Associations between health-related quality of life and the decision to perform surgery for childhood intermittent exotropia.

OBJECTIVE: To assess associations between health-related quality of life (HRQOL) and the decision to perform strabismus surgery for children with intermittent exotropia. DESIGN: Retrospective chart review. PARTICIPANTS: Children with intermittent exotropia. METHODS: Included subjects, identified in a clinical practice, had assessment of HRQOL using the intermittent exotropia questionnaire (IXTQ), comprising child, proxy, and parent components (parent domains: function, psychosocial, and surgery). The IXTQ scores were evaluated for association with surgery, along with standard clinical measures: prism and alternate cover test (PACT), stereoacuity, and control score (mean of the 3 most recent scores). Included data were from preoperative examination (surgical cohort) or from most recent follow-up examination (nonsurgical cohort). Univariate and multivariate logistic regression analyses were performed, and relative risk (RR) ratios were calculated. Spearman rank correlations were calculated to identify highly correlated items. MAIN OUTCOME MEASURES: Association of individual factors with the decision to perform surgery, calculated using RR ratios. RESULTS: One hundred six children with intermittent exotropia (median age, 6 years; range, 2-16 years) were eligible for inclusion. Nineteen (18%) of 106 underwent surgery. Using all available data, the IXTQ proxy score, IXTQ parent function score, IXTQ parent psychosocial score, distance control score, near control score, near PACT, and Randot Preschool stereoacuity (Stereoptical Co, Inc, Chicago, IL) were associated with undergoing surgery (P<0.1). Sixty-nine of 106 patients had complete data on all factors identified in univariate analysis and were included in multivariate analyses. Fourteen (20%) of these 69 patients underwent surgery. In multivariate analyses, poor distance control score (RR, 1.83; 95% confidence interval [CI], 1.25-2.68) and reduced IXTQ parent function score (RR, 0.96; 95% CI, 0.92-0.99) were associated with surgical intervention. Repeat multivariate analyses retaining only 1 of the highly correlated items showed IXTQ proxy, IXTQ parent psychosocial, larger near PACT, and worse near control were also associated with surgery. CONCLUSIONS: After accounting for poorer exodeviation control at distance, reduced parent and proxy HRQOL were associated with undergoing strabismus surgery for childhood intermittent exotropia. Recognizing reduced parental HRQOL may be important, with a possible role for educational or counselling interventions.

Quantifying diplopia with a questionnaire.

PURPOSE: To report a diplopia questionnaire (DQ) with a data-driven scoring algorithm. DESIGN: Cross-sectional study. PARTICIPANTS: To optimize questionnaire scoring, 147 adults with diplopic strabismus completed both the DQ and the Adult Strabismus-20 (AS-20) health-related quality-of-life (HRQOL) questionnaire. To assess test-retest reliability, 117 adults with diplopic strabismus. To assess responsiveness to surgery, 42 adults (46 surgeries). METHODS: The 10-item AS-20 function subscale score (scored 0-100) was defined as the gold standard for severity. A range of weights was assigned to the responses and the gaze positions (from equal weighting to greater weighting of primary and reading). Combining all response option weights with all gaze position weights yielded 382848 scoring algorithms. We then calculated 382848 Spearman rank correlation coefficients comparing each algorithm with the AS-20 function subscale score. MAIN OUTCOME MEASURES: To optimize scoring, Spearman rank correlation coefficients (measuring agreement) between DQ scores and AS-20 function subscale scores. For test-retest reliability, 95% limits of agreement and intraclass correlation coefficient (ICC). For responsiveness, change in DQ score. RESULTS: For the 382 848 possible scoring algorithms, correlations with AS-20 function subscale score ranged from -0.64 (best correlated) to -0.55. The best-correlated algorithm had response option weights of 5 for rarely, 50 for sometimes, and 75 for often, and gaze position weights of 40 for straight ahead in the distance, 40 for reading, 1 for up, 8 for down, 4 for right, 4 for left, and 3 for other, totaling 100. There was excellent test-retest reliability with an ICC of 0.89 (95% confidence interval, 0.84-0.92), and 95% limits of agreement were 30.9 points. The DQ score was responsive to surgery with a mean change of 51 ± 34 (P<0.001). CONCLUSIONS: We have developed a data-driven scoring algorithm for the DQ, rating diplopia symptoms from 0 to 100. On the basis of correlations with HRQOL, straight-ahead and reading positions should be highly weighted. The DQ has excellent test-retest reliability and responsiveness, and may be useful in both clinical and research settings.

Comparing outcome criteria performance in adult strabismus surgery.

PURPOSE: To evaluate the performance of motor, diplopia, and health-related quality of life (HRQOL) criteria when analyzing outcomes of adult strabismus surgery. DESIGN: Cohort study. PARTICIPANTS: We studied 159 adults undergoing 171 strabismus surgeries. METHODS: All patients underwent clinical assessment preoperatively and 6 weeks postoperatively, including completion of Adult Strabismus-20 HRQOL questionnaires. Preoperatively, strabismus was classified as either diplopic (n = 117), nondiplopic (n = 38), or atypical diplopic (n = 16). To assess performance of motor, diplopia, and HRQOL criteria, success was defined a priori and applied separately and in combinations. For success: (1) motor criteria, <10 prism diopters by simultaneous prism cover test; (2) diplopia criteria, none or only rare in primary distance and for reading; (3) HRQOL criteria, exceeding previously reported 95% limits of agreement (LOA). MAIN OUTCOME MEASURES: Surgical success rate when applying motor, diplopia, and HRQOL criteria alone and in combinations. RESULTS: Overall, success rates were 90% for motor criteria, 74% for diplopia criteria, and 60% for HRQOL criteria. Combining criteria, the highest success rate was for motor plus diplopia criteria (67%) and the lowest success rate was when combining motor, diplopia, and HRQOL criteria (50%). CONCLUSIONS: Applying motor criteria alone yields the highest success rates when evaluating outcomes in adult strabismus surgery, but motor criteria do not fully represent the patient's postoperative status. Combining diplopia criteria with motor criteria provides a more clinically relevant standard for judging the success of adult strabismus surgery. For HRQOL criteria, exceeding 95% LOA at 6 weeks postoperatively seems to be a difficult hurdle to clear for some individual patients, and evaluating change in HRQOL score may be more useful in cohort studies.

Comparison of Methods for Measuring Cyclodeviation.

PURPOSE: To compare the double-Maddox rod test with other methods of measuring cyclodeviation DESIGN: Retrospective cohort study. METHODS: We retrospectively identified 153 adults in a clinical practice with cyclodeviation assessed using double-Maddox rods, of whom 105 were also assessed using fusible synoptophore targets, 73 using nonfusible synoptophore targets, 118 using single-Maddox rod, and 43 using fundus photography. Relationships between double-Maddox rod and other tests were evaluated by calculating mean differences with 95% confidence intervals (CI), intraclass correlation coefficients (ICC), and Bland-Altman plots with linear regression. RESULTS: Synoptophore cross-in-circle targets and the largest (of right or left) single-Maddox rod values were similar to double-Maddox values (mean differences: -1.2° and 0.1°, respectively; ICC: 0.79 and 0.82, respectively). Synoptophore house targets measured less excyclodeviation (mean difference: -2.7°; ICC: 0.71). Mean summed single-Maddox rod values were somewhat similar to double-Maddox values (mean difference: 1.5°; ICC: 0.85), but differences increased with greater cyclodeviation (r2 = 0.2678; P < .001). Fundus photographs showed large, uncorrelated differences compared with double-Maddox rod test, when summing right and left eyes and when using the largest of right or left (mean differences: 12.2° and 6.2°; ICC: -0.02 and 0.21, respectively), and differences increased with greater cyclodeviation (r2 = 0.4094; P < .001 and r2 = .1143; P= .03, respectively). CONCLUSIONS: There was good agreement between double-Maddox and the largest single- Maddox test values and synoptophore cross-in-circle targets but poorer agreement with other tests. Further study is needed to understand which measurements best reflect true cyclodeviation and relationships with symptoms.

Quality of life and functional vision across pediatric eye conditions assessed using the PedEyeQ.

PURPOSE: To evaluate eye-related quality of life (ER-QOL) and functional vision across a wide range of pediatric eye conditions, using the Pediatric Eye Questionnaires (PedEyeQ). METHODS: A total of 1,037 children with an eye condition and 254 visually normal controls, across 0-4, 5-11, and 12-17 years age groups, completed the following questionnaires: Child PedEyeQ (Functional Vision, Bothered by Eyes/Vision, Social, Frustration/Worry domains), Proxy PedEyeQ (same domains plus Eye Care), and Parent PedEyeQ (Impact on Parent and Family, Worry about Child's Eye Condition, Worry about Child's Self-perception and Interactions, and Worry about Functional Vision domains). The primary eye condition was classified as amblyopia (n = 171), cataract (n = 99), cerebral visual impairment (CVI; n = 50), cornea (n = 20), eyelid (n = 35), glaucoma (n = 24), nystagmus (n = 57), orbital (n = 19), pupil/iris (n = 7), refractive error (n = 119), retina (n = 82), strabismus (n = 332), and uveitis (n = 22). RESULTS: PedEyeQ domain scores (scaled 0-100) were significantly worse across eye conditions, compared with controls. Child PedEyeQ greatest differences were on the Bothered by Eyes/Vision domain (nystagmus 5-11 years, -26 points [95% CI, -39 to -12]; nystagmus 12-17 years, -45 [95% CI, -61 to -28]). Proxy PedEyeQ differences were greatest on Functional Vision (CVI 0-4 years, -45 [95% CI, -56 to -34]; CVI 5-11 years, -58 [95% CI, -72 to -43]; nystagmus 12-17 years, -50 [95% CI, -69 to -31]). Parent PedEyeQ differences were greatest on Worry about Child's Functional Vision (CVI 0-4 years, -64; 95% CI -77 to -50). CONCLUSIONS: The PedEyeQ detects reduced ER-QOL and functional vision across pediatric eye conditions, and across age groups, indicating its utility for clinical practice and clinical trials.

Association of Visual Acuity with Eye-Related Quality of Life and Functional Vision Across Childhood Eye Conditions.

PURPOSE: We evaluated relationships between visual acuity (VA) and eye-related quality of life and functional vision in children, across a spectrum of pediatric eye conditions, using the Pediatric Eye Questionnaire (PedEyeQ). DESIGN: Cross-sectional study. METHODS: Three hundred ninety-seven children (5-11 years of age) with an eye condition and 104 visually normal control subjects completed the Child PedEyeQ (functional vision, bothered by eyes/vision, social, and frustration/worry domains). One parent for each child completed the Proxy PedEyeQ (same domains as child plus eye care) and parent PedEyeQ (impact on parent and family, worry about child's eye condition, worry about child's self-perception and interactions, and worry about functional vision domains). Each domain was Rasch-scored and Spearman rank correlations were calculated to evaluate relationships between better-seeing-eye and worse-seeing-eye VA and PedEyeQ domain score. RESULTS: There was a significant relationship between poorer better-seeing-eye VA and lower (worse) PedEyeQ score on 2 of 4 child domains (e.g., functional vision, r = -0.1474; P = .005), on 2 of 5 proxy PedEyeQ domains (e.g., functional vision, r = -0.2183; P < .001), and on 2 of 4 parent PedEyeQ domains (e.g., impact on parent and family, r = -0.1607; P = .001). Worse-seeing-eye VA was associated with lower PedEyeQ scores across all child, proxy and parent domains (P < .01 for each) with the exception of the child social domain (P = .15). CONCLUSIONS: Both better-seeing-eye and worse-seeing-eye VA were associated with functional vision and eye-related quality of life in children, assessed using the PedEyeQ, although other factors may also influence relationships. These data further validate using the PedEyeQ across pediatric eye conditions.

Understanding the Impact of Residual Amblyopia on Functional Vision and Eye-related Quality of Life Using the PedEyeQ.

PURPOSE: To evaluate the effect of residual amblyopia on functional vision and eye-related quality of life (ER-QOL) in children and their families using the Pediatric Eye Questionnaire (PedEyeQ). DESIGN: Prospective cross-sectional study. METHODS: Seventeen children with residual amblyopia (no current treatment except glasses), 48 visually normal controls without glasses, and 19 controls wearing glasses (aged 8-11 years) completed the Child 5-11 year PedEyeQ. One parent for each child completed the Proxy 5-11 PedEyeQ, Parent PedEyeQ. Rasch-calibrated domain scores were calculated for each questionnaire domain and compared between amblyopic children and controls. RESULTS: PedEyeQ scores were significantly lower (worse) for children with residual amblyopia than for controls without glasses across all domains: Child PedEyeQ greatest mean difference 18 points worse on Functional vision domain (95% confidence interval [CI] -29 to -7; P < .001); Proxy PedEyeQ greatest mean difference 31 points worse on Functional vision domain (95% CI -39 to -24; P < .001); Parent PedEyeQ greatest mean difference 34 points worse on the Worry about child's eye condition domain (95% CI -46 to -22; P < .001). Compared with controls wearing glasses, PedEyeQ scores were lower for residual amblyopia on the Child Frustration/worry domain (P = .03), on 4 of 5 Proxy domains (P ≤ .05), and on 3 of 4 Parent domains (P ≤ .05). CONCLUSIONS: Residual amblyopia affects functional vision and ER-QOL in children. Parents of amblyopic children also experience lower quality of life. These data help broaden our understanding of the everyday-life impact of childhood residual amblyopia.

Eye-related quality of life and functional vision in children wearing glasses.

PURPOSE: To evaluate eye-related quality of life (ER-QOL) and functional vision in children wearing glasses using the Pediatric Eye Questionnaire (PedEyeQ). METHODS: Children 5-17 years of age with normal visual acuity who wore glasses for correction of refractive error and with no other eye condition or treatment and control subjects who did not wear glasses, along with 1 parent for each child, were prospectively enrolled. Children completed the Child 5-11 or 12-17 PedEyeQ (four domains); parents completed the Proxy 5-11 or 12-17 questionnaires (perceived effect on child; five domains) and also the Parent questionnaire (effect on parent themselves; four domains). Each domain was Rasch-scored (converted to 0-100), and scores between groups were compared. RESULTS: A total of 40 subjects and 99 non-glasses-wearing controls, along with 1 parent for each child, were included. Children 5-11 and 12-17 years of age who wore glasses had lower PedEyeQ scores across all domains compared with controls (mean differences, -6 to -15; P ≤ 0.04 for each domain). Proxy scores were also lower for glasses wearers across age groups and domains (mean differences, -4 to -18; P ≤ 0.02), and Parent scores were lower for parents of children wearing glasses (mean differences, -6 to -18; P < 0.001 for each domain). CONCLUSIONS: In this study, glasses wearers had reduced ER-QOL and functional vision compared with control subjects. Parents of children wearing glasses also experience reduced quality of life.

Association of Strabismus With Functional Vision and Eye-Related Quality of Life in Children.

Importance: Strabismus is common, affecting 2% to 4% of children, but how children and their families are affected in everyday life is poorly understood. Objective: To evaluate the association of strabismus with functional vision and eye-related quality of life in children and their families using the Pediatric Eye Questionnaire (PedEyeQ). Design, Setting, and Participants: This cross-sectional study was conducted between December 2017 and October 2019 and included 91 children with strabismus and 166 visually normal controls across 3 age groups (0-4, 5-11, and 12-17 years) who were enrolled at Mayo Clinic, Rochester, Minnesota, and Retina Foundation of the Southwest, Dallas, Texas. Exposures: Children completed the child PedEyeQ (5 to 11- and 12 to 17-year versions: functional vision, bothered by eyes/vision, social, and frustration/worry domains); parents completed the proxy (0 to 4-, 5 to 11-, and 12 to 17-year versions: functional vision, bothered by eyes/vision, social, frustration/worry, and eye care domains) and the parent PedEyeQ (impact on parent and family, worry about child's eye condition, worry about child's self-perception and interactions, and worry about child's functional vision domains). Rasch-calibrated PedEyeQ scores were calculated for each domain and converted to 0 (worst) to 100. Main Outcomes and Measures: PedEyeQ domain scores. Results: Of 91 participants with strabismus, 41 (45.1%) were girls, 74 (81.3%) were white, 4 (4.4%) were Asian, 5 (5.5%) were more than 1 race, 5 (5.5%) were African American, and 2 (2.2%) were American Indian/Alaska Native. Child PedEyeQ domain scores were lower with strabismus vs visually normal controls among children ages 5 to 11 years and the greatest mean (SD) difference was in functional vision (12 [14] points; 95% CI, 6-18; P = .001), and among children ages 12 to 17 years, the greatest mean (SD) difference was in frustration/worry (27 [13] points; 95% CI, 18-36; P < .001). Proxy PedEyeQ domain scores were also lower with strabismus. The greatest difference among children ages 0 to 4 years was in functional vision (13 [9] points; 95% CI, 9-16; P < .001), among children ages 5 to 11 years was in functional vision (26 [10] points; 95% CI, 22-30; P < .001); and among children ages 12 to 17 years was in functional vision (21 [12] points; 95% CI, 12-30; P < .001), social (21 [13] points; 95% CI, 12-30; P < .001), and frustration/worry (21 [13] points; 95% CI, 12-30; P < .001). Parent PedEyeQ domain scores were lower with strabismus; the greatest difference was in worry about child's eye condition (38 [14] points; 95% CI, 34-42; P < .001). Conclusions and Relevance: Strabismus is associated with reduced functional vision and eye-related quality of life in children. Parents of children with strabismus also experience a reduced quality of life. These findings advance our understanding of how strabismus affects children and their families and should be considered when defining patient management goals.

Validation of the Pediatric Eye Questionnaire in Children with Visual Impairment.

PURPOSE: To evaluate the recently developed Pediatric Eye Questionnaire (PedEyeQ) in visually impaired and visually normal children, as an initial validation of the PedEyeQ. DESIGN: Questionnaire validation study. METHODS: A total of 48 children with visual impairment (retinal, cortical, and corneal conditions) and 59 visually normal controls were enrolled at 2 centers. Five- to 17-year-old children completed the Child PedEyeQ (5-11- and 12-17-year-old versions), and parents completed the Proxy PedEyeQ (0-4-, 5-11-, and 12-17-year-old versions) and the Parent PedEyeQ. Rasch scores were calculated by age group for each distinct domain within each age-specific PedEyeQ (converted to 0-100 for interpretation). Domains for the Child PedEyeQ are functional vision, bothered by eyes/vision, social, and frustration/worry. Proxy PedEyeQ domains are functional vision, bothered by eyes/vision, social, frustration/worry, and eye care. Parent PedEyeQ domains are impact on parent and family, worry about child's eye condition, worry about child's self-perception/interactions, and worry about child's functional vision. For each domain, median PedEyeQ scores were compared between visually impaired and visually normal cohorts. RESULTS: Child 5-11 and 12-17 PedEyeQ scores were significantly lower (worse) for visually impaired children than for controls for each domain (P < .007), except for the social domain in 5-11-year-old children (P = .13). In addition, Proxy 0-4-, 5-11-, and 12-17-year-old PedEyeQ scores, and Parent PedEyeQ scores, were lower for visually impaired children across all domains (P < .001). CONCLUSIONS: Visually impaired children and their parents had significantly lower (worse) PedEyeQ scores than visually normal controls, across functional vision and eye-related quality of life domains, demonstrating known-group validity of the PedEyeQ in children with visual impairment.

Development of Pediatric Eye Questionnaires for Children With Eye Conditions.

PURPOSE: To develop patient-derived Pediatric Eye Questionnaires (PedEyeQ) to separately assess eye-related quality of life (ER-QOL) and functional vision in children with eye conditions. DESIGN: Questionnaire development study. METHODS: A total of 444 children (0 to <18 years old), across 10 diagnostic categories, were enrolled at 2 sites. All parents (n = 444) and 277 children (5 to <18 years old) completed master questionnaires, developed from patient-derived concerns. Factor analysis was performed to identify unidimensional domains (eigenvalue >1.0) and Rasch analyses (differential item functioning, targeting, fit) to reduce items (separate analyses for subjects aged 0-4, 5-11, and 12-17 years and for each factor). RESULTS: The Child 5- to 11-year-old PedEyeQ consisted of 4 unidimensional domains/questionnaires: functional vision, bothered by eyes/vision, social, frustration/worry (10 items each). The Child 12- to 17-year-old PedEyeQ consisted of the same 4 domains (total 39 items). The Proxy 0- to 4-year-old PedEyeQ consisted of 3 questionnaires/domains: functional vision, bothered by eyes/vision, social (total 29 items). The Proxy 5- to 11-year-old PedEyeQ consisted of 5 questionnaires/domains: functional vision, bothered by eyes/vision, social, frustration/worry, eye care (total 39 items), as did the Proxy 12- to 17-year-old PedEyeQ (total 42 items). The Parent PedEyeQ consisted of 4 questionnaires/domains: impact on parent/family, worry regarding child's eye condition, worry regarding child's self-perception and interactions, worry regarding child's visual function (total 35 items). Rasch look-up tables were created for scoring. CONCLUSIONS: By following a rigorous approach, we have developed Pediatric Eye Questionnaires for separately assessing functional vision and ER-QOL domains in children of any age and with any eye condition.

Test-retest variability of cyclodeviations measured using the double Maddox rod test.

The double Maddox rod test is often used to measure cyclodeviations and to monitor change over time. The purpose of this study was to estimate test-retest reliability and the amount of cyclodeviation that would be considered real change using 95% limits of agreement. We retrospectively studied 86 clinically stable patients with double Maddox rod measurements 5-175 days apart (median, 69 days). The range of cyclodeviation at the first measurement was 6° incyclodeviation to 15° excyclodeviation. We calculated the half width of the 95% limits of agreement to be 4.7°, which means that a change of ≥5° in cyclodeviation would be considered a real change. The threshold of 5° should be used when assessing change between two measurements made with double Maddox rods.

Dose Effect and Stability of Postoperative Cyclodeviation After Adjustable Harada-Ito Surgery.

PURPOSE: To report the dose-response relationship of adjustable Harada-Ito surgery, and postoperative stability of cyclodeviation. DESIGN: Retrospective cohort study. METHODS: Twenty patients underwent a unilateral adjustable Harada-Ito advancement of the anterior fibers of the superior oblique tendon, with or without resection. The surgical dose was calculated as resection plus advancement plus adjustment. Cyclodeviation was measured using double Maddox rods at preoperative and postoperative time points. We assessed dose effect by calculating change in cyclodeviation from preoperatively to postadjustment and evaluated the dose-response relationship by Spearman rank methods. Postoperative stability of cyclodeviation was assessed by comparing postadjustment measurements with 1-day and 6-week measurements, and also 1- and 5-year measurements, when available. RESULTS: Change in cyclodeviation from preoperatively to postadjustment ranged from 7 to 20 degrees of incyclo effect. There was a dose effect of 1.3 degrees per mm (rs = 0.52, P = .019). Postoperative incyclodeviation unwound a mean of 3.6 degrees ± 2.5 degrees between postadjustment and day 1. Overall change between postadjustment and the 6-week window was 6.5 ± 2.9 degrees in an excyclo direction. Unwinding continued with a mean change of 0.2 ± 2.6 degrees between the 6-week and 1-year windows, and of 0.4 ± 2.1 degrees between 1-year and 5-years windows. CONCLUSIONS: Adjustable Harada-Ito advancement of the anterior fibers of the superior oblique tendon has a dose response of 1.3 degrees per millimeter, but its effect regresses over time. Based on our data, an immediate postoperative target incyclodeviation of 7 degrees is reasonable.

Patient-derived questionnaire items for patient-reported outcome measures in pediatric eye conditions.

PURPOSE: To identify specific health-related quality of life (HRQOL) and functional vision concerns of children with eye conditions, and create comprehensive lists of potential questionnaire items as a first step in developing patient-reported outcome measures. METHODS: Children experiencing a range of pediatric eye conditions, along with one of their parents, were interviewed to identify specific concerns. Transcribed interviews were reviewed, and specific HRQOL and functional vision concerns were coded independently by two reviewers. Coded concerns were reviewed to formulate questions to address specific child concerns (derived from child and parent interviews) and specific parent concerns. Questions were grouped into bins of like questions. Two comprehensive lists of questions were formulated, one addressing child-related concerns and one addressing parent-related concerns. RESULTS: This study included 180 children and 328 parents. A total of 614 individual child questions were grouped into 36 bins (eg, appearance, coordination, glasses, learning), and 589 parent questions were formulated and grouped into 61 bins (eg, having to assist the child, worry about deterioration, time off work, safety). CONCLUSIONS: Using rigorous methods based on individual interviews, we identified a comprehensive list of patient- and parent-derived questionnaire items that address functional vision and HRQOL concerns of children with eye conditions and of their parents. We plan to use this large pool of potential questionnaire items to develop a formal set of pediatric outcome measures, and this pool of questions may also be a resource for future research.

Bilateral childhood visual impairment: child and parent concerns.

PURPOSE: To identify specific health-related quality of life and visual function concerns affecting children with bilateral visual impairment as expressed by children or one of their parents (proxy) and concerns affecting the parents themselves. METHODS: A total of 37 children <16 years of age with visual impairment (visual acuity worse than 20/70 in the better eye) and one parent for each child were prospectively enrolled. Semistructured individual interviews were performed with children 5-15 years of age (n = 16) and with one parent for each child (ages 0-15 years, N = 37). Interview transcripts were analyzed using NVivo software. Categories of concern were identified from both child and parent interviews, from which broad themes were identified. The frequencies of the themes and specific categories of concerns were calculated. RESULTS: Regarding the child's experience, categories of concern were grouped into 6 themes: visual function (expressed by 13 of 16 children [81%] and 33 of 37 parents [89%]), treatment (63% and 54%), emotions (50% and 68%), social (50% and 70%), physical discomfort (50% and 22%), and worry (38% and 8%). Concerns expressed regarding the parents' own experience were grouped into 5 themes: worry (100%), compensate-adjust for condition (89%), treatment (84%), emotions (81%), and affects family (46%). CONCLUSIONS: Individual interviews identified a wide spectrum of concerns in children with visual impairment and their parents, affecting functional, emotional, social and physical domains. Specific concerns will be used to develop patient-derived questionnaires for quantifying the effects of visual impairment on children and parents in everyday life.

Symptoms in Children with Intermittent Exotropia and Their Impact on Health-Related Quality of Life.

PURPOSE: In childhood intermittent XT, the frequency and type of symptoms have not been rigorously studied. We aimed to identify specific symptoms in children with intermittent XT, their frequency, and effects on health-related quality of life (HRQOL). METHODS: 35 children (5-13 years) with intermittent XT without previous surgery were enrolled in a prospective cohort study. Specific symptoms were identified from a previous study involving child and parent interviews and formulated as a 22-item symptom questionnaire. A frequency Likert-type rating scale was used and a response of "sometimes" or more was considered consistent with having the symptom. All 35 children, along with one parent for each child, also completed the patient-derived intermittent XT HRQOL questionnaire (IXTQ) with Child, Proxy, and Parent components. The frequency of symptoms was calculated, and the relationship between individual symptom question scores and Child, Proxy, and Parent HRQOL scores, was evaluated in multivariate linear regression analyses. RESULTS: The mean number of specific symptoms was 7 (range 2 to 19). The most frequently reported were: rubbing the eye (29 [83%] of 35), problems with eyes in the sun, and the eyes feeling tired (each 22, 63%). Lower (worse) Child IXTQ HRQOL scores were associated with symptoms of difficulty focusing eyes (P=0.0007), double vision (P=0.007), eyes hurting (P=0.006), and problems with eyes in the sun (P=0.06). There were weak associations between Proxy IXTQ and Parent IXTQ scores and child symptoms. Overall, 7 symptom questions were associated with reduced HRQOL in multivariate models. CONCLUSION: Children with intermittent XT frequently experience symptoms, some of which impact the child's HRQOL. Formal assessment of symptoms may aid understanding of the effects of intermittent XT on an individual child, and could use just the 7 symptom questions associated with reduced HRQOL.

Lookup Tables Versus Stacked Rasch Analysis in Comparing Pre- and Postintervention Adult Strabismus-20 Data.

PURPOSE: We compare two methods of analysis for Rasch scoring pre- to postintervention data: Rasch lookup table versus de novo stacked Rasch analysis using the Adult Strabismus-20 (AS-20). METHODS: One hundred forty-seven subjects completed the AS-20 questionnaire prior to surgery and 6 weeks postoperatively. Subjects were classified 6 weeks postoperatively as "success," "partial success," or "failure" based on angle and diplopia status. Postoperative change in AS-20 scores was compared for all four AS-20 domains (self-perception, interactions, reading function, and general function) overall and by success status using two methods: (1) applying historical Rasch threshold measures from lookup tables and (2) performing a stacked de novo Rasch analysis. Change was assessed by analyzing effect size, improvement exceeding 95% limits of agreement (LOA), and score distributions. RESULTS: Effect sizes were similar for all AS-20 domains whether obtained from lookup tables or stacked analysis. Similar proportions exceeded 95% LOAs using lookup tables versus stacked analysis. Improvement in median score was observed for all AS-20 domains using lookup tables and stacked analysis (P < 0.0001 for all comparisons). CONCLUSIONS: The Rasch-scored AS-20 is a responsive and valid instrument designed to measure strabismus-specific health-related quality of life. When analyzing pre- to postoperative change in AS-20 scores, Rasch lookup tables and de novo stacked Rasch analysis yield essentially the same results. TRANSLATIONAL RELEVANCE: We describe a practical application of lookup tables, allowing the clinician or researcher to score the Rasch-calibrated AS-20 questionnaire without specialized software.

Incorporating Health-related Quality of Life Into the Assessment of Outcome Following Strabismus Surgery.

PURPOSE: To evaluate changes in health-related quality of life (HRQOL) in adult strabismus patients classified as surgical failures by standard motor and diplopia criteria. DESIGN: Prospective cohort study evaluating outcomes. METHODS: Adults undergoing strabismus surgery in a single clinical practice, with preoperative and 1-year-postoperative Adult Strabismus-20 HRQOL questionnaires, were included. Motor and diplopia criteria were applied to classify outcomes (success, partial success, or failure). For those classified as failure, the medical record of the 1-year examination was reviewed to determine whether the patient reported subjective improvement. We evaluated improvement in HRQOL, defined as exceeding 95% limits of agreement on at least 1 of the 4 Adult Strabismus-20 domains. We compared proportions exceeding 95% limits of agreement in those reporting subjective improvement vs those who did not. RESULTS: Forty of 227 patients (18%) were classified as failure by motor and diplopia criteria, with 39 of 40 able to exceed Adult Strabismus-20 95% limits of agreement. Overall, 21 of 39 (54%) showed improved HRQOL by exceeding 95% limits of agreement on at least 1 of the 4 Adult Strabismus-20 domains (54% vs predicted 10% by chance alone; P < .0001). Twenty-five patients (64%) reported subjective improvement, of whom 16 (64%) showed improved HRQOL exceeding 95% limits of agreement. CONCLUSIONS: Many apparent surgical failures report subjective improvement, often reflected in improved HRQOL scores. We propose incorporating quantitative HRQOL criteria into the assessment of strabismus surgery outcomes, defining success as either meeting motor and diplopia criteria or showing improvement in HRQOL beyond test-retest variability.

Childhood esotropia: child and parent concerns.

PURPOSE: To identify specific health-related quality of life (HRQOL) concerns affecting children with esotropia as expressed by children or one of their parents (proxy) and concerns affecting the parents themselves. METHODS: Sixty children with esotropia (0-17 years of age) and 1 parent for each child were prospectively enrolled. Individual semistructured interviews were conducted with children aged 5-17 years (n = 40) and 1 parent each for child ages 0-17 years. Transcripts of recorded interviews were evaluated using NVivo software. Specific concerns were identified from both child and parent interviews and coded. From these specific codes, broad themes were identified. Frequency of each theme was calculated, along with the frequency of specific codes within each theme. RESULTS: Regarding the child's experience 6 broad themes were identified: visual function (mentioned by 32 of 40 children (80%) and by 50 of 60 parents (proxy assessment of child, 83%), treatment (78% and 85%), emotions (65% and 67%), social (58% and 68%), physical (58% and 32%), and worry (45% and 7%). Regarding the parents' own experience, 5 broad themes were identified: treatment (59 of 60 parents, 98%), worry (97%), emotions (82%), compensation for condition (80%), and affects family (23%). CONCLUSIONS: A wide range of concerns were identified from interviews of children with esotropia and their parents. Concerns reflect the impact of esotropia in physical, emotional, and social domains, and specific concerns will be used for the development of questionnaires to quantify the effects of esotropia on children's and parents' quality of life.