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Childhood represents a critical window for the emergence and treatment of mental health disorders, yet many are not being identified, or are identified too late to receive adequate intervention. This systematic review (Prospero registration: CRD42022299560) aimed to determine the effectiveness and acceptability of parent reported universal mental health screening (UMHS) to improve the early identification of children at-risk of mental health difficulties, and to identify barriers and enablers that may influence parental engagement. Six databases were searched in February 2022 for peer-reviewed, primary research. Studies conducted in targeted populations, evaluating psychometric properties, or focused on screening non-psychological problems were excluded. Ten studies examined parent reported (n = 3,464 parents) UMHS for children from birth to 18 years, suggesting an overall scarcity of research. Findings are presented in a table of study characteristics and a narrative summary of acceptability, effectiveness, barriers, and enablers. Quantitative findings indicated that parents generally support and accept UMHS. Research assessing effectiveness was limited, although two studies indicated increased referrals and referral adherence following positive screens. Confidentiality and stigma were commonly identified barriers. Quality assessment using the Mixed Methods Appraisal Tool indicated that studies varied in quality, meeting four to seven of the seven quality criteria. Understanding and addressing parent attitudes to UMHS across settings is necessary for the successful implementation of screening and improvement of child mental health outcomes. More high-quality research studies, including randomized controlled trials are therefore needed to examine the acceptability and effectiveness of UMHS for parents and their children.
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Tamizaje Masivo , Padres , Humanos , Niño , Padres/psicología , Trastornos Mentales/diagnóstico , Trastornos Mentales/prevención & control , Adolescente , Preescolar , Salud MentalRESUMEN
Albinism is a spectrum disorder causing foveal hypoplasia, nystagmus, and hypopigmentation of the iris and fundus along with other visual deficits, which can all impact vision. Albinism is also associated with amblyogenic factors which could affect monocular visual acuity. The foveal appearance in albinism can range from mild foveal hypoplasia to that which is indistinguishable from the peripheral retina. The appearance can be quickly and easily graded using the Leicester Grading System in the clinic. However, interquartile ranges of 0.3 logMAR for the grades associated with albinism limit the accuracy of the grading system in predicting vision. Here, we discuss the potential role of nystagmus presenting evidence that it may not be a major source of variability in the prediction of visual acuity. We also show that interocular differences in visual acuity are low in albinism despite high levels of amblyogenic factors indicating that active suppression of vision in one eye in albinism is uncommon.
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Albinismo , Humanos , Agudeza Visual , Fóvea Central , Fondo de Ojo , IrisRESUMEN
Purpose: This is the first systematic comparison of visual field (VF) deficits in people with albinism (PwA) and idiopathic infantile nystagmus (PwIIN) using static perimetry. We also compare best-corrected visual acuity (BCVA) and optical coherence tomography measures of the fovea, parafovea, and circumpapillary retinal nerve fiber layer in PwA. Methods: VF testing was performed on 62 PwA and 36 PwIIN using a Humphrey Field Analyzer (SITA FAST 24-2). Mean detection thresholds for each eye were calculated, along with quadrants and central measures. Retinal layers were manually segmented in the macular region. Results: Mean detection thresholds were significantly lower than normative values for PwA (-3.10 ± 1.67 dB, P << 0.0001) and PwIIN (-1.70 ± 1.54 dB, P < 0.0001). Mean detection thresholds were significantly lower in PwA compared to PwIIN (P < 0.0001) and significantly worse for left compared to right eyes in PwA (P = 0.0002) but not in PwIIN (P = 0.37). In PwA, the superior nasal VF was significantly worse than other quadrants (P < 0.05). PwIIN appeared to show a mild relative arcuate scotoma. In PwA, central detection thresholds were correlated with foveal changes in the inner and outer retina. VF was strongly correlated to BCVA in both groups. Conclusions: Clear peripheral and central VF deficits exist in PwA and PwIIN, and static VF results need to be interpreted with caution clinically. Since PwA exhibit considerably lower detection thresholds compared to PwIIN, VF defects are unlikely to be due to nystagmus in PwA. In addition to horizontal VF asymmetry, PwA exhibit both vertical and interocular asymmetries, which needs further exploration.
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Albinismo , Enfermedades Genéticas Ligadas al Cromosoma X , Nistagmo Congénito , Humanos , Campos Visuales , Escotoma/diagnóstico , Escotoma/etiología , RetinaRESUMEN
Purpose: Our primary aim was to compare adult full-field ERG (ffERG) responses in albinism, idiopathic infantile nystagmus (IIN), and controls. A secondary aim was to investigate the effect of within-subject changes in nystagmus eye movements on ffERG responses. Methods: Dilated Ganzfeld flash ffERG responses were recorded using DTL electrodes under conditions of dark (standard and dim flash) and light adaptation in 68 participants with albinism, 43 with IIN, and 24 controls. For the primary aim, the effect of group and age on ffERG responses was investigated. For the secondary aim, null region characteristics were determined using eye movements recorded prior to ffERG recordings. ffERG responses were recorded near and away from the null regions of 18 participants also measuring the success rate of recordings. Results: For the primary aim, age-adjusted photopic a- and b-wave amplitudes were consistently smaller in IIN compared with controls (P < 0.0001), with responses in both groups decreasing with age. In contrast, photopic a-wave amplitudes increased with age in albinism (P = 0.0035). For the secondary aim, more intense nystagmus significantly reduced the success rate of measurable responses. Within-subject changes in nystagmus intensity generated small, borderline significant differences in photopic b-wave peak times and a-and b-wave amplitudes under scotopic conditions with standard flash. Conclusions: Age-adjusted photopic ffERG responses are significantly reduced in IIN adding to the growing body of evidence of retinal abnormalities in IIN. Differences between photopic responses in albinism and controls depend on age. Success at obtaining ffERG responses could be improved by recording responses at the null region.
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Albinismo , Enfermedades Genéticas Ligadas al Cromosoma X , Nistagmo Congénito , Nistagmo Patológico , Adulto , Humanos , Nistagmo Patológico/diagnóstico , Movimientos OcularesRESUMEN
Nipah virus (NiV) poses a significant threat to human and livestock populations across South and Southeast Asia. Vaccines are required to reduce the risk and impact of spillover infection events. Pigs can act as an intermediate amplifying host for NiV and, separately, provide a preclinical model for evaluating human vaccine candidate immunogenicity. The aim of this study was therefore to evaluate the immunogenicity of an mRNA vectored NiV vaccine candidate in pigs. Pigs were immunized twice with 100 µg nucleoside-modified mRNA vaccine encoding soluble G glycoprotein from the Malaysia strain of NiV, formulated in lipid nanoparticles. Potent antigen-binding and virus neutralizing antibodies were detected in serum following the booster immunization. Antibody responses effectively neutralized both the Malaysia and Bangladesh strains of NiV but showed limited neutralization of the related (about 80% amino acid sequence identity for G) Hendra virus. Antibodies were also capable of neutralizing NiV glycoprotein mediated cell-cell fusion. NiV G-specific T cell cytokine responses were also measurable following the booster immunization with evidence for induction of both CD4 and CD8 T cell responses. These data support the further evaluation of mRNA vectored NiV G as a vaccine for both pigs and humans.
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Anticuerpos Neutralizantes , Anticuerpos Antivirales , Infecciones por Henipavirus , Virus Nipah , Vacunas Virales , Animales , Virus Nipah/inmunología , Virus Nipah/genética , Porcinos , Infecciones por Henipavirus/prevención & control , Infecciones por Henipavirus/inmunología , Anticuerpos Neutralizantes/inmunología , Anticuerpos Neutralizantes/sangre , Vacunas Virales/inmunología , Vacunas Virales/administración & dosificación , Anticuerpos Antivirales/sangre , Anticuerpos Antivirales/inmunología , Enfermedades de los Porcinos/inmunología , Enfermedades de los Porcinos/prevención & control , Enfermedades de los Porcinos/virología , ARN Mensajero/genética , ARN Mensajero/inmunología , Inmunogenicidad Vacunal , Inmunización Secundaria , Citocinas/inmunología , Vacunas Sintéticas/inmunología , Liposomas , NanopartículasRESUMEN
BACKGROUND/AAIMS: Congenital stationary night blindness (CSNB) is an inherited retinal disease that is often associated with high myopia and can be caused by pathological variants in multiple genes, most commonly CACNA1F, NYX and TRPM1. High myopia is associated with retinal degeneration and increased risk for retinal detachment. Slowing the progression of myopia in patients with CSNB would likely be beneficial in reducing risk, but before interventions can be considered, it is important to understand the natural history of myopic progression. METHODS: This multicentre, retrospective study explored CSNB caused by variants in CACNA1F, NYX or TRPM1 in patients who had at least 6 measurements of their spherical equivalent of refraction (SER) before the age of 18. A mixed-effect model was used to predict progression of SER overtime and differences between genotypes were evaluated. RESULTS: 78 individuals were included in this study. All genotypes showed a significant myopic predicted SER at birth (-3.076D, -5.511D and -5.386D) for CACNA1F, NYX and TRPM1 respectively. Additionally, significant progression of myopia per year (-0.254D, -0.257D and -0.326D) was observed for all three genotypes CACNA1F, NYX and TRPM1, respectively. CONCLUSIONS: Patients with CSNB tend to be myopic from an early age and progress to become more myopic with age. Patients may benefit from long-term myopia slowing treatment in the future and further studies are indicated. Additionally, CSNB should be considered in the differential diagnosis for early-onset myopia.