Development of the Parental Questionnaire for Cerebral Visual Impairment in Children Younger than 72 Months.
Ontology highlight
ABSTRACT: Background and purpose
Cerebral visual impairment (CVI) is an underdiagnosed condition in children, and its assessment tools have focused on older children. We aimed to develop a parental questionnaire for cerebral visual impairment (PQCVI) for screening CVI in young children.Methods
The PQCVI comprised 23 questions based on a modified version of Houliston and Dutton's questionnaire for older children. The PQCVI with neurocognitive function tests was applied to 201 child-parent pairs with typically developing children younger than 72 months (age 32.4±20.1 months, mean±standard deviation). The children were classified into six age groups. The normative data, cutoff scores, and internal reliability were assessed and item analysis was performed. We referred to the total score for all questions as the cerebral visual function (CVF) score.Results
The normative data showed that the CVF score and the scores corresponding to ventral-stream and dorsal-stream visual functions plausibly increased with age. The scores rapidly reached 90% of their maximum values up to the age of 36 months, after which they increased slowly. Cronbach's alpha for all questions across all age groups was 0.97, showing excellent consistency. The item difficulty and item discrimination coefficients showed that the questions were generally adequate for this age stage.Conclusions
The PQCVI items produced reliable responses in children younger than 72 months. The rapid increase in scores before the age of 3 years supports the importance of early identification of CVI. Following additional clinical verification, the PQCVI may be useful for CVI screening.
SUBMITTER: Moon JH
PROVIDER: S-EPMC8242312 | biostudies-literature |
REPOSITORIES: biostudies-literature
Similar Datasets
Project description:Cerebral Visual Impairment (CVI) has become the leading cause of children's visual impairment in developed countries. Since CVI may negatively affect neuropsychomotor development, an early diagnosis and characterization become fundamental to define effective habilitation approaches. To date, there is a lack of standardized diagnostic methods to assess CVI in children, and the role of visual functions in children's neuropsychological profiles has been poorly investigated. In the present paper, we aim to describe the clinical and neuropsychological profiles and to investigate the possible effects of visual functions on neuropsychological performance of a cohort of children diagnosed with CVI. Fifty-one children with CVI were included in our retrospective analysis (inclusion criteria: verbal IQ > 70 in Wechsler scales; absence of significant ocular involvement). For each participant, we collected data on neuropsychological assessment (i.e., cognitive, cognitive visual, and learning abilities), basic visual functions (e.g., Best Corrected Visual Acuity-BCVA, contrast sensitivity, and ocular motor abilities) and global development features (e.g., neurological signs and motor development delay) based on standardized tests, according to patients' ages. The results showed that oculomotor dysfunction involving saccades and smooth pursuit may be a core symptom of CVI and might have a significant impact on cognitive visual and other neuropsychological abilities. Furthermore, visual acuity and contrast sensitivity may influence cognitive, cognitive visual, and academic performances. Our findings suggest the importance of a comprehensive assessment of both visual and neuropsychological functions in children when CVI is suspected, which is needed to provide a more comprehensive functional profile and define the best habilitation strategy to sustain functional vision.
| S-EPMC9221908 | biostudies-literature
Project description:In clinical practice Cerebral Visual Impairment (CVI) is typically diagnosed by observation of abnormal visually guided behaviors which indicate higher visual function deficits (HVFDs) suggesting abnormal brain development or brain damage in a child with a suitable clinical history. HVFDs can occur even in the presence of good visual acuity and may remain undiagnosed because the good visual acuity does not prompt further investigation. This leads to a lack of understanding of the child's visual perceptual difficulties. In a prospective study, we determined the spectrum of HVFDs in a group of children with history suggestive of brain damage or disruption of brain development and an independent diagnosis of CVI in comparison with typically developing children with a structured 51 question inventory, the Higher Visual Function Question Inventory (HVFQI-51) adapted from the Cerebral Vision Impairment Inventory, CVI-I. Here, we show that the HVFQI-51 can detect a range of HVFDs in children with CVI with good visual acuity and clearly distinguishes these children from typically developing children. HVFDs in our study group could mostly be attributed to dorsal stream visual processing dysfunction though the spectrum varied between children. We report on the inclusion of the "not applicable" response option in analysis providing a picture of HVFDs more in tune with the overall disability of each child. We also propose a subset of 11 questions (Top-11) which discriminate between children with CVI vs. behaviors seen in typical children: this provides both a potential screening tool for initial assessment of HVFDs and a measure of CVI-related impairment, and needs further validation in a secondary independent sample.
| S-EPMC8636735 | biostudies-literature
Project description:BackgroundCerebral Visual Impairment (CVI) is a very common finding in children affected by Cerebral Palsy (CP). In this paper we studied the characteristics of CVI of a large group of children with CP and CVI, describing their neurovisual profiles according to three different age subgroups (subgroup 1: infants 6 months-2 years; subgroup 2: pre-school age 3-5 years; subgroup 3: school age ≥ 6 years).MethodsWe enrolled 180 subjects (104 males, mean age 66 ± 42.6 months; range 6-192 months) with CP and CVI for the study. We carried out a demographic and clinical data collection, neurological examination, developmental or cognitive assessment, and a video-recorded visual function assessment including an evaluation of ophthalmological characteristics, oculomotor functions, and basic visual functions. In school-aged children, we also performed an evaluation of their cognitive-visual profiles.ResultsThere were signs of CVI in all the three subgroups. Subgroup 1 (62 children) and subgroup 2 (50 children) were different for fixation (p = 0.02), visual acuity (p = 0.03) and contrast sensitivity (p < 0.01), being more frequently impaired in younger children. Comparing subgroup 2 with subgroup 3 (68 children), the older children presented more frequently myopia (p = 0.02) while the younger ones esotropia (p = 0.02) and alteration in smooth pursuit (p = 0.03) and saccades (p < 0.01). Furthermore, fixation, smooth pursuit, visual acuity, contrast sensitivity and visual filed (p < 0.01) were more frequently impaired in younger children (subgroup 1) compared to the older ones. Multiple correspondence analysis (MCA) confirmed the different neurovisual profiles according to age: younger children with CP showed more signs of CVI compared to the older ones. 34 out of 68 children belonging to subgroup 3 underwent the cognitive visual evaluation; an impairment of cognitive visual skills was detected in 21 subjects.ConclusionYounger children with CP showed more signs of CVI compared to the older ones, likely for the physiological maturation of visual system and mechanisms of neuroplasticity. In this direction, we suggest an early neurovisual evaluation to detect any weak visual functions.
| S-EPMC8924515 | biostudies-literature
Project description:INTRODUCTION:Cerebral Visual Impairment (CVI) is the most common cause of visual impairment in children in the developed world and appears to be more prevalent in children with additional support needs (ASN). There is an urgent need for routine screening for CVI, particularly in children with ASN, however, current screening questionnaires for CVI have limited validation. The aim of this study was to evaluate two screening tools: the Five Questions and the CVI Questionnaire. Additionally, the distribution of CVI across neurodevelopmental disorders is unknown. This too was investigated. METHODS:An online survey was completed by 535 parents. The survey was advertised via social media, CVI websites and parent email systems of four schools. The survey comprised of the Five Questions, the CVI Questionnaire and additional questions regarding the child's diagnoses. Whether or not a child had a diagnosis of CVI and/or additional neurodevelopmental disorders was based on parental report. RESULTS:Based on parent reports, both the screening tools accurately screened for CVI diagnoses in children. The Five Questions and the CVI Questionnaire have construct validity (as determined through factor analysis), high internal consistency (as determined by Cronbach's alpha) and convergent validity (as determined by correlation analysis of the raw scores of each questionnaire). This study also highlights that among children with neurodevelopmental disorders, a large proportion have parent-reported CVI (23%-39%) and potential CVI (6.59-22.53%; as identified by the questionnaires). CONCLUSION:The current study demonstrates that the Five Questions and CVI Questionnaire have good convergent validity, internal consistency and a reliable factor structure and may therefore be suitable as screening tools. The study also highlights that reported or potential CVI is evident in a large proportion of children with neurodevelopmental disorders.
| S-EPMC6435113 | biostudies-literature
Project description:PurposeThis article provides recommendations for screening children aged 36 to younger than 72 months for eye and visual system disorders. The recommendations were developed by the National Expert Panel to the National Center for Children's Vision and Eye Health, sponsored by Prevent Blindness, and funded by the Maternal and Child Health Bureau of the Health Resources and Services Administration, United States Department of Health and Human Services. The recommendations describe both best and acceptable practice standards. Targeted vision disorders for screening are primarily amblyopia, strabismus, significant refractive error, and associated risk factors. The recommended screening tests are intended for use by lay screeners, nurses, and other personnel who screen children in educational, community, public health, or primary health care settings. Characteristics of children who should be examined by an optometrist or ophthalmologist rather than undergo vision screening are also described.ResultsThere are two current best practice vision screening methods for children aged 36 to younger than 72 months: (1) monocular visual acuity testing using single HOTV letters or LEA Symbols surrounded by crowding bars at a 5-ft (1.5 m) test distance, with the child responding by either matching or naming, or (2) instrument-based testing using the Retinomax autorefractor or the SureSight Vision Screener with the Vision in Preschoolers Study data software installed (version 2.24 or 2.25 set to minus cylinder form). Using the Plusoptix Photoscreener is acceptable practice, as is adding stereoacuity testing using the PASS (Preschool Assessment of Stereopsis with a Smile) stereotest as a supplemental procedure to visual acuity testing or autorefraction.ConclusionsThe National Expert Panel recommends that children aged 36 to younger than 72 months be screened annually (best practice) or at least once (accepted minimum standard) using one of the best practice approaches. Technological updates will be maintained at http://nationalcenter.preventblindness.org.
| S-EPMC4274336 | biostudies-literature
Project description:Cerebral visual impairment (CVI) is a major cause of low vision in children due to impairment in projection and/or interpretation of the visual input in the brain. Although acquired causes for CVI are well known, genetic causes underlying CVI are largely unidentified. DNAs of 25 patients with CVI and intellectual disability, but without acquired (eg, perinatal) damage, were investigated by whole-exome sequencing. The data were analyzed for de novo, autosomal-recessive, and X-linked variants, and subsequently classified into known, candidate, or unlikely to be associated with CVI. This classification was based on the Online Mendelian Inheritance in Man database, literature reports, variant characteristics, and functional relevance of the gene. After classification, variants in four genes known to be associated with CVI (AHDC1, NGLY1, NR2F1, PGAP1) in 5 patients (20%) were identified, establishing a conclusive genetic diagnosis for CVI. In addition, in 11 patients (44%) with CVI, variants in one or more candidate genes were identified (ACP6, AMOT, ARHGEF10L, ATP6V1A, DCAF6, DLG4, GABRB2, GRIN1, GRIN2B, KCNQ3, KCTD19, RERE, SLC1A1, SLC25A16, SLC35A2, SOX5, UFSP2, UHMK1, ZFP30). Our findings show that diverse genetic causes underlie CVI, some of which will provide insight into the biology underlying this disease process.
| S-EPMC4930090 | biostudies-literature
Project description:Background: There is limited research on brain lesions in children with cerebral visual impairment (CVI) of heterogeneous etiologies and according to associated subtyping and vision dysfunctions. This study was part of a larger project establishing data-driven subtypes of childhood CVI according to visual dysfunctions. Currently there is no consensus in relation to assessment, diagnosis and classification of CVI and more information about brain lesions may be of potential diagnostic value. Aim: This study aimed to investigate overall patterns of brain lesions and associations with level of visual dysfunction and to compare the patterns between the classification subgroups in children with CVI. Methods: School-aged children with CVI received ophthalmological and neuro-psychological/developmental assessments to establish CVI-related subtyping. Other pediatric information was collected from medical records. MRI scans were coded according to a semi-quantitative template including brain regions (right hemisphere, left hemisphere, visual pathways) and summed for total scores. Non-parametric analyses were conducted. Results: 28 children had clinical brain MRI scans available [44% of total sample, Group A (lower severity of visual dysfunctions) n = 16, Group B (higher severity) n = 12]. Total brain scores ranged between 0 and 18 (Group A mdn = 7, IQR = 0.8-10.0, Group B mdn = 10, IQR = 6.5-11.8) and were widespread across regions. 71 per cent had post-geniculate visual pathway damage. The median total brain and hemisphere scores of Group B were higher than subgroup A but differences did not reach statistical significance. No statistically significant associations were found between brain scores and vision variables (acuity, contrast sensitivity). Conclusion: This study found a spread of lesions across all regions on the brain scans in children with congenital CVI. The majority had damage in the postgeniculate visual pathways and visual cortex region suggesting this is an area of interest and potentially informative for diagnosis. However the subtyping classification did not show differences in number or region of lesions though the trend was higher toward Group B. This study confirms the complex diffuse and variable nature of brain lesions in children with congenital CVI, many of whom have other neurological impairments.
| S-EPMC8770951 | biostudies-literature
Project description:PurposeTo recommend a standardized approach for measuring progress toward national goals to improve preschool children's eye health.MethodsA multidisciplinary panel of experts reviewed existing measures and national vision-related goals during a series of face-to-face meetings and conference calls. The panel used a consensus process, informed by existing data related to delivery of eye and non-eye services to preschool children.ResultsCurrently, providers of vision screening and eye examinations lack a system to provide national- or state-level estimates of the proportion of children who receive either a vision screening or an eye examination. The panel developed numerator and denominator definitions to measure rates of children "who completed a vision screening in a medical or community setting using a recommended method, or received an eye examination by an optometrist or ophthalmologist at least once between the ages of 36 to <72 months." A separate measure for children with neurodevelopmental disorders and measures for eye examination and follow-up were also developed. The panel recommended that these measures be implemented at national, state, and local levels.ConclusionsStandardized performance measures that include all eye services received by a child are needed at state and national levels to measure progress toward improving preschool children's eye health.
| S-EPMC4274339 | biostudies-literature
Project description:This article provides a rationale for developing an integrated data system for recording vision screening and eye care follow-up outcomes in preschool-aged children. The recommendations were developed by the National Expert Panel to the National Center for Children's Vision and Eye Health at Prevent Blindness and funded by the Maternal and Child Health Bureau of the Health Resources and Services Administration, US Department of Health and Human Services. Guidance is provided regarding specific elements to be included, as well as the characteristics and architecture of such a data system. Vision screening for preschool-aged children is endorsed by many organizations concerned with children's health issues. Currently, there is a lack of data on the proportion of children screened and no effective system to ensure that children who fail screenings access appropriate comprehensive eye examinations and follow-up care.The expansion of currently existing, or developing integrated health information systems, which would include child-level vision screening data, as well as referral records and follow-up diagnosis and treatment, is consistent with the proposed national approach to an integrated health information system (National Health Information Infrastructure). Development of an integrated vision data system will enhance eye health for young children at three different levels: (1) the child level, (2) the health care provider level, and (3) an epidemiological level.It is critical that the end users, the professionals who screen children and the professionals who provide eye care, be involved in the development and implementation of the proposed integrated data systems. As essential stakeholders invested in ensuring quality eye care for children, this community of professionals should find increasing need and opportunities at local, state, and national levels to contribute to cooperative guidance for data system development.
| S-EPMC4274341 | biostudies-other
Project description:PurposeChildren with cerebral visual impairment (CVI) often have abnormal visual orienting behaviors due to impaired or damaged visual cortex. Alternatively, visual-cortical function is intact but visual information is not transformed downstream into an appropriate oculomotor output (visuomotor dysfunction). We examined visual, anatomic, and oculomotor assessments to distinguish visuomotor dysfunction from CVI associated with severely reduced visual-cortical response.MethodsWe reviewed the medical records from children with CVI having abnormal visual orienting behaviors, normal ocular examinations, and born near term. Relevant data were visual evoked potentials (VEPs), Teller card acuity, eye movements recorded by video-oculography (VOG), and neuroimaging (magnetic resonance imaging [MRI]) including diffusion tensor imaging (DTI) tractography.ResultsThirty subjects had visuomotor dysfunction based on a normal VEP; of these 33% had a normal MRI and 67% had white matter abnormalities associated with metabolic disease and/or decreased volume of brain parenchyma. VOG recordings showed smooth pursuit gains were uniformly reduced and saccades were dysmetric but followed the main sequence. Ten subjects had severe CVI based on VEPs at noise levels; visual acuities and MRI findings overlapped those of the visuomotor dysfunction group. Developmental delay, seizures, microcephaly, and hypotonia were common across all groups. All subjects with an abnormal conventional MRI had abnormal metrics on DTI tractography from the occipital lobe.ConclusionsA subset of patients with CVI have abnormal visual orienting behaviors despite a normal VEP (visuomotor dysfunction). A majority have abnormal white matter metrics on tractography suggesting a downstream defect in sensorimotor transformation. Clinically, visuomotor dysfunction is indistinguishable from severe CVI.
| S-EPMC8132015 | biostudies-literature