Anxiety and depression in parents of children born with esophageal atresia: An international online survey study

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2021

Anxiety and depression in parents of children born with esophageal atresia:

An international online survey study

Wallace, Vuokko

Elsevier BV

Tieteelliset aikakauslehtiartikkelit

© 2021 The Authors

CC BY-NC-ND https://creativecommons.org/licenses/by-nc-nd/4.0/

http://dx.doi.org/10.1016/j.pedn.2021.02.016

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Anxiety and Depression in Parents of Children Born with Esophageal Atresia: An International Online Survey Study

Vuokko Wallace, DClinPsych

^a,b,

⁎ , Kirsi Honkalampi, PhD

^b

, Elizabeth Sheils, MSc

^a

aDepartment of Psychology, University of Bath, UK

bDepartment of Psychology, University of Eastern Finland, Finland

a b s t r a c t a r t i c l e i n f o

Article history:

Received 21 September 2020 Revised 14 February 2021 Accepted 14 February 2021

Keywords:

Esophageal atresia Parental anxiety Parental depression Feeding difﬁculties Caring stress Online survey

Purpose:Caring for a chronically ill child can be stressful and requires additional care from parents. Parental mental health and stress may impact both parental and child wellbeing, leading to maladaptive parenting practices, and interference with illness management. The aim of the study was to explore the levels of anxiety and depression in parents of children born with esophageal atresia (EA) and to investigate whether parental anxiety and depression were associated with child's medical characteristics or with parental factors.

Design and methods:An international online survey was developed and disseminated to parents of children born with EA, aged 0–12 years, via EA patient charity social network sites. A one-way between subjects ANOVA and post hoc statistical analyses were used to examine differences in mean scores of parental anxiety and depression between sub-groups that described child's medical characteristics and parental characteristics.

Results:A total of 240 parents completed the survey from 17 different countries. Of these, nearly 70% self- reported raised levels of anxiety, whilst 38% self-reported raised levels of depression. Statistically signiﬁcant differences in mean scores of parental anxiety and depression were found between sub-groups that described the child's feeding problems, parental age, and perceived support for caring, caring stress and money matters.

Conclusion:Child's feeding problems related to esophageal atresia, and parental factors, such as younger age, perceived lack of support for caring, caring stress, and money worries, may contribute to the increased levels of parental anxiety and depression in parents of children born with EA.

⁎ Corresponding author at: University of Bath, Claverton Down, 10 West, Bath BA2 7AY, UK.

E-mail address:vw273@bath.ac.uk(V. Wallace).

Introduction

Esophageal Atresia (EA) is a rare congenital malformation of the esophagus, which leads to an inability to swallow and requires surgical repair in infancy (Spitz, 2007). It commonly occurs together with tracheoesophageal atresia (TEF), which is an abnormal connection between the trachea and esophagus (Lee & Meeker, 2018). Other complex- ities include a long gap EA presentation, which poses additional surgical challenges and a more complicated post-surgery management (Séguier- Lipszyc et al., 2005), and VACTERL association, in which at least two or three of the associated anomalies (vertebral defects, anal atresia, cardiac defects, tracheoesophagealﬁstula, renal anomalies, and limb abnormalities) are present, adding to the overall complexity (Raam et al., 2011).

Medical advances have increased survival rates of babies born with EA to 95% in the Western world (Sfeir et al., 2013). Follow-up studies have found that most children born with EA achieve full oral feeds fol- lowing the surgical repair, however, abnormal esophageal function and associated morbidity, such as esophageal stricture, gastro-

esophageal reﬂux, and related respiratory problems (e.g., recurrent respiratory infections and tracheomalacia) remain common (Donoso et al., 2016;Koivusalo et al., 2013;Porcaro et al., 2017). Research has shown that feeding and swallowing disorders are common complications of surgical esophageal repair (Conforti et al., 2015) and that these problems often prevail into adulthood, impacting the quality of life in adults with EA (Gibreel et al., 2017). It has been suggested that feeding difﬁculties can lead to prolonged and stressful mealtimes, and a higher risk of aspiration with potential pulmonary consequences (Ramsay & Birnbaum, 2013), as well as to growth retardation in babies and children with EA (Puntis et al., 1990).

Caring for a chronically ill child requires additional care from parents, including daily medical care, attending hospital appointments andﬁnancial management. These additional responsibilities can impact parents' employment, social life, and health-related quality of life (HRQoL) (Grootenhuis & Bronner, 2009). The needs of carers, and the relational and social aspects of caring are often unrecognized (ten Hoopen et al., 2020), even though it is known that stressful life events, ongoing stresses, and psychosocial factors increase the likelihood of depression (e.g.,Brown & Harris, 1978;Harris & Craig, 2006).

Research has demonstrated that parents who care for a chronically ill child are more likely to report poorer mental health and more

https://doi.org/10.1016/j.pedn.2021.02.016

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parenting stress compared to parents of unaffected children (Cohn et al., 2020;Cousino & Hazen, 2013). Previous literature reviews investigating parental anxiety and depression in parents of children with chronic health conditions, have found elevated levels of both anxiety (16%) (Pinquart, 2019a) and depression (20.9%) (Pinquart, 2019b). Parental mental health is important because it may impact both parental and child wellbeing. Parental anxiety and depression have been found to predict child mental health and development (e.g. Vänskä et al., 2017). Poor parental mental health and parenting stress can also lead to parents using maladaptive parenting practices and interfere with ef- fective illness management (Pinquart, 2018;Streisand et al., 2008).

Previous research has attempted to explore parental mental health in families affected by EA, mainly in single-center, quality-of-life studies.

Research has suggested that child related factors, such as the child's age or illness related factors, might be related to parental mental health.

Faugli et al. (2009)found that parents experienced most challenges related to their child's EA during the infancy and toddlerhood/preschool years. Similarly,Witt et al. (2019)found that the parents of younger children born with EA (aged 2–7 years) reported lower mental health compared to the parents of older children and adolescents. In another study, parents of older children and adolescents (aged 8–17) born with VACTERL association self-reported anxiety and depression at levels that were comparable to a non-clinical sample (Kassa et al., 2020).

Theseﬁndings would suggest that parental mental health might im- prove over time, whilst the child born with EA grows and develops through childhood into adolescence. The severity of the child's EA (Witt et al., 2019); the presence of associated anomalies (Rozensztrauch et al., 2020); lung infections (Faugli et al., 2009); and feeding problems (Dellenmark-Blom et al., 2015;Rozensztrauch et al., 2020) have been indicated as the child's illness related factors impacting parental mental health and family functioning in families affected by EA.

Further,Dellenmark-Blom et al. (2016)has suggested that feeding difﬁ- culties in children and adolescents with EA may lead to social and emotional strains and impact child's relationships and interaction with other people.

A literature review focusing on chronically ill children found that anxiety, depression, and poorer psychological adjustment in parents were not associated with child related medical characteristics but with parents' younger age, social problems, problems in daily life, and parenting stress instead (van Oers et al., 2014). Mothers have also been found to report lower mental health compared to fathers (Witt et al., 2019). In a study on posttraumatic stress in parents of children with EA, it was found that the severity of the parents' posttraumatic stress symptoms was related to the parents' subjective evaluation of their child's health status and quality of life rather than being associated with the objective severity of their child's EA (Le Gouëz et al., 2016).

To date, the mental health of parents of children born with EA has not been investigated using validated anxiety and depression instru- ments in an international sample. Given the lack of research on the mental health of parents of children born with EA, the aim of the study was to explore the levels of anxiety and depression in parents of children born with esophageal atresia (EA). Given the unique challenges related to feeding and swallowing problems in EA (Conforti et al., 2015), this study also aimed to investigate whether parental anxiety and depression were associated with child's medical characteristics (child's age, EA complexity, and associated feeding problems reported by parents) or with parental factors (parental age, perceived support for caring, parental stress and money worries).

Design and methods Design

The current study employed a cross-sectional, explorative research design and purposive sampling methods, recruiting participants through an international online survey.

Ethical considerations

Ethical approval for this study protocol was obtained from the Uni- versity of Bath Psychology Research Ethics Committee. The online survey contained an introductory page with details of the study, ethical approval, the name and contact details of the researcher, and a consent form to complete. To take part in the study, participants needed access to a computer, tablet, or a smart phone. Participants completed the survey anonymously, without providing any identiﬁable information. After completing the survey, the participants were presented with a page with debrief information, including details of organizations that provide mental health support, if needed.

Participants

A total of 310 parents of children born with EA, aged 0–12 years, took part in an online survey during the data collection period. From these, 240 submitted surveys were used in the statistical analysis as they were fully completed, they met the inclusion criteria, and the respondents had consented to taking part in the study. Whilst most survey submissions came from the UK (176 submissions, 73.3%), surveys were submitted from 17 countries. These included Australia and New Zealand (24 submissions, 10.0%), US and Canada (19 submissions, 7.9%), countries of the EU (16 submissions, 6.7%), Africa (2 submissions, 0.8%) and Asia (1 submission, 0.4%).

Inclusion and exclusion criteria

The inclusion criteria were explained in the study advertisement and checked in the beginning of the survey via background questions. Par- ticipants needed to be parents of children born with EA (aged 0–12 years). The large age range allowed the current study to investigate parental mental health throughout childhood years and to examine whether there were differences in mean scores of parents' anxiety and depression between different child age groups. Both mothers and fathers were invited to take part in the study and both parents from the same family were able tofill in the survey. The participants were re- quired to have access to a computer, and to understand and be able to write English sufficiently tofill in an online survey. The participants were asked to submit their responses at the end of the survey for them to be included in the study. If the survey responses were not submitted, they were excluded from the sample. There were no questions related to the current medical circumstances of the family, e.g., whether the child was currently at home or in hospital.

Development and dissemination of the online survey

The survey was developed using Qualtrics software and it was disseminated via a UK based patient charity for EA. Before disseminating the survey, the usability of the survey was piloted by university staff to confirm that it could be easily accessed and completed online using the survey link. The survey included a question asking participants to provide feedback on the study, and in response to the feedback, minor changes were made to increase the usability of the survey after the first 24 responses. The changes included adding an option to save a half-filled survey to return to it at later stage. Thefirst 24 responses were included in the sample.

The participants were recruited through EA patient social network sites, such as private Facebook groups for families affected by EA. The study advertisement with a URL link to the online survey was posted 8 times on private social network sites for EA patients over a 4-month period. The advertisement was also posted on the patient charity's website and emailed to the members of the charity. Additionally, the study advertisement was forwarded by the collaborating US, Canada, Australia, and New Zealand EA patient groups. Once a parent completed the survey, theﬁle was automatically entered into a Qualtrics database.

V. Wallace, K. Honkalampi and E. Sheils Journal of Pediatric Nursing 60 (2021) 77–82

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After the survey closed, the complete set ofﬁles were exported into a statistical software package (IBM SPSS 26) for data analysis.

Background questionnaire

To explore the role of parental age and child related characteristics on mean scores of parents' anxiety and depression, additional background questions were incorporated into the survey. The participants' responses to the background questions deﬁned the parent age category and the child categories for statistical analysis. It is important to note that the current study focused on the mental health of the parents of children born with EA, and that child related information was used to describe the different contexts for parents. No children took part in the study, and there were no child-parent pairs used in the analysis.

The participants were asked to state their own age and the age of their child born with EA. The participants were also asked to state, using a multiple-choice question, whether their child had 1) esophageal atresia/tracheoesophageal atresia (EA/TEF), or 2) whether they had a long gap EA/TEF, or EA/TEF with any additional associated conditions (e.g., VACTERL). This question was used to deﬁne EA complexity category, as a long gap EA presentation and EA with associated conditions increase the surgical challenges, related complications, and overall complexity (Raam et al., 2011;Séguier-Lipszyc et al., 2005). Referral to a speech and language therapist (SALT) is recommended for children's feeding difﬁculties in EA (IJsselstijn et al., 2016). To detect the presence of feeding related problems, parents were asked to state whether their child born with EA had had a speech and language therapy assessment or treatment for swallowing or eating related problems, or not. Please seeTable 1for the child related categories.

Measures

To investigate the levels of anxiety and depression in parents of children born with EA, the questions from the Hospital Anxiety and Depres- sion Scale (HADS) (Zigmond & Snaith, 1983) were incorporated into the survey. The HADS is a 14-item self-report measure for anxiety and depression, and it provides four categories of severity for the subscales of depression and anxiety: normal (0–7), mild (8–10), moderate (11–14) and severe (15–21). The HADS has been shown to be a reliable and valid self-report measure for anxiety and depression (Bjelland et al., 2002), and it has demonstrated high concurrent validity with the Beck Depression Inventory scale (BDI;Beck et al., 1961;Hansson et al., 2009).

To deﬁne parental characteristics related to care-related factors such as support for caring, caring stress and money matters, the questions from the Adult Carer Quality of Life Questionnaire (AC-QoL;Elwick et al., 2010) were incorporated into the survey. The AC-QoL is a 40- item self-report measure for adult carer quality of life in eight separate domains. It provides three categories of care-related quality of life scores (low 0–5, mid-range 6–10, and high 11–15) for eight subscales (Elwick et al., 2010). The parents' scores (low, mid-range or high QoL) on the‘Support for caring’,‘Caring stress’and‘Money matters’subscales were used in the analysis as the parental care-related categories (please seeTable 1for the parent categories). The three sub-scales used in the current study measure the extent of perceived emotional, practical, and professional support; the extent of mental and physical stress from caring; and how carers feel about theirﬁnancial situation.

Lower scores on AC-QoL indicate that the carer might need more support for caring (Elwick et al., 2010). The AC-QoL has been shown to be a reliable and valid measure to assess caregivers' perceived challenges and resources related to caring. It has also demonstrated satisfactory convergent and discriminant validity with measures of burden, resil- ience, satisfaction, physical and mental health (Joseph et al., 2012;

Negri et al., 2019).

Statistical analysis

Descriptive statistics were used to describe the levels of depression and anxiety in the survey population of parents of children born with EA (aged 0–12 years). To explore the role of the child and parent characteristics on mean scores of parents' anxiety and depression, seven categories were formed. These categories were formed based on the parents' answers in the background questionnaire (child age, EA complexity, EA related feeding problems, parent age, and the parents' scores on the Support for caring, Caring stress, and Money matters sub-scales of the AC-QoL measure). Please seeTable 1for the child and parent related categories.

A series of One-way ANOVA and Fisher's Least Signiﬁcant Difference (LSD) post-hoc analyses were conducted using SPSS 26, to explore differences in mean scores of parents' anxiety and depression between the sub-groups in each of the child and parent categories (1) Child age, 2) EA complexity, 3) SALT needs for swallowing, 4) Parental age, 5) Support for caring, 6) Caring stress, and 7) Money matters). The effect sizes were calculated by Partial Eta Squared on SPSS and they were de- ﬁned as small (ηp²< 0.06), medium (ηp²= 0.06–0.14) and large (ηp²> 0.14). Two respondent's HADS depression scores were excluded from statistical analysis, as they were outliers.

Results

Child characteristics

Of the respondents' children born with EA, a total of 74 (30.8%) were under 2 years old, 132 (55%) were aged between 2 and 7 years and 34 Table 1

Categories for statistical analysis.

Parent sub-groups

n (%) Notes

Child related categories*

Child age 1 0–2 years 74 (31) **

2 2–7 years 132 (55) 3 8–12 years 34 (14) EA

complexity

1 EA/TEF only 130 (54) Complex EA deﬁned as a long gap, VACTERL, and/or other associated conditions**

2 Complex EA 110 (46) SALT needs

for swallowing

1 NO 144 (60) SALT assessment or treatment for swallowing and eating related issues?

**

2 YES 96 (40)

Parent related categories

Parental age 1 18–29 years 35 (15) **

2 30–39 years 151 (63) 3 40–45+

years

54 (23) Support for

Caring

1 Low QoL 92 (38) ***

2 Mid-range QoL

100 (42)

3 High QoL 48 (20)

Caring stress 1 Low QoL 26 (11) ***

2 Mid-range QoL

128 (53)

3 High QoL 86 (36)

Money matters

1 Low QoL 88 (37) ***

2 Mid-range QoL

103 (43)

3 High QoL 49 (20)

EA = Esophageal Atresia.

TEF = Tracheoesophageal atresia.

VACTERL = vertebral defects, anal atresia, cardiac defects, tracheoesophagealﬁstula, renal anomalies, and limb abnormalities.

SALT = Speech and language therapy.

AC-QoL = Adult Carer Quality of Life measure (Subscales rating: Low QoL (0–5 points), mid-range QoL (6–10 points), high QoL (11+ points)).

* Child related information demonstrates the child related factors for parents.

** Based on parental response on the background questionnaire.

*** Based on parental score on AC-QoL sub-scale.

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(14.29%) were aged between 8 and 12 years. A total of 130 (54.2%) respondents indicated that their child had an esophageal atresia and/or tracheoesophageal atresia (EA/TEF), whilst 110 (45.8%) respondents indicated that their child had a more complex type of EA (including a long gap presentation, VACTERL association, and other associated conditions). A total of 96 (40%) of the respondents reported that their child had had a speech and language therapy assessment or treatment for a swallowing or eating related issues, whilst 144 (60%) did not. Please seeTable 1for breakdown of the child characteristics.

Parental characteristics

Of the 240 respondents, 224 were mothers (93.3%), 15 (6.3%) were fathers, and one (0.4%) was a carer. A total of 35 (14.6%) of the parents were aged between 18 and 29 years, 151 (62.9%) were aged between 30 and 39 years, and 54 (22.5%) were over 40 years of age. In terms of parents' care-related factors, a total of 92 (38.3%) parents reported low quality of life on the Support for Caring subscale of the AC-QoL, whereas 100 (41.7%) parents reported mid-range QoL and 48 (20.0%) parents reported high QoL. On the Caring Stress subscale of AC-QoL, 26 (10.8%) parents reported low QoL, 128 (53.3%) mid-range QoL and 86 (35.8%) high QoL. In the Money Matters subscale of AC-QoL, 88 (36.7%) parents reported low QoL, 103 (42.9%) parents reported mid-range QoL and 49 (20.4%) parents reported high QoL. Please seeTable 1for breakdown of the parent characteristics.

Parental anxiety and depression

Descriptive statistics showed that 167 (69.6%) parents self-reported raised anxiety levels, with a mean anxiety score of 9.91 (SD= 4.34) on the HADS measure. From these, 62 parents (25.8%) self-reported mild anxiety, 68 (28.3%) moderate anxiety and 37 (15.4%) severe anxiety. A total of 92 (38.3%) parents self-reported raised levels of depression, with a mean depression score of 6.54 (SD= 4.10), on the HADS measure. From these 51 parents (21.3%) self-reported mild depression, 33 (13.8%) moderate depression and 8 (3.3.%) severe depression. Please seeTable 2for a summary of parental anxiety and depression scores.

Parental anxiety and depression and associated child characteristics

A series of one-way between subjects ANOVA tests were conducted to compare the effects of child's age, EA complexity, and SALT needs for swallowing on parental anxiety and depression. It was found that there were no statistically signiﬁcant differences in the parents' HADS scores between the sub-groups in the child's age category for anxiety F(2,237) = 0.09,p= .915, or depressionF(2,235) = 0.57,p= .569.

Similarly, there were no statistically signiﬁcant differences in the parents' HADS scores between the sub-groups in the child's EA complexity category for anxiety F(2,238) = 1.23, p= .269, or depression F (1,236) = 2.23,p= .137. Analysis of variance showed a main effect of SALT needs for swallowing on both parental anxietyF(1,238) = 7.47 p= .007,ηp²= 0.03, and parental depressionF(1,236) = 5.67,p=

.018,ηp²= 0.02, indicating a small effect size. Please see breakdown of parents' HADS scores for anxiety and depression in each child category inTable 3.

Parental anxiety and depression and associated parent characteristics

A one-way between subjects ANOVA was conducted to compare the effects of parental age on parental anxiety and depression. Analysis of variance showed a main effect of parental age on both parental anxiety F(2,237) = 6.96, p= .001, ηp² = 0.06, and parental depression F(2,235) = 7.92,p< .001,ηp²= 0.06, indicating a medium effect size.

Post hoc analyses using Fisher's Least Signiﬁcant Difference (LSD) test indicated that parents' anxiety and depression scores were higher for younger parents (aged 18–29 years) compared to 30–39 years olds (anxietyp< .001, depressionp< .001) and to over 40-year olds (anxietyp= .009, depressionp= .005), but there were no signiﬁcant differences between the older age groups (parents aged 30–39 years and over 40 years) for either anxiety (p= .408), or depression (p= .420).

A series of one-way between subjects ANOVA tests were conducted to compare the effects of parental care-related categories (Support for caring, Caring stress and Money matters) on parental anxiety and depression. Analysis of variance showed a main effect of Support for caring, Caring stress, and Money matters categories on both parental anxiety [Support for caring:F(2,237) = 23.22,p< .001,ηp²= 0.16; Car- ing stress:F(2,237) = 36.69,p< .001,ηp²= 0.24; Money matters:

F(2,237) = 8.85,p< .001,ηp²= 0.07] and parental depression [Support for caring:F(2,235) = 21.23,p< .001,ηp²= 0.15; Caring stress:

F(2,235) = 48.33,p< .001,ηp²= 0.29; Money matters:F(2,235) =

Table 2

Parental anxiety and depression scores.

Categories Parental HADS

anxiety scores

Parental HADS depression scores

n (%) n (%)

Normal (0–7) 73 (30) 148 (62)

Mild (8–10) 62 (26) 51 (21)

Moderate (11–14) 68 (28) 33 (14)

Severe (15–21) 37 (15) 8 (3)

Total 240 240

HADS = Hospital Anxiety and Depression Scale: normal (0–7), mild (8–10), moderate (11–14) and severe (15–21).

Table 3

HADS anxiety and depression scores in parents of children born with EA.

Categories Parental anxiety HADS

scores

Parental depression HADS scores

Mean SD pvalue Mean SD pvalue

Child categories

Child age p = .915 p = .569

Under 2 years 9.78 4.18 6.69 4.66

2–7 years 10.02 4.65 6.65 3.83

8–12 years 9.77 3.42 5.77 3.85

EA complexity p = .269 p = .137

EA/TEF only 9.62 4.17 6.09 3.81

Complex EA 10.25 4.52 7.07 4.38

SALT needs for swallowing p = .007* p = .018*

No 9.29 4.12 5.94 3.78

Yes 10.83 4.51 7.43 4.42

Parent categories

Parental age p < .001*** p < .001***

18–29 years 12.31 3.89 8.77 4.09

30–39 years 9.35 4.30 5.99 4.09

40–45+ years 9.91 4.26 6.63 3.70

Support for caring p < .001*** p < .001***

Low QoL 11.95 3.80 8.54 4.04

Mid-range QoL 9.26 4.01 5.72 3.49

High QoL 7.35 4.34 4.40 3.79

Caring Stress p < .001*** p < .001***

Low QoL 13.58 3.92 11.77 3.65

Mid-range QoL 10.91 3.70 7.13 3.67

High QoL 7.30 3.93 4.07 2.87

Money Matters p < 001*** p < .001***

Low QoL 11.35 3.93 8.46 4.11

Mid-range QoL 9.34 4.40 5.53 3.58

High QoL 8.51 4.27 5.20 3.91

Total HADS scores 9.91 4.34 6.54 4.10

EA = Esophageal Atresia.

TEF = Tracheoesophageal atresia.

VACTERL = vertebral defects, anal atresia, cardiac defects, tracheoesophagealﬁstula, renal anomalies, and limb abnormalities.

SALT = Speech and language therapy.

HADS= Hospital Anxiety and Depression Scale: normal (0–7),mild (8–10),moderate (11–14)andsevere (15–21).

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15.36,p< .001,ηp²= 0.12], indicating medium or large effect sizes. Post hoc analyses using LSD test indicated that, in all three parental care- related categories, parents' anxiety and depression scores were higher in low QoL groups compared to mid-range QoL groups [Support for caring (anxietyp< .001, depressionp< .001); Caring stress (anxietyp= .001, depressionp< .001); Money matters (anxietyp= .001, depressionp< .001)], and compared to high QoL groups [Support for caring (anxiety p < .001, depression p < .001); Caring stress (anxiety p< .001, depressionp< .001); Money matters (anxietyp< .001, depressionp< .001)]. Please see breakdown of parents' HADS scores for anxiety and depression in each parent category inTable 3.

Discussion

To our knowledge, this was theﬁrst study to speciﬁcally look at parental anxiety and depression in parents of children born with EA, using a validated questionnaire. Our aim was to explore the levels of anxiety and depression in parents of children born with EA (aged 0–12 years).

We also aimed to investigate whether parental anxiety and depression were associated with child's medical characteristics (child age, EA complexity and associated feeding problems reported by parents) or with parental factors (age, perceived support for caring, parental stress and money worries).

The study found that the levels of both anxiety and depression in parents of children born with EA were higher than those previously reported in parents of children with chronically ill children. In previous review studies, 16% of parents were found to have elevated levels of anxiety and 20.9% of depression (Pinquart, 2019a, 2019b). In the current study, a total of 69.6% of parents self-reported elevated levels of anxiety, whilst 38.3% self-reported elevated levels of depression (HADS score 8 or above). Higher levels of anxiety and depression found in the current study might be partly explained by the overrepresentation of mothers in the study as previous research has found mothers to report lower mental health compared to fathers (Witt et al., 2019). It is important to note that even though many people in the sample reported moderate to severe anxiety (43.7%) and depression (27.1%), the mean scores for the total sample were either within normal (depression) or mild (anxiety) range.

Previous researchﬁndings on the impact of child characteristics on parental mental health differ between a literature review on chronically ill children and studies on EA. A literature review byvan Oers et al.

(2014)found that child's medical characteristics, such as child's age or the severity of illness, were not related to parental anxiety or depression in parents of chronically ill children. However, studies speciﬁcally on EA have suggested that child's age (Faugli et al., 2009;Kassa et al., 2020;

Witt et al., 2019), EA severity (Witt et al., 2019), associated anomalies (Rozensztrauch et al., 2020), and feeding problems (Dellenmark-Blom et al., 2015;Rozensztrauch et al., 2020) might have an impact on parental mental health or family functioning. The current study partly sup- ported thefindings byvan Oers et al. (2014), as statistically significant differences were not found in the levels of parental anxiety or depression between child's age, or EA complexity sub-groups. Even though parents' anxiety and depression scores were higher in the complex EA sub-group compared to EA/TEF only sub-group, these differences were not statistically significant.

Previous research has demonstrated that EA related eating and drinking problems can impact the quality of life of children and adolescents born with EA (Dellenmark-Blom et al., 2015), and impact the family functioning, particularly in the emotional domain (Rozensztrauch et al., 2020). The current study found that there was a statistically significant difference, even though with only a small effect size, in the levels of parental anxiety and depression between parents of children with and without feeding problems. This preliminaryﬁnding supports previous research and provides further insight into the unique challenge of EA related feeding problems and their impact on parental mental health.

In line with previous research (van Oers et al., 2014), this study found that younger parents self-reported higher levels of both anxiety and depression compared to parents aged 30 and over, suggesting that younger parents might be particularly vulnerable for poorer mental health outcomes when caring for a child born with EA. Similarly, this study found that perceived support for caring, caring stress and money matters were all associated with parental anxiety and depression, as statistically significant differences were found in mean scores of parents' anxiety and depression between sub-groups in all care- related categories. Thisfinding suggests that if parents of children born with EA report low care-related quality of life, they are also more likely to experience higher levels of anxiety and depression compared to those parents who report mid-range or high care-related quality of life. Thisfinding supports previous research in parents with other chronic conditions, as anxiety, depression and poorer psychological adjustment have been found to be associated with parents' social problems, problems in daily life and parenting stress (van Oers et al., 2014).

Practice implications

This study utilized online EA patient groups and online survey methods to reach parents in a rare disease population and achieved a relatively large and international sample. Even though online data collection in social network sites might create a sampling bias, these methods provide the potential to reach groups with unusual or rare health conditions (Acher et al., 2016).

Ourfindings can help health professionals to identify those parents who might be at increased risk of developing poorer mental health outcomes: younger parents; parents with less support for caring; parents who feel more stressed as a result of caring; and parents withfinancial worries. A uniquefinding related to EA, was a difference found in the levels of parental anxiety and depression between parents of children with and without EA related feeding problems. This preliminaryfinding calls for further research in this area, taking a multidisciplinary perspec- tive. It also calls for a recommendation for clinicians to enquire about child's feeding difficulties in detail, including their impact on parental wellbeing, regardless of the child's overall EA complexity.

Limitations

One of the common challenges for online survey designs is uneven gender distribution of participants recruited through social media net- works. Previous research has suggested that females might be particularly overrepresented in mental health related studies (Thornton et al., 2016), which could partly explain the overrepresentation of mothers in the current study. Due to the small number of fathers taking part, there was no statistical analysis of differences in parental anxiety and depression between mothers and fathers. Future studies might explore different ways of improving recruitment of fathers or recruiting speciﬁc child-parent pairs.

Due to the constraints of the survey design, a non-standardized background questionnaire was used to enquire about children's feeding problems and EA complexity. Even though this provided a useful method for an explorative study, future research might add a standardized questionnaire for feeding and swallowing related problems. Inves- tigating this issue further might be particularly important for developing emotional and practical support for parents caring for their child with EA. One should also note that EA related studies have used different methods of defining EA severity or complexity (e.g.,Dellenmark-Blom et al., 2015;Rozensztrauch et al., 2020). Varied findings in this area call for further research in the specific impact of illness-related factors on parental mental health.

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Conclusion

In the current study, parents of children born with EA self-reported higher levels of anxiety and depression compared to previously reported levels in parents of children with chronic health conditions. It was found that parental anxiety and depression in parents of children born with EA, might be associated more with parental characteristics, such as parental age and care-related factors, rather than child characteristics, such as child's age or illness complexity. Even with the limitations related to the online survey methods, this study adds to the limited research on mental health in parents of children born with EA.

Declaration of Competing Interest

We have no known conﬂict of interest to disclose.

Acknowledgments

We would like to thank all the parents taking part in the study, and the Tracheo-Oesophageal Fistula Support (TOFS), Registered Charity in England and Wales (327735), for theirﬁnancial support and invaluable help with participant recruitment. We would like to acknowledge the patient-led approach used in this study, including the main author (VW) being born with EA. We would also like to thank Dr. James Greg- ory for his valuable and helpful comments on an earlier draft.

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