Bundesgesundheitsblatt Gesundheitsforschung Gesundheitsschutz 2012;55:558–67. 7. Go¨tz I, Go¨tz M. Reproduction and parenting. In: Bluebond-Langner M, Lask B, Angst DB, eds. Psychosocial aspects of cystic fibrosis. London, United Kingdom: Arnold; 2001:172–87. 8. Anonymous. Abstracts of the 36th European Cystic Fibrosis Conference. Lisbon, Portugal, 2013 Jun 12–15. J Cyst Fibros 2013;12:s36. 9. Frankl M, Hjelte L. Parents who have cystic fibrosis – experiences and aspects of parenthood. J Cyst Fibros 2004;3:s103 [Abstract]. 10. Erwander I. Experiences of parenthood among women and men with cystic fibrosis. 24th European Cystic Fibrosis Conference, 2001 Jun 6–9, Vienna. J Cyst Fibros (Nullnummer) Book of Abstracts 2001:371. 11. Colpaert K, Havermans T. CF and parenting. Presented at the 32nd European CF Conference, Brest, 2009 June 10–13. Slides online available. Available from: http://www.ecfs.eu/projects/internationalnurse-specialist-group-cf/presentations [last accessed 8 Aug 2013]. 12. Li A, Fulbrook P, Bell S. Journey to parenthood: experience of adults with Cystic fibrosis. Presented at the 8th Australasian CF Conference, 2009, 28 Aug to 1 Sept, Brisbane. 13. Cammidge S, Latchford G, Duff A, Etherington C. When women with CF become mothers: a qualitative study of psychosocial impact and adjustment. J Cyst Fibros 2013;12:s37 (No. WS18.5) [Abstract]. 14. Tsang A, Moriarty C, Towns S. Contraception, communication and counseling for sexuality and reproductive health in adolescents and young adults with CF. Paediatr Respir Rev 2010;11:84–9. 15. Simcox AM, Duff AJ, Morton AM, et al. Decision making about reproduction and pregnancy by women with cystic fibrosis. Br J Hosp Med 2009;70:639–43. 16. Domsch H, Lohaus A. [Parent Stress Questionnaire] Elternstressfragebogen (ESF): Manual. Go¨ttingen: Hogrefe, 2010. 17. Abidin RR. Parenting stress index. Charlottesville (VA): Pediatric Psychology Press; 1990. 18. Feizi A, Najmi B, Salesi A, et al. Parenting stress among mothers of children with different physical, mental, and psychological problems. J Res Med Sci 2014;19:145–52. 19. Epifanio MS, Genna V, Vitello MG, et al. Parenting stress and impact of illness in parents of children with coeliac disease. Pediatr Rep 2013;5:81–5. 20. Bluebond-Langner M, Lask B, Angst DBH. Psychosocial aspects of cystic fibrosis. London, United Kingdom: Arnold; 2001. 21. Jessup M, Parkinson C. ‘All at the sea’: the experience of living with cystic fibrosis. Qual Health Res 2010;20:352–64. 22. Simcox AM, Hewison J, Duff AJ, et al. Decision-making about pregnancy for women with cystic fibrosis. Br J Health Psychol 2009; 14:323–42. 23. Gerhardt CA, Vannatta K, McKellop JM, et al. Comparing parental distress, family functioning, and the role of social support for caregivers with and without a child with juvenile rheumatoid arthritis. J Pediatr Psychol 2003;28:5–15.
ISSN: 0963-8288 (Print) 1464-5165 (Online) Journal homepage: http://www.tandfonline.com/loi/idre20
Parenting stress in mothers with cystic fibrosis Gerald Ullrich, Ingrid Bobis & Burkhard Bewig To cite this article: Gerald Ullrich, Ingrid Bobis & Burkhard Bewig (2015): Parenting stress in mothers with cystic fibrosis, Disability and Rehabilitation To link to this article: http://dx.doi.org/10.3109/09638288.2015.1031290
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RESEARCH PAPER
Parenting stress in mothers with cystic fibrosis* Gerald Ullrich1, Ingrid Bobis2, and Burkhard Bewig2 Private Practice, Schwerin, Germany and 2Adult CF Centre, University Hospital Schleswig-Holstein, Kiel, Germany
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Abstract
Keywords
Aim: To assess the parenting experience of mothers with cystic fibrosis (CF) and to compare with normative data. Methods: Cross-sectional study with a validated generic parental stress questionnaire (PSQ). This PSQ differentiates four components of parental stress: main factor ‘‘parental stress’’, compounding factor ‘‘role restrictions’’, protective factors ‘‘support from spouse’’, and ‘‘social support’’. Cut-off scores categorise results as ‘‘normal’’, ‘‘borderline’’ or ‘‘concerning’’. Sample: Seventy-three women were informed by their local CF centre. Of these, 36 enrolled and had a first-born child aged 1–12 years (consistent with reference values of the PSQ). Of these, 31 (86%) returned the PSQ. Mean age of mothers was 32.6 years ± 6.9 years, mean age of first-born child was 5.2 years ± 3.4 years. Most of the mothers had one biological child, five women had two children and one had three children. Results: Parental stress scores were normally distributed, the same applies for contributing factors and for the two protective factors. Favourable scores were twice as frequent as concerning scores. Mothers of younger children scored slightly better than mothers of school-aged children. Conclusion: In line with the only comparable study, mothers with CF seem to be a remarkably resilient group who mostly cope well with parental stress even in the face of a progressive, chronic disease requiring time-consuming treatment.
Adults, cystic fibrosis, parenthood, stress History Received 28 September 2014 Revised 6 March 2015 Accepted 16 March 2015 Published online 10 April 2015
ä Implications for Rehabilitation
Today, motherhood is increasingly becoming an option in fertile women with cystic fibrosis. The additional burden of parenting seems to be rewarded by fulfilling essential personal goals. CF clinics should routinely address a possible wish for a child and to discuss it, openly.
Introduction Cystic fibrosis (CF) is one of the most common, life-shortening genetic diseases affecting young people of Caucasian background. It is a complex, multi-system disorder, with a heterogeneous presentation, a variable rate of progression and peak adverse effects occurring at varying times, mostly starting in childhood [1]. The primary impairments in CF affect respiratory and digestive structures and functions, but also fertility. In male, infertility is almost certain [2], while female are considered fertile, and women in stable health status and wishing to become pregnant, today are encouraged to do so [2–5]. The majority of mortality and morbidity is related to respiratory impairment, and the expected lifespan has increased into the fourth decade. Today, people with CF are however healthier than ever before when they enter adult life [6]. Not surprisingly then, parenthood is becoming an issue [5,7]. One of the workshops at the 2013 European Cystic Fibrosis Conference in Lisbon, Portugal, was even entitled ‘‘The pregnancy epidemic’’
*Portions of this work were presented at the 37th ECFC in Gothenburg, 11–14 June 2014. Address for correspondence: Dr Gerald Ullrich, Reutzstr. 1, 19055 Schwerin, Germany. E-mail: [email protected]
[8]. However, CF research almost completely missed the topic of parenthood. So far, there is only one systematic study from Sweden [9], though not yet published as a full paper. A mixed sample of 35 mothers and fathers with CF was assessed regarding parental stress and further aspects. Other studies [10–13], all of them with a qualitative approach, focus on the life experience of parents with CF, in general, but so far are unpublished, either. The lack of empirical data, on the life experience of parents with CF and specifically on the parenting experience, is a drawback for health caregivers since they are expected to counsel beyond the medical pros and cons of pregnancy in CF but also to include aspects of parenthood [2,14,15]. As long as respective data are lacking, this extended counselling will likely be omitted. Therefore, we aimed to systematically assess (a) the life experience of mothers with CF by means of semi-structured interviews and a questionnaire developed for the purpose of this study and (b) parenting stress by means of a standardised, validated questionnaire. The focus of the present article is on parenting stress in mothers with CF.
Methods This is a cross-sectional, mixed-methods study (qualitative and quantitative measures). The present part of the study is based on
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quantitative measures. Data were mainly assessed with a validated generic parental stress questionnaire (PSQ).
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Measures We used the ‘‘Elternstressfragebogen’’ [16], which is similar to established PSQs, such as the Parenting Stress Index [17]. The present PSQ refers to a transactional concept of stress which implies that stress is not qualified in itself but as a result of the individual’s perception of options to cope with the respective challenge. The instrument differentiates four subscales (components) of parenting stress: a main factor ‘‘parental stress’’ (17 items), a compounding factor ‘‘role restrictions’’ (7 items), and two buffering factors, namely ‘‘support from spouse’’ (7 items) and ‘‘social support’’ (7 items). Items are summed up to scores and cut-off scores classify results either as ‘‘normal’’, ‘‘borderline’’ or ‘‘concerning’’. The latter is interpreted as requiring further assessment. Categories refer to Stanine values and are equivalent to the following frequencies: 77% (‘‘normal’’), 12% (‘‘borderline’’), and 11% (‘‘concerning’’). High scores on ‘‘parental stress’’/‘‘role restriction’’ and low scores on ‘‘support from spouse’’/‘‘social support’’ indicate negative results. There are reference values both pooled for parents and exclusively for mothers and for fathers. Results may further be differentiated according to age of the child (infants and pre-schoolers versus school-aged children). Reference values are available for parents of children aged 1–12 years. An additional item of the PSQ refers to life events during the last year (occurrence; impact), and a final item asks about the general stressfulness of looking after the respective child (compared to siblings). Both of these items are descriptive (no normative data). This PSQ yielded satisfactory psychometric properties with internal consistencies ranging from a 0.76 to 0.92 and retest reliability ranging from rtt 0.76 to 0.91 [16]. An additional questionnaire was designed specifically for the purpose of the study. It explored the life experience of mothers with CF and assessed demographic and health-related data. This article only refers to data concerning the status of the former pregnancy (planned, unplanned) as well as to socio-demographic and health-related items. Health-related data were assessed by recall of respondents but not by medical records. Among others, respondents were asked about their FEV1 prior to pregnancy, 12 months after birth and at the time of the study. Respondents were to tell the respective FEV1 value or to indicate that they ‘‘don’t know/remember’’.
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We announced our study to all German CF centres by means of a nationwide mailing list. CF centres that were willing to cooperate disclosed the number of eligible women. Inclusion criteria were: (1) the mother had to be capable to understand and respond to the questionnaires, (2) her first-born child was at least 1 year and younger than 13 years (due to the reference values of the PSQ). We then sent invitation letters for each mother to the respective centre where staff forwarded our letter to the mother’s address. If the respective mother was willing to participate, she filled in the informed consent and a short demographic sheet and returned it in a prepaid envelope. Afterwards, we mailed the set of study questionnaires to each participant together with a postagepaid return envelope.
Sample Local CF centres reported on 73 ‘‘eligible’’ mothers. Of these, 44 signed informed consent. However, it turned out that centre staff had forwarded our invitation letters also to mothers not meeting the second inclusion criteria (i.e. their first-born child being younger than 1 year or older than 12 years), so the true number of participants eligible for PSQ assessment was 36 only. Of these, 31 filled in and returned the PSQ. Demographic details of the sample are summarised in Table 1. The majority of participants had given birth to one child and most of the mothers were currently in their thirties (Figure 1). Age at birth was 27 years ± 5.8 years with four mothers being younger than 20 years (youngest mother 16 years). Pregnancy had been planned in 25/31 women. Planning of pregnancy was unrelated to age. At the time of birth, most of the mothers were living with their partner/husband, while 23% were living with their parents. At the time of the study, 87% of women were still in partnership with the child’s father, while 13% either had separated meanwhile or had no contact to him right from the beginning. Almost half of the women (48%) currently earned money out of part-time employment. The mother’s CF was diagnosed most often during the first 2 years of life (74%), however 16% of participants were diagnosed late, as adolescents or as adults. Prior to becoming a mother, 19% of respondents denied taking enzymes as part of their routine CF therapy, and 13% neither used enzymes nor inhaled antibiotics, at that time. The average time devoted to daily CF treatment (including physiotherapy and sports) before pregnancy was 87 min (± 63 min), and was almost the same at the time of the study (83 min ± 53 min).
Results Statistical analysis As the main outcome, we used frequencies of the respective score categories (normal, borderline, concerning) in the sample of mothers with CF compared to the expected frequencies in a given (unselected) sample. Statistical significance was intended to be tested by 2-tests. Comparisons of the four subscale means were performed as a second-line outcome. Statistical significance was tested by t-tests. All statistical analyses were performed with GraphPad Prism 6.01 (GraphPad Software, Inc., La Jolla, CA). Further analyses, such as concerning life events, the mothers’ overall judgement about the stressfulness of parenting the firstborn child, or associations of scores with demographic variables were analysed, descriptively only. Procedure The study protocol was approved by the Institutional Review Board at the University Hospital Kiel, Germany.
As described, we will first report about the categorical results (i.e. number of clinically significant scores), next about comparison of means (which – even if elevated – may or may not be clinically meaningful), and finally about associations of PSQ scores to various variables (from an exploratory point of view). Frequency distributions Figures 2–5 show the results of descriptive frequency comparisons regarding the four PSQ scales in terms of either normal, borderline, or concerning scores. Over the four scales, there were only nine ‘‘concerning’’ scores (stemming from nine different subjects, i.e. no subject yielded two ‘‘concerning’’ scores) and 12 ‘‘borderline’’ scores. There were two women who scored high (i.e. concerning or borderline scores) on three of the four PSQ scales, indicating profound problems. One participant had a family background of migration and the other one made a narrative comment indicating a ‘‘chaotic’’ family life and that family therapy was already in operation.
Parenting stress
DOI: 10.3109/09638288.2015.1031290
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Table 1. Demographic and biomedical characteristics of the sample (n ¼ 31).
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Demographic aspects Age of mother (years; mean ± SD) Age of first-born child (years; mean ± SD) Age category of first-born child Infant (53 year) Pre-schooler (3–5 years) Primary school-age (6–9 year) Secondary school-age (49 years) Sex of first-born child (F/M%) Number of biological children One Two Three Married/living in partnership At least part-time employment (i.e. mother with own salary at the time of the study) Health-related aspects Mother’s age at CF diagnosis At birth/within first 2 years Up to 12 years During adolescence As an adult Current body mass index [BMI, kg/m2] (mean ± SD; range) BMI518 kg/m2 Current FEV1 in Perc. pred. (mean ± SD) FEV1550% CF-related diabetes Treatment with pancreatic enzymes prior to pregnancy Chronic infection with Pseudomonas aeruginosa Amount of time (minutes) devoted every day to CF therapies (i.e. including physiotherapy and sports) Prior to first pregnancy (mean ± SD; median) Currently (mean ± SD; median)
32.6 ± 6.9 5.2 ± 3.4 10/31 10/31 5/31 6/31 17:14
(32%) (32%) (16%) (19%) (55%/45%)
25/31 5/31 1/31 27/31 15/31
(81%) (16%) (3%) (87%) (48%)
23/31 (74%) 3/31 (10%) 2/31 (6%) 3/31 (10%) 20.8 ± 2.6; 16.8–28.3 4/31 (13%) 67 ± 17 (n ¼ 23)a 1/23 (4%) 12/31 (39%) 25/31 (81%) 27/31 (87%) 87 ± 63; 85 (n ¼ 28) 83 ± 53; 60 (n ¼ 29)
a
Thirteen mothers were unaware of their FEV1 or missing data.
Figure 2. Parenting stress in mothers of school-aged children (CF2) and of younger children (CF1) compared to the expected frequencies of ‘‘normal’’, ‘‘borderline’’ and ‘‘concerning’’ scores. Figure 1. Age distribution of participants and respective age of mother at giving birth to their first child.
Of note, favourable scores (i.e. high protective impact, low parenting stress or low contributing impact) were twice as frequent as concerning scores. Due to the low frequencies of scores in the ‘‘borderline’’ or ‘‘concerning’’ range, statistical tests regarding frequency distributions could not be computed.
stress in CF mothers of school-aged children compared to healthy mothers of primary school children. Also, there was a trend (p ¼ 0.08) to more social support in CF mothers of school-aged children compared to healthy mothers of children beyond primary school (Table 2). Remarkably, even though time pressure (due to the burden of CF treatment) was objectively higher for mothers with CF, their perception of role restrictions was almost identical to normative data.
Comparison of means Comparison of statistical means showed no statistically significant difference between CF mothers and reference values. However, there was a trend (p ¼ 0.09) to higher parenting
Exploratory data analysis If the child was a boy, ‘‘parental stress’’ tend to be higher (p ¼ 0.09), while the other three PSQ subscales showed no difference.
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trend to more favourable results in the group of working mothers (Table 4). Global rating and life events
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Figure 3. Compounding factor role restriction (RR) in CF mothers of school-aged children (CF2) and of younger children (CF1) compared to the expected frequencies of ‘‘normal’’, ‘‘borderline’’ and ‘‘concerning’’ scores.
Only the subscale on parenting stress highly correlated with mother’s overall judgement of the stressfulness of parenting the first-born child (r ¼ 0.78; p50.001), while the other scales were unrelated. Eight of the 10 mothers of school-aged children and 7/14 mothers of younger children considered parenting the child ‘‘somewhat’’ stressful, while only two mothers said that parenting the child was ‘‘very’’ stressful. Additional burden due to life events in the preceding year occurred in a number of families: divorce (3%), death of a relative (10%), serious illness or accident (23%), financial constraints (19%), or substantial alteration of the setting at home (e.g. moving house; 23%). If death of a relative, a serious illness, or financial constraints occurred, these were considered ‘‘somewhat’’ of ‘‘very’’ stressful, the other events were considered ‘‘hardly’’ stressful.
Discussion
Figure 4. Protective factor Support from Spouse (Spou) in CF mothers of school-aged children (CF2) and of younger children (CF1) compared to the expected frequencies of ‘‘normal’’, ‘‘borderline’’ and ‘‘concerning’’ scores.
Figure 5. Protective factor Social Support (Supp) in CF mothers of school-aged children (CF2) and of younger children (CF1) compared to the expected frequencies of ‘‘normal’’, ‘‘borderline’’ and ‘‘concerning’’ scores.
None of the subscales showed a difference according to whether or not pregnancy had been planned. Also, PSQ results were unrelated to mother’s age, FEV1, or amount of time spent for CF therapies. In order to describe the experience of child rearing from these mothers’ point of view, we report on the five most- and leastfrequently endorsed items of the PSQ (Table 3). Mothers who were at least part-time employed (6/11 mothers of school-aged children and 9/20 mothers of younger children) also responded similar to non-working mothers, or even showed a
The experience of parenting of mothers with CF so far did not attract much attention of the scientific community. This is the first study to report on the parenting experience of 31 mothers aged 21 years to 45 years, who mostly gave birth to one child. The parenting experience was assessed from a clinical point of view, i.e. focussing on parenting stress, and by means of an established, validated PSQ. The primary aim was to ascertain whether or not mothers with CF more often scored in the range categorised as ‘‘borderline’’ or even as ‘‘concerning’’ compared to the respective frequencies in an unselected sample. Remarkably, mothers with CF showed similar or even more favourable results for most of the PSQ scales. However, there was a trend to elevated ‘‘parenting stress’’ in mothers of school-aged children. Of note, while support from spouse was remarkably high in CF mothers of infants and pre-schoolers, this was less than average (though not statistically significant) for CF mothers of school-aged children. The vulnerable condition of young mothers with CF might trigger higher social support and more support from spouse, which might fade away as the child grows. The only other study, though so far only presented at a conference, also assessed the parenting experience by means of a PSQ in a sample of (n ¼ 35) Swedish mothers or fathers with CF [9]. This study, too, reported results comparable to normative data. Both studies, then, describe mothers with CF as a remarkably resilient group. To our knowledge, there are no studies on parenting that focus on chronically ill parents of healthy children. More often, the reverse is under study, namely how healthy parents experience rearing a chronically ill child. Typically, these parents show elevated parenting stress (e.g. [18,19]). Why are CF mothers of healthy children so normal? First, there will of course be kind of a positive bias due to a ‘‘need to be normal’’, which is well-known in the CF literature (e.g. [20]). Second, there will also be a positive bias due to response shift effects, namely that people with CF have another personal scale of stressors and strains. As one mother puts it in a telephone interview performed with a subset of mothers: ‘‘And for me, it is actually an incentive because I can see how Sarah is growing up, what she is doing, because I want to be part of it. And when I look at ‘normal’ mothers at the kindergarten (or elsewhere), who whine about things, I say then: ‘What sort of worries do you have?!’’
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Table 2. Comparison of means (PSQ subscales) between mothers with CF and normative values of three groups of healthy mothers (according to Domsch & Lohaus [16]). Infants and pre-schoolers
School-aged children Healthy
PSQ Subscale (M[SD])
CF n ¼ 20
Parental stressa Role restrictiona Support from spouseb Social supportb
13.04 9.45 16.00 12.95
(8.67) (4.82) (5.77) (4.66)
Healthy n ¼ 523 16.22 10.18 14.05 12.17
(8.92) (5.02) (4.75) (4.62)
c
CF n ¼ 11 22.64 8.36 12.05 12.18
Forms 1–4 n ¼ 694
(8.37) (4.23) (4.37) (4.35)
17.33 8.99 13.80 11.01
(10.27) (4.75) (4.64) (4.91)
Forms 5–6 n ¼ 668c 17.79 8.37 13.91 9.68
(10.08) (4.82) (5.04) (4.76)
Forms 1 to 4 and Forms 5 to 6 are synonymous to Grade level 1 to 4 (primary school) and grades 5 to 6 (beyond primary school), respectively. Lower scores more favourable. b Higher scores more favourable. c Reduced sample size regarding ‘‘Support from spouse’’: n ¼ 625 and n ¼ 583, respectively.
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a
Table 3. The five most- and least-frequently endorsed PSQ-items. Item title
Percentage a
Highest approval 1. There are people around who look after my child 2. With respect to child rearing, my partner and I fully agree on all aspects 3. My partner understands my concerns 4. In difficult situations with my child; I cope well 5. If I have questions about my child, there are friends or other people I can refer to Lowest approvala 1. I repeatedly feel embarrassed about my child’s behaviour 2. As a mother, there are some tasks that I have difficulty coping with 3. At times I feel helpless about my child’s behaviour 4. My child is demanding 5. (a) There are days when I feel uncertain about rearing my childb (b) Supervising my child’s homework is a challenging task for meb
87 87 83 78 77 9 13 16 22 26
a
Responses to the PSQ, both agreement and disagreement, were consolidated from two options to one option. b Different wording according to age of the child: (a) infants and pre-schoolers, (b) school-aged children. Table 4. Employment status and parenting stress in mothers with CF. Scales and groups PSQ scale: Parenting stress (main factor)* Mothers of infants and pre-schoolers (9 working, 11 non-working)a Mothers school-aged children (6 working, 5 non-working) PSQ scale: Role restriction (compounding factor)* Mothers of infants and pre-schoolers (9 working, 11 non-working) Mothers school-aged children (6 working, 5 non-working) PSQ scale: Support from spouse (buffering factor)** Mothers of infants and pre-schoolers (9 working, 11 non-working)b Mothers school-aged children (6 working, 5 non-working) PSQ scale: Social support (buffering factor)** Mothers of infants and pre-schoolers (9 working, 11 non-working)c Mothers school-aged children (6 working, 5 non-working)
Working M (SD)
Not working M (SD)
9.4 (7.0) 22.7 (7.7)
16.6 (8.9) 22.6 (10.1)
8.2 (5.6) 7.3 (2.9)
10.5 (3.9) 9.6 (5.6)
18.8 (6.0) 12.8 (5.3)
13.7 (4.5) 11.2 (3.2)
14.9 (3.4) 13.04 (8.67)
11.4 (5.1) 10.02 (4.7)
*Lower scores more favourable. **Higher scores more favourable. a p ¼ 0.06. b p ¼ 0.04. c p ¼ 0.09.
Third, a response shift effect may also result from the fact, that motherhood had mostly not been self-evident for these young women with CF, since many of them were raised with the grim outlook of dying early and not being able to lead a full adult life [12,21]. During our interviews, as well as in narrative comments to the purpose-designed questionnaire, a number of respondents referred to the ‘‘right’’ of the woman with CF to become a mother (similarly [22]). If this ‘‘right’’ is questioned, either by virtue of
the disease or by critical comments of parents or healthcare professionals, finally becoming a mother will then likely be described in even more positive terms. Last but not least, positive (or rather ‘‘normal’’) PSQ results will probably be a consequence of raising healthy (i.e. ‘‘normal’’) children. Therefore, the initial question, why CF mothers are so normal, should perhaps be reworded: Why should they be different?!
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Limitations and future directions This study has several limitations. First, we relied on normative data provided by the validated instrument, but did not include a control group. This may be misleading [23]. However, reference scores of this PSQ are rather up-to-date, since this measure was published only recently. Second, we used a convenience sample and women with discouraging experience of motherhood might have avoided enrolment. However, we clearly advised cooperating CF centres not to select candidates but to forward our invitation letter to all eligible mothers. Third, although our study is the largest on mothers with CF, the sample size still was small, at least regarding in-between group comparisons (working versus not working CF mothers). Despite these limitations, our study indicated that motherhood is a viable option in woman with CF. Future studies will show whether the trend to less favourable results for mothers of older children may be replicated.
Acknowledgements We appreciate the support from following colleagues: CF nurse specialist Kerscher and PD Dr Fischer (Munich), Dipl. Soz.-Pa¨d. Becker (Essen), Dr Sauer-Heilborn (Hanover), Prof. Bargon (Frankfurt/Main), PD Dr Kappler (Munich), Dr Dieninghoff (Cologne), Dr Hammermann (Dresden), Dr Heyder (Stuttgart), Dr Mainz (Jena), PD Dr Mellies (Essen), Dr Na¨hrlich (Gießen), Dr Ko¨ster (Oldenburg), Dr Claßen (Bremen), Dr Kinder (Neubrandenburg), Prof. Skopnik (Worms), Prof. Hebestreit (Wu¨rzburg), Dr Breuel (Rostock), Dr Heuer (Hamburg), Dr Smaczny (Frankfurt/Main), Dr Vogl-Vosswinkel (Munich), Dr Rietschel (Cologne), Dr Wiebel/Dr Po¨mpeler (Heidelberg), Dr Bu¨sing (Osnabru¨ck), Prof. Ballmann (Bochum), PD Dr Gru¨ber (Frankfurt/Oder). Finally, we are grateful to Prof. Gratiana Steinkamp for helpful comments to a prior draft of this article.
Declaration of interest This study was supported in total by Novartis Pharma GmbH, Germany. Dr Bobis and Prof. Bewig wish to confirm that there are no known conflicts of interest associated with this publication and there has been no significant financial support for this work that could have influenced its outcome. Dr Ullrich declares to have been paid for substantial parts of the research the results of which are described in this article. He also received a professional fee for preparation of the article. Research funding and source of payment were identical, namely Novartis Pharma GmbH, Germany. The authors declare no conflicts of interests. The authors alone are responsible for the content and writing of this article.
References 1. Hodson ME, Geddes D, Bush A. Cystic fibrosis. London, United Kingdom: Arnold Publ; 2007.
Disabil Rehabil, Early Online: 1–6
2. Sawyer SM. Sexual and reproductive health. In: Hodson M, Geddes D, Bush A, eds. Cystic fibrosis. London, United Kingdom: Arnold Publ; 2007:279–90. 3. Edenborough FP. Women with cystic fibrosis and their potential for reproduction. Thorax 2001;56:649–55. 4. Wexler ID, Johannesson M, Edenborough FP, et al. Pregnancy and chronic progressive pulmonary disease. Am J Respir Crit Care Med 2007;175:300–5. 5. Edenborough FP, Borgo G, Knoop C, et al. Guidelines for the management of pregnancy in women with cystic fibrosis. J Cyst Fibros 2008;7:s2–32. 6. Hardt Hvd, Schwarz C, Ullrich G. [Adults with cystic fibrosis. It’s not just about longevity] Erwachsene mit Mukoviszidose. Es geht um mehr als die Lebensdauer. Bundesgesundheitsblatt Gesundheitsforschung Gesundheitsschutz 2012;55:558–67. 7. Go¨tz I, Go¨tz M. Reproduction and parenting. In: Bluebond-Langner M, Lask B, Angst DB, eds. Psychosocial aspects of cystic fibrosis. London, United Kingdom: Arnold; 2001:172–87. 8. Anonymous. Abstracts of the 36th European Cystic Fibrosis Conference. Lisbon, Portugal, 2013 Jun 12–15. J Cyst Fibros 2013;12:s36. 9. Frankl M, Hjelte L. Parents who have cystic fibrosis – experiences and aspects of parenthood. J Cyst Fibros 2004;3:s103 [Abstract]. 10. Erwander I. Experiences of parenthood among women and men with cystic fibrosis. 24th European Cystic Fibrosis Conference, 2001 Jun 6–9, Vienna. J Cyst Fibros (Nullnummer) Book of Abstracts 2001:371. 11. Colpaert K, Havermans T. CF and parenting. Presented at the 32nd European CF Conference, Brest, 2009 June 10–13. Slides online available. Available from: http://www.ecfs.eu/projects/internationalnurse-specialist-group-cf/presentations [last accessed 8 Aug 2013]. 12. Li A, Fulbrook P, Bell S. Journey to parenthood: experience of adults with Cystic fibrosis. Presented at the 8th Australasian CF Conference, 2009, 28 Aug to 1 Sept, Brisbane. 13. Cammidge S, Latchford G, Duff A, Etherington C. When women with CF become mothers: a qualitative study of psychosocial impact and adjustment. J Cyst Fibros 2013;12:s37 (No. WS18.5) [Abstract]. 14. Tsang A, Moriarty C, Towns S. Contraception, communication and counseling for sexuality and reproductive health in adolescents and young adults with CF. Paediatr Respir Rev 2010;11:84–9. 15. Simcox AM, Duff AJ, Morton AM, et al. Decision making about reproduction and pregnancy by women with cystic fibrosis. Br J Hosp Med 2009;70:639–43. 16. Domsch H, Lohaus A. [Parent Stress Questionnaire] Elternstressfragebogen (ESF): Manual. Go¨ttingen: Hogrefe, 2010. 17. Abidin RR. Parenting stress index. Charlottesville (VA): Pediatric Psychology Press; 1990. 18. Feizi A, Najmi B, Salesi A, et al. Parenting stress among mothers of children with different physical, mental, and psychological problems. J Res Med Sci 2014;19:145–52. 19. Epifanio MS, Genna V, Vitello MG, et al. Parenting stress and impact of illness in parents of children with coeliac disease. Pediatr Rep 2013;5:81–5. 20. Bluebond-Langner M, Lask B, Angst DBH. Psychosocial aspects of cystic fibrosis. London, United Kingdom: Arnold; 2001. 21. Jessup M, Parkinson C. ‘All at the sea’: the experience of living with cystic fibrosis. Qual Health Res 2010;20:352–64. 22. Simcox AM, Hewison J, Duff AJ, et al. Decision-making about pregnancy for women with cystic fibrosis. Br J Health Psychol 2009; 14:323–42. 23. Gerhardt CA, Vannatta K, McKellop JM, et al. Comparing parental distress, family functioning, and the role of social support for caregivers with and without a child with juvenile rheumatoid arthritis. J Pediatr Psychol 2003;28:5–15.
