Quality of life in intermittent exotropia: child and parent concerns

Abstract

Objective

To identify specific health related quality of life (HRQOL) concerns for children with intermittent exotropia (IXT) and their parents.

Methods

24 children (5–17 years) with IXT and one parent for each child were recruited. Individual interviews, with the child and then the parent, were audio taped and transcribed. Transcripts were reviewed, phrases regarding effects of IXT on HRQOL recorded, and specific topic areas identified. Topic frequency was analyzed to determine: child’s perception of their own HRQOL, parent’s perception of child’s HRQOL, and parent’s own HRQOL.

Results

Child interviews generated 18 topics. Worry [10 (42%) of 24] was most frequently mentioned. Parent interviews generated 22 topics regarding their child’s HRQOL. The most frequently mentioned topic was Comments from others [15 (63%) of 24]. Regarding the parent’s own HRQOL, 14 topics were identified; the most frequently mentioned was Worry regarding possible surgery [15 (63%) of 24].

Conclusions

Multiple individual interviews revealed specific HRQOL concerns such as Worry in children with IXT and their parents. We will use concerns identified to develop condition-specific HRQOL instruments for IXT.

Introduction

Intervention in childhood intermittent exotropia (IXT) aims to preserve binocular function^1–3 and to address any social stigma associated with manifest strabismus.^{4, 5} Nevertheless, the natural history of IXT^{1, 6–8} and indications for intervention are poorly defined, partly due to a lack of robust outcome measures.^{6, 8–10} Formal assessment of health-related quality of life (HRQOL) may therefore be particularly helpful in clarifying indications for intervention as well as potential benefits from treatment. Others have compared HRQOL between patients with IXT and control subjects using generic questionnaires¹¹ (Powell CJ et al Quality of Life in Intermittent Exotropia. Poster presentation, AAPOS 2007), but not with condition-specific instruments. A condition-specific instrument is designed to better capture HRQOL effects specific to a given condition, and therefore is more likely to be sensitive to changes over time and changes with treatment.^{12, 13} A condition-specific HRQOL instrument may be warranted for IXT because it is a common condition¹⁴ and differs from other forms of childhood strabismus in its intermittency and often normal binocular function. As a first step towards developing condition-specific HRQOL questionnaires, we interviewed children with IXT and their parents to identify specific HRQOL concerns. We also aimed to compare and contrast HRQOL concerns perceived by the parent and child, since such concerns may influence management of this condition.

Patients and Methods

Institutional Review Board approval was obtained and the parent of each child gave informed consent before participating. Children aged 8 years or older provided informed assent. All procedures and data collection were conducted in a manner compliant with the Health Insurance Portability and Accountability Act.

Patients

Twenty-four children (age range 5 to 17, median 10.5 years) with IXT who had not undergone previous surgery were recruited. Subjects were identified in outpatient clinics and by searching clinical databases. Seventeen subjects were female and for 21 subjects, race was self-reported as ‘White.’ All children had divergence excess or basic type exotropia: no child had a near angle ≥10 pd greater than distance (convergence insufficiency type exotropia). Patients with co-existing ocular pathology were excluded. Median angle of deviation by alternating prism cover test at distance was 20 prism diopters (pd) (range 10 pd to 40 pd). ‘Control’ and ‘stereoacuity’ are not reported since we have found both control and stereoacuity to vary throughout the day in children with IXT,^{15, 16} and we did not make multiple measurements in this particular cohort. For each child, one accompanying parent or legal guardian was also recruited. Patients and parents were required to be comfortably conversant in the English language in order to be included.

Methods

Individual interviews, first with the child and then with the parent, were conducted and audio taped. All interviews were conducted by a research orthoptist (SRH) and were performed separately from the clinical exam, some before the exam and some after. For the child, a series of 10 open-ended questions (Table 1) were used as the basis for the interview, and discussion was encouraged for each question. Each child was asked each question. The duration of interviews ranged from approximately two to five minutes depending on the individual. The child was given the choice to be interviewed with or without the parent present. For the parent, a series of 12 open-ended questions (Table 2) were used as the basis for the interview, and discussion was encouraged for each question. Each parent was asked each question. Both parent and child questions were selected after informal discussion within our research group, but were chosen to be general and open-ended to capture as many concerns as possible. The duration of interviews ranged from approximately three to 15 minutes, depending on the individual. The parent was given the choice of being interviewed alone (if possible) or with the child present.

Table 1.

Interview questions for children with intermittent exotropia.

1. What bothers you about your eyes?

2. How are your eyes different from other children’s eyes?

3. What things are hard to do because of your eyes?

4. Tell me how your eyes feel?

5. What do other children say to you about your eyes?

6. What do grown ups say to you about your eyes?

7. What do you wish you could change about your eyes?

8. How much do you worry about your eyes?

9. What do other people think about you?

10. Is there anything else about your eyes that bothers you or that you want to tell me?

Table 2.

Interview questions for parents of children with intermittent exotropia.

1. What bothers you most about your child’s eyes?

2. How much do you notice your child’s eye wandering out or your child closing one eye?

3. What activities do you associate with your child’s eye wandering out? What makes it worse or better?

4. What do you do or not do with your child because they have intermittent exotropia?

5. How does your child’s intermittent exotropia affect you and your family?

6. How do other people react when they notice your child has intermittent exotropia? How does that make you feel?

7. In what ways does intermittent exotropia affect your child?

8. In what ways does intermittent exotropia affect your child’s ability to interact with other children or adults?

9. How would life change for you and your child if the intermittent exotropia was corrected?

10. What concerns for the future do you have regarding your child’s intermittent exotropia?

11. What are the main issues or concerns for you regarding the treatment or management of your child’s intermittent exotropia?

12. Can you describe any other ways intermittent exotropia affects you or your child that we haven’t covered?

Child and parent interviews were transcribed and the transcripts were reviewed independently by one clinician (WEA) and two non-clinicians (DAL and PAK). Each investigator independently recorded any phrases pertaining to the effects of IXT on quality of life, resulting in three lists of phrases for each child and three lists of phrases for each parent. For each child and for each parent the three lists of phrases were combined, duplicate phrases were removed, and responses reviewed to identify themes or topic areas. Any phrases describing pure symptoms but not any effect of symptoms on HRQOL were removed. If the phrase indicated the child or parent was troubled by the symptom, then the phrase was retained. For example the phrase ‘my eye wanders’ was removed whereas ‘I’m bothered by my eyes going in and out’ was retained.

Once data collection was completed, two investigators (DAL and SRH) reviewed the final list of phrases and topics for all children and all parents to agree on the assignment of each phrase to a topic. If a response could be assigned to more than one topic area, the allocation was agreed by discussion. We considered a topic to have been mentioned whether it occurred once or multiple times during an interview, and so topic frequency was determined by analyzing whether a topic was mentioned at least once by individual children and parents. Overall topic frequency was calculated for the children’s responses. For the parents, phrases and topics were analyzed according to whether they reflected the parent’s perception of their child’s quality of life, or whether they reflected the parent’s own quality of life concerns. Topics relating to the child’s HRQOL that were common to both the parent group and the child group were analyzed to determine whether or not they were mentioned by parent-child pairs (i.e., within the same family).

Results

Child interviews

For the 24 children with IXT, a total of 106 phrases were extracted, and 18 topic areas were apparent. The number of topics mentioned per child ranged from zero (3 patients) to 11 (median 3). The overall topic frequency is shown in Table 3. The most frequently mentioned topic was Worry [10 (42%) of 24] including phrases such as ‘I worry a lot about my eyes,’ ‘I worry about it getting worse,’ and ‘I worry that people will think I’m weird.’ Troubled by blurriness was mentioned by 8 (33%) of 24 patients e.g. ‘I don’t like it wandering because it goes all blurry.’ Comments from others e.g. ‘kids ask what is wrong with my eye,’ ‘people say ‘your eye is drifting” was also mentioned by 8 (33%) of 24 patients. The next most frequently mentioned topics [7 (29%) of 24 children each] were: Ocular discomfort e.g. ‘My eyes hurt when I’m tired,’ Self consciousness including phrases such as ‘My eyes are different from other kids’ and ‘it kind of feels a little bit embarrassing,’ and Wish didn’t have it e.g. ‘I wish my eyes would stay straight’ and ‘wish I could stop them from going in and out.’

Table 3.

Topic frequency of health related quality of life concerns related to the child with intermittent exotropia, shown as expressed by the child themselves and by the parent.

Concerns expressed by the child	Number of children (%)	Concerns expressed by the parent about the child	Number of parents (%)
Worry	10 (42%)	Comments from others	15 (63%)
Troubled by blurriness	8 (33%)	Appearance to others	9 (38%)
Comments from others	8 (33%)	Troubled by need to correct exotropia	6 (25%)
Ocular discomfort	7 (29%)	Reading	5 (21%)
Self consciousness	7 (29%)	Self consciousness	5 (21%)
Wish didn’t have it	7 (29%)	Relationships †	4 (17%)
Appearance to others	4 (17%)	Self confidence†	4 (17%)
Self esteem	4 (17%)	Concentration†	3 (13%)
Bothered by eyes*	3 (13%)	Self esteem	3 (13%)
Reading	3 (13%)	Clinic visits†	2 (8%)
Appearance to self*	2 (8%)	Computer†	2 (8%)
Depth perception	2 (8%)	Eye contact†	2 (8%)
Monocular eye closure*	2 (8%)	Ocular discomfort	2 (8%)
Ocular sensation*	2 (8%)	School†	2 (8%)
Troubled by need to correct exotropia	2 (8%)	Troubled by blurriness	2 (8%)
Exotropia triggered by staring*	1 (4%)	Worry	2 (8%)
Parent’s need to correct*	1 (4%)	Depth perception	1 (4%)
Troubled by double vision*	1 (4%)	Exotropia triggered by television†	1 (4%)
		Eye fatigue†	1 (4%)
		General awareness of deviation†	1 (4%)
		Headaches†	1 (4%)
		Wish didn’t have it	1 (4%)

^*= Topics mentioned only by children

^†= Topics mentioned only by parents

Parent interviews

For the 24 parents, a total of 217 phrases were extracted. Ninety-six (44%) of 217 phrases related to the child’s quality of life; phrases covered 22 topic areas. One-hundred and twenty-one (56%) of 217 phrases related to the parent’s quality of life; phrases covered 14 topic areas.

Concerns expressed by the parents relating to their child’s HRQOL

The overall frequency of the 22 topics mentioned by parents regarding their child’s HRQOL is shown in Table 3. The number of topics mentioned per parent ranged from zero (1 parent) to 9 (median 3). The most frequently mentioned topic was Comments from others [15 (63%) of 24] including phrases such as ‘people sometimes comment on my child’s eye’ and ‘he doesn’t like it if somebody makes a comment.’ The next most frequently mentioned topics were Appearance to others [9 (38%) of 24] e.g. ‘friends think her eye looks weird’ and Troubled by need to correct exotropia [6 (25%) of 24] e.g. ‘closes eyes and shakes head to get it [eye] in.’ Concerns regarding Reading (‘complained about difficulties reading’), and Self-consciousness (‘she is a little embarrassed about it’) were made by 5 (21%) of 24 parents.

Concerns expressed by the parents relating to their own HRQOL

The overall frequency of the 14 topics mentioned by parents regarding their own HRQOL is shown in Table 4. Due to the large number of phrases describing specific worries, topics relating to worry were divided into individual areas of concern rather than grouping all types of worry together. The number of topics mentioned per parent ranged from one to eight (median 3). The most frequently mentioned topics were Worry related, specifically Worry regarding possible surgery [15 (63%) of 24], including phrases such as ‘worry about needing surgery’, ‘worry about surgery making it worse,’ ‘worry about risks of surgery,’ and Worry regarding possible deterioration [14 (58%) of 24] e.g. ‘worried it will get worse,’ ‘worried about losing depth perception’ and ‘I don’t want one eye to give up.’ The topic Worry regarding other potential effects of IXT [11 (46%) of 24], included worry over individual concerns, such as ability to drive in the future, potential affects on schooling, and potential affects on personality. Worry regarding others noticing was mentioned by 8 (33%) of 24 parents, including phrases such as ‘worry that people will think he is mentally handicapped,’ and ‘worried about the social thing.’ Seven (29%) of 24 parents mentioned phrases regarding Comments from others e.g. ‘people say there is something wrong with my child’s eyes’ and ‘other people ask if we notice.’

Table 4.

Topic frequency of health related quality of life concerns of parents whose children have intermittent exotropia (IXT).

Concerns expressed by parents	Number of Parents (%)
Worry regarding possible surgery	15 (63%)
Worry regarding possible deterioration	14 (58%)
Worry regarding other potential effects of IXT	11 (46%)
Worry regarding others noticing	8 (33%)
Comments from others	7 (29%)
Parent’s need to correct	6 (25%)
Bothered by eyes	5 (21%)
Clinic visits	5 (21%)
Wish didn’t have it	5 (21%)
Forced to accept it	4 (17%)
Guilt	3 (13%)
Appearance to others	1 (4%)
Lack of definitive medical plan	1 (4%)
Parent-child relationship	1 (4%)

Comparison of child and parent responses regarding child’s HRQOL

Regarding the child’s HRQOL, seven topics were mentioned only by the children with IXT and not by the parents (Table 3). Three (13%) of the 24 children mentioned that they were Bothered by their eyes, e.g. ‘bothered that my eyes go out.’ Other topics mentioned only by children were: Appearance to self, Monocular eye closure, Ocular sensation, Troubled by double vision, Parent’s need to correct, and Exotropia triggered by staring (Table 3).

Eleven topics were mentioned by the parents but were not mentioned by the children (Table 3). Four (17%) parents thought IXT affected their child’s Self confidence using phrases such as ‘sometimes he holds himself back because of his eyes,’ ‘it affects self confidence,’ and ‘Relationships’ e.g. ‘affects his ability to interact with others.’ Three (13%) of the 24 parents perceived IXT to cause problems with their child’s Concentration, e.g. ‘he lacks some concentration because of it.’ Other topics mentioned by parents only were: Clinic visits, Computer, Eye contact, School, Eye fatigue, General awareness of deviation, Headaches, and Exotropia triggered by TV (Table 3).

There were 11 topics relating to the child’s HRQOL that were mentioned in both child and parent interviews (Table 3). Analysis of these 11 topics by parent-child pairs (i.e. parent and child within the same family) showed poor agreement between parents and their children (Table 5). Among topics mentioned by both parents and children, the topics with the best agreement were Reading (2 (33%) of 6), Appearance to others (3 (30%) of 10) and Comments from others (4 (21%) of 19, Table 5).

Table 5.

The 11 topics relating to the child’s health related quality of life that were mentioned in both child and parent interviews. Data show the percent agreement between parent-child pairs (i.e., topics mentioned by both the parent and the child within the same family).

Topic	Number of families where topic mentioned by either parent or child	Parent-child agreement	Mentioned by child but not by parent	Mentioned by parent but not by child
Comments from others	19	4 (21%)	4 (21%)	11 (58%)
Child worry	11	1 (9%)	9 (82%)	1 (9%)
Self consciousness	11	0 (0%)	7 (64%)	4 (36%)
Appearance to others	10	3 (30%)	1 (10%)	6 (60%)
Troubled by blurriness	9	1 (11%)	7 (78%)	1 (11%)
Ocular discomfort	8	1 (13%)	6 (75%)	1 (13%)
Troubled by need to correct exotropia	7	1 (14%)	1 (14%)	5 (71%)
Wish didn’t have it	7	1 (14%)	6 (86%)	0 (0%)
Reading	6	2 (33%)	1 (17%)	3 (50%)
Self esteem	6	1 (17%)	3 (50%)	2 (33%)
Depth perception	3	0 (0%)	2 (67%)	1 (33%)

Discussion

We have identified a range of specific HRQOL concerns affecting children with IXT and their parents. The topics most frequently expressed by children with IXT were Worry concerning their eyes, Comments from others, and that they were Troubled by blurriness. Parents identified the main effects of IXT on their children to be dealing with Comments from others. Interviews also identified affects of the child’s IXT on the HRQOL of the parents themselves, primarily worry relating to the possibility of surgery, the possibility of deterioration, and the possibility of other effects of IXT.

Nearly half of children interviewed expressed worry related to their eye condition. There are few data on the presence or nature of worry in children with strabismus, but worry regarding health has been identified even in normal children, affecting 69% in a study by Silverman et al.¹⁷ Whether children with IXT worry more about their eyes than normal children will be analyzed in the next phase of our study. It is possible, especially in younger children, that the word ‘worry’ encompassed other, more specific concerns such as self-consciousness that children were not able to articulate.

Concern regarding Comments from others was expressed by one third of children, most phrases indicating that comments were from other children. Also, more than half of the parents identified Comments from others as affecting their child’s HRQOL. The age at which children may become aware of strabismus in others was studied by Paysse et al⁴ who found that at approximately 6 years of age children began to express an overall negative response to playing with dolls with strabismus. It is unclear whether Comments from others should influence management decisions but assessing such comments appears important in evaluating HRQOL in IXT.

Children expressed concern regarding blurriness of vision more frequently than we would have predicted. Expressions related to being Troubled by blurriness were associated with awareness of the eye wandering. It is possible that children used the word ‘blurry’ to describe diplopia, although patients with IXT are typically thought to suppress. Alternatively, it may be that comments relate to experiences of children when not wearing their habitual refractive correction [prescribed in 10 (42%) of 24 children]. Nevertheless, in the context of the interview it was clear that the experience of blurriness was associated with the episode of the XT itself. We speculate that in IXT a child’s observations of blurry vision may indicate the effect of accommodative or motor convergence mechanisms used to control the exodeviation, but this remains controversial.^{18, 19} Alternatively, it may be that children describe as blur the change in visual experience noticed when suppression occurs.

Analysis of parent-child pairs showed poor agreement, with most parents raising concerns that were different than those raised by their child and vice versa. Our finding of poor parent-child concordance is consistent with the report by Powell et al (Quality of Life in Intermittent Exotropia. Powell CJ et al. Poster presentation, AAPOS 2007) who found poor correlation between parent and child scores on all PedsQL²⁰ questionnaire sub-scales. Lack of agreement between self and proxy-reporting, especially in social or emotional domains, is well recognized^20–23 and creates challenges for management decisions. For children with IXT, the differences between child and parent concerns raise important questions regarding the indications for and aims of treatment: are we treating the child or the parent? Self-reporting is considered the standard measure of quality of life,^{21, 24} but in children this cannot always be achieved. Where possible a combination of parent and self-reporting has been recommended.^21–23 Using data from this present study, we aim to develop questionnaires enabling self and parental reporting of HRQOL in IXT, based on concerns identified in this present study.

Regarding the effect of IXT on the HRQOL of parents themselves, the most frequently occurring topics were worry-related. It is interesting to note that, without exception, worry was caused by potential, not actual effects of IXT, i.e., the possibility of surgery, deterioration, or others noticing the strabismus. It is possible that transference of parental concerns may lead to worry in children.^25–27 Some parental comments in our study suggest that medical advice conveys a sense of uncertainty as to the likely course of disease and the optimal treatment strategy. Improved understanding of the natural history of IXT and indications for surgery may lead to improved parental education and result in reduced parental worry and possibly reduced child worry.

There are several potential weaknesses to our study. The majority of children recruited were female, although this is consistent with the reported prevalence of IXT.²⁸ We did not find a noticeable difference between concerns reported by females and those reported by males, but this may be worthy of further study. To create a less threatening atmosphere, we did not insist on parents leaving the room when the child was interviewed. As a result, most of the younger children (<10 years of age) were interviewed in the presence of their parents. This may have limited the openness of the child, although there was no indication that the parent’s presence restricted what the child said. When interviewing parents of younger children, it was not always possible to ask children to leave so that parents were alone for their interview. Again, this may have limited how much the parent revealed concerning particular concerns. An additional potential weakness is that we did not standardize the timing of interviews relative to the clinical exam. It is possible that parent and child responses might have differed before versus after the examination.

We have identified a range of HRQOL concerns affecting children with IXT. Parental perceptions of the effects of IXT on their child’s HRQOL differed from concerns identified by the child themselves. We also identified concerns affecting the HRQOL of the parents. The specific concerns identified in this study will be used to develop patient-derived, condition-specific HRQOL questionnaires both for children with IXT and parents of children with IXT. Such questionnaires should prove valuable in assessing the needs of an individual patient, determining criteria for intervention, and improving our understanding of the nature of IXT.