Background: The seacoasts of the Japanese Arc are fringed by many gravel beaches owing to active tectonic uplift and intense denudation caused by heavy rainfall. These gravel beaches are inhabited by gobies of the genus Luciogobius that burrow into the gravel sediment and live interstitially. Although their habitat and morphology (e. g., reduced fins, elongated, scale-less body, and highly segmented vertebral column) are highly unusual among fishes, little is known on how their morphological evolution has facilitated the colonization of interstitial habitats and promoted extensive diversification. We conducted thorough sampling of Luciogobius and related species throughout Japan, and performed molecular phylogenetic analysis to explore the patterns of morphological evolution associated with gravel beach colonization.
Purpose-To assess the health-related quality of life (HRQOL) of parents of children with intermittent exotropia using the newly developed condition-specific Parent Intermittent Exotropia Questionnaire (Parent IXTQ) and the generic PedsQL Family Impact Module (PedsQL FIM) and to compare the performance of both instruments.Methods-One parent was recruited for each of 59 children with intermittent exotropia (age range, 3 to 16; median, 7 years) and for each of 29 visually normal children (age range, 5 to 13; median, 8 years). The parent completed the Parent IXTQ and the PedsQL FIM. For each questionnaire, we compared median HRQOL scores between the intermittent exotropia group and visually normal group. We also calculated normal thresholds, which were defined as the 5th percentile score in the cohort of parents of visually normal children. The proportion of subnormal scores between questionnaires in parents of children with intermittent exotropia was compared.Results-The Parent IXTQ score was worse in the intermittent exotropia group than in the visually normal group (70.6 vs 94.1, p < 0.0001), whereas the PedsQL FIM scores were similar (97.9 vs 95.8, p = 0.8). More parents scored below normal using the Parent IXTQ than using the PedsQL FIM (31% vs 12%; p = 0.008, McNemar's test). Conclusions-TheParent IXTQ detects subnormal HRQOL in parents of children with intermittent exotropia more often than the PedsQL FIM. It is possible that parental worry may influence management decisions in children with intermittent exotropia and therefore parental HRQOL is worthy of further study.
he Intermittent Exotropia Questionnaire (IXTQ) is a patientderived, intermittent exotropia-specific instrument designed to evaluate health-related quality of life (HRQOL) in children with intermittent exotropia and their parents. 1,2 The IXTQ consists of 3 parts: the 12-item child IXTQ (completed by the child to assess the child's HRQOL), the 12-item proxy IXTQ (completed by the parent to assess the child's HRQOL), and the 17-item parent IXTQ (completed by the parent regarding his or her own HRQOL). 1 The child and proxy questionnaires each have a single subscale. The parent questionnaire contains 3 subscales: psychosocial, function, and surgery. The IXTQ is reliable and valid for assessing HRQOL in children with intermittent exotropia. [1][2][3] It is available for download free of charge at http://pedig.jaeb.org/.The IXTQ was originally developed using classical test theory. Rasch analysis may be used to modify and improve existing HRQOL instruments. [4][5][6][7][8][9] In the present study, Rasch analysis was used to refine the existing IXTQ, removing items that do not contribute meaningful information to the instrument and ensuring that response options are properly interpreted. Methods Patient CohortParents gave written informed consent, and children gave written assent when required. The protocol was approved by the institutional review boards of the Mayo Clinic, Jaeb Center for Health Research, and other local sites involved in the study. Data were collected and analyzed in accordance with the Health Insurance Portability and Accountability Act guidelines. The IXTQ was completed by 575 parents of 575 children aged 1 through 16 years with intermittent exotropia at the time of their child's clinic examination, enrolled from May 15, 2008, through July 24, 2013. The 295 children aged 5 years or older completed the age-appropriate child IXTQ. Parents and children completed the IXTQ as part of routine care in the strabismus practice of one of the authors (J.M.H., n = 110) or at the enrollment examination for 1 of 2 ongoing randomized clinical trials being conducted by the Pediatric Eye Disease Investigator Group (NCT 01032603 [n = 69] and NCT 01032330 [n = 396]). Child questionnaires were administered to children aged 5 through 7 years by study personnel. All 8-to 17-year-old child, proxy, and parent questionnaires were self-administered. Patient demographics are reported in eTable 1 in the Supplement. Statistical AnalysisBefore Rasch analysis, items with floor and ceiling effects on the child, proxy, and parent IXTQs were eliminated as described in the eMethods in the Supplement. Rasch analysis was performed on each of the 4 IXTQs using the analytic methods that we have applied previously (eMethods in the Supplement). 9 The performance and structure of the Rasch-IMPORTANCE The Intermittent Exotropia Questionnaire (IXTQ) is a patient, proxy, and parental report of quality of life specific to children with intermittent exotropia. We refine the IXTQ using Rasch analysis to improve reliability and validity.OBSERVATI...
Objective To assess associations between health-related quality of life (HRQOL) and the decision to perform strabismus surgery for children with intermittent exotropia (XT). Design Retrospective chart review Participants Children with intermittent XT 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, Surgery). 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 (surgical cohort), or most recent follow-up (non-surgical cohort) examinations. Univariate and multivariate logistic regression analyses were performed and relative risk (RR) ratios 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 relative risk ratios. Results 106 children with intermittent XT (median age 6, range 2 to 16 years) were eligible for inclusion. 19 (18%) of 106 underwent surgery. Using all available data, IXTQ Proxy score, IXTQ Parent-Function score, IXTQ Parent-Psychosocial score, distance control score, near control score, near PACT, and Preschool Randot stereoacuity were associated with undergoing surgery (P<0.1). 69 of 106 patients had complete data on all factors identified in univariate analysis and were included in multivariate analyses. 14 (20%) of these 69 underwent surgery. In multivariate analyses poor distance control score (RR 1.83, confidence interval [CI] 1.25, 2.68) and reduced IXTQ Parent-Function score (RR 0.96, CI 0.92, 0.99) were associated with surgical intervention. Repeat multivariate analyses retaining only one of 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 XT. Recognizing reduced parental HRQOL may be important, with a possible role for educational or counselling interventions.
Background We developed and validated the Jaeb Visual Acuity Screener (JVAS), a computerized visual acuity-based screening program for children that employs a rapid, age-specific, standardized algorithm for vision screening in the medical home that is available for download at no cost. Methods A total of 175 children aged 3 to <8 (median, 6) years were screened with the JVAS before undergoing a complete eye examination (gold standard). The JVAS presented 2 large single surround optotypes (20/100 and 20/80) and then 5 optotypes at a predetermined, age-specific normal threshold. Failure on the gold standard examination was determined using recently published referral criteria and published visual acuity norms for age. We evaluated the sensitivity and specificity of the JVAS for detecting reduced visual acuity, amblyopia, and amblyopia risk factors. JVAS pass/fail paradigms evaluated were inability to identify 3 of 4, 3 of 5, and 4 of 5 age-appropriate optotype presentations. Results Screening testability for the JVAS was high, at 100%. Sensitivity of the JVAS ranged from 88% to 91%, and specificity from 73% to 86%, with positive predictive value ranging from 66% to 79% and negative predictive value from 92% to 93% (ranges reflect different pass/fail paradigms). Conclusions The new JVAS provides an effective and practical method for screening 3- to 7-year-olds using any Windows-based computer. Providing the JVAS free-of-charge to pediatricians and school systems would standardize currently fragmented visual acuity-based screening practices.
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