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The Visual Acuity Outcome and Relevant Factors Affecting Visual Improvement in Pediatric Sporadic Chiasmatic-Hypothalamic Glioma Patients Who Received Surgery.

ABSTRACT: Background: The role and effectiveness of primary surgical treatment for sporadic chiasmatic-hypothalamic glioma (CHG) are not clear. The present study was to describe sporadic CHG visual acuity (VA) outcomes after surgery and to analyze the relevant factors affecting VA improvement. Methods: Forty-five pediatric sporadic CHG patients who met the inclusion criteria were included in a retrospective study. All patients received primary intratumor partial resection. Disease characteristics, treatment strategies, complications, and VA outcome were analyzed. Univariate and multivariate analyses were performed to identify relevant factors of VA improvement. Receiver operating characteristic (ROC) analysis was performed to evaluate the predictive accuracy of measurement indexes. Results: There were 77 eyes of 45 children suffering from various levels of VA impairment before surgical treatment, and only 13 eyes had normal vision. Patients with resection extents >70, 50-70, and <50% accounted for 26.67, 24.44, and 48.89%, respectively. The percentages of VA maintained and deteriorated in normal vision eyes were 61.54 and 38.46%. The percentages of VA improved, maintained, and deteriorated in visually impaired eyes after surgery were 29.87, 45.45, and 24.68%, respectively. There was a positive correlation between the IVA level and VA improvement. There was no significant difference in VA improvement between the different resection extents. Blindness occurred in ~4.4%. Approximately 11.1% of the children had complications that affected quality of life, which correlated with resection extent. IVA and tumor size were correlated with VA improvement. The AUC for IVA + tumor size predicting VA improvement was 0.831. The cutoff points for IVA level and tumor volume were 4.5 and 43.50 cm3, respectively. Conclusions: IVA and tumor size were correlated with VA improvement after primary intratumor partial resection. Children with IVA ? level 5 were more likely to achieve visual improvement after decompression surgery, but decompression had limited effectiveness on vision improvement in patients with tumor volumes ? 43.50 cm3. Performing resections < 50% was safe and did not reduce the effect of decompression to improve VA.

SUBMITTER: Liao C

PROVIDER: S-EPMC7466562 | biostudies-literature | 2020

REPOSITORIES: biostudies-literature

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The Visual Acuity Outcome and Relevant Factors Affecting Visual Improvement in Pediatric Sporadic Chiasmatic-Hypothalamic Glioma Patients Who Received Surgery.

Liao Chihyi C Zhang Heng H Liu Zhiming Z Han Zhe Z Li Chunde C Gong Jian J Liu Wei W Ma Zhenyu Z Tian Yongji Y

Frontiers in neurology 20200819

<b>Background:</b> The role and effectiveness of primary surgical treatment for sporadic chiasmatic-hypothalamic glioma (CHG) are not clear. The present study was to describe sporadic CHG visual acuity (VA) outcomes after surgery and to analyze the relevant factors affecting VA improvement. <b>Methods:</b> Forty-five pediatric sporadic CHG patients who met the inclusion criteria were included in a retrospective study. All patients received primary intratumor partial resection. Disease characteri ...[more]

PMID: 32973646

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Project description:ImportanceOptic neuritis (ON) in children is uncommon. There are limited prospective data for visual acuity (VA) outcomes, associated diseases, and neuroimaging findings. Prospective data from a large sample would be useful for counseling families on treatment decisions and prognosis.ObjectiveTo prospectively study children with a first episode of ON, describe VA after 6 months, and ascertain the network's (Pediatric Eye Disease Investigator Group and Neuro-Ophthalmology Research Disease Investigator Consortium) ability to enroll pediatric patients with ON prospectively.Design, setting, and participantsThis nonrandomized cohort study was conducted from September 20, 2016, to July 20, 2018, at 23 sites in the United States and Canada in pediatric ophthalmology or neuro-ophthalmology clinics. A total of 44 children (aged 3-15 years) presented with a first episode of ON (visual loss, pain on eye movements, or both) within 2 weeks of symptom onset and at least 1 of the following in the affected eye: a distance high-contrast VA (HCVA) deficit of at least 0.2 logMAR below age-based norms, diminished color vision, abnormal visual field, or optic disc swelling. Exclusion criteria included preexisting ocular abnormalities or a previous episode of ON.Main outcomes and measuresPrimary outcomes were monocular HCVA and low-contrast VA at 6 months. Secondary outcomes were neuroimaging, associated diagnoses, and antibodies for neuromyelitis optica and myelin oligodendrocyte glycoprotein.ResultsA total of 44 children (mean age [SD], 10.2 [3.5] years; 26 boys [59%]; 23 White individuals [52%]; 54 eyes) were enrolled in the study. Sixteen patients (36%) had bilateral ON. Magnetic resonance imaging revealed white matter lesions in 23 children (52%). Of these children, 8 had myelin oligodendrocyte glycoprotein-associated demyelination (18%), 7 had acute disseminated encephalomyelitis (16%), 5 had multiple sclerosis (11%), and 3 had neuromyelitis optica (7%). The baseline mean HCVA was 0.95 logMAR (20/200), which improved by a mean 0.76 logMAR (95% CI, 0.54-0.99; range, -0.70 to 1.80) to 0.12 logMAR (20/25) at 6 months. The baseline mean distance low-contrast VA was 1.49 logMAR (20/640) and improved by a mean 0.72 logMAR (95% CI, 0.54-0.89; range, -0.20 to 1.50) to 0.73 logMAR (20/100) at 6 months. Baseline HCVA was worse in younger participants (aged <10 years) with associated neurologic autoimmune diagnoses, white matter lesions, and in those of non-White race and non-Hispanic ethnicity. The data did not suggest a statistically significant association between baseline factors and improvement in HCVA.Conclusions and relevanceThe study network did not reach its targeted enrollment of 100 pediatric patients with ON over 2 years. This indicates that future treatment trials may need to use different inclusion criteria or plan a longer enrollment period to account for the rarity of the disease. Despite poor VA at presentation, most children had marked improvement by 6 months. Associated neurologic autoimmune diagnoses were common. These findings can be used to counsel families about the disease.

Project description:ContextIndividuals treated for pediatric craniopharyngioma, a rare, grade 1 brain tumor, frequently develop hypothalamic obesity, a complication often recalcitrant to intervention. Although hypothalamic obesity is known to adversely impact quality of life, less is known about how caregivers and patients experience this condition.ObjectiveOur goal was to examine the approaches that families take towards weight management and the impact on social function in individuals with craniopharyngioma and obesity. Individuals with craniopharyngioma without obesity were included as a comparison.Subjects and methodsAdult caregivers of children <18y with craniopharyngioma completed a web-based survey posted by a patient advocacy organization between February and July 2020. Questions related to the child's diagnosis, medications, lifestyle modifications, and social function along with research priorities. Descriptive statistics were generated. Linear regression was used to assess the independent effects of obesity and other covariates on social function.ResultsOf 106 respondents, 60 (57%) reported their child had obesity at the time of survey completion. In contrast, only 6 (5.7%) had obesity prior to craniopharyngioma diagnosis. A majority (92%) of those with obesity had tried limiting calories or carbohydrates; 31% and 69% found these helpful, respectively. Thirty-eight percent had tried weight loss medications (stimulants, metformin, GLP1R-agonists, and topiramate) and 48% found at least one helpful. Both stimulant and anti-depressant use were reported more frequently with obesity. An index (T-score) reflecting social function was lower in the cohort than a population reference, 41 (SD 11) vs. 50 (SD 10), p<0.001. In a linear model, both older age and obesity were independently associated with greater social impairment. Ninety-four percent of respondents caring for a child with obesity (and 79% of all respondents) identified "improving treatments and prevention for hypothalamic obesity" as a key research priority.ConclusionsOnly a minority of individuals with hypothalamic obesity had trialed medication, even though many reported that lifestyle modification was inadequate. Furthermore, social function was significantly impaired overall in survivors compared to a reference cohort, and even more so in individuals with obesity. These findings highlight the opportunity to improve social functioning as an additional potential benefit of improved treatments for hypothalamic obesity.

Project description:BackgroundVisual outcome is one of the main issues in the treatment of optic pathway glioma in childhood. Although the prognostic factors of low vision have been discussed extensively, no reliable indicators for visual loss exist. Therefore, we aimed to define initial and evolving factors associated with long-term vision loss.MethodsWe conducted a multicenter historical cohort study of children treated in France with up-front BB-SFOP chemotherapy between 1990 and 2004. Visual acuity performed at the long-term follow-up visit or within 6 months prior was analyzed. Logistic regression analysis was used to estimate the effects of clinical and radiological factors on long-term visual outcome.FindingsOf the 180 patients in the cohort, long-term visual acuity data were available for 132 (73.3%) patients (median follow-up: 14.2 years; range: 6.1-25.6). At the last follow-up, 61/132 patients (46.2%) had impaired vision, and 35 of these patients (57.3%) were partially sighted or blind. Multivariate analysis showed that factors associated with a worse prognosis for long-term visual acuity were an age at diagnosis of < 1 year (OR 3.5 [95% CI: 1.1-11.2], p = 0.04), tumor extent (OR 4.7 [95% CI: 1.2-19.9], p = 0.03), intracranial hypertension requiring one or more surgical procedures (OR 5.6 [95% CI: 1.8-18.4], p = 0.003), and the need for additional treatment after initial BB-SFOP chemotherapy (OR 3.5 [95% CI: 1.1-11.9], p = 0.04). NF1 status did not appear as a prognostic factor, but in non-NF1 patients, a decrease in tumor volume with contrast enhancement after BB-SFOP chemotherapy was directly associated with a better visual prognosis (OR 0.8 [95% CI: 0.8-0.9], p = 0.04).InterpretationOur study confirms that a large proportion of children with optic pathway glioma have poor long-term outcomes of visual acuity. These data suggest new prognostic factors for visual acuity, but these results need to be confirmed further by large- and international-scale studies.

Project description:ImportanceThere is substantial socioeconomic and individual burden from uncorrected refractive error (URE) and chronic ocular disease. Understanding the association of visual acuity (VA) reduction with URE and the adults most likely to benefit from refraction may help support clinical decision-making in ophthalmologic care and maximize patient outcomes.ObjectivesTo assess the magnitude of VA improvement associated with URE among adults under ophthalmic care who obtain low vision rehabilitation (LVR) services and identify the characteristics of the patients who are most likely to experience improvement.Design, setting, and participantsThis retrospective case series assessed patients 20 years or older who were new to the LVR clinics from August 1, 2013, to December 31, 2015, and who had habitual VA between 20/40 and counting fingers (not including) and underwent refraction. Data analysis was performed from April 4, 2018, to December 20, 2019.ExposuresPatient demographics and clinical data, including habitual and refraction VA, refraction, and disease diagnosis. Habitual VA was categorized as mild (VA worse than 20/40 and at least 20/60), moderate (VA worse than 20/60 and better than 20/200), severe (VA 20/200 or worse and better than 20/500), and profound (VA 20/500 or worse) vision impairment (VI).Main outcomes and measuresAt least 2-line VA improvement and any VA improvement (≥1-line) by refraction.ResultsAmong the 2923 patients new to LVR clinics, 1773 (mean [SD] age, 70 [18.2] years; 1069 [60.3%] female) were included in these analyses. The mean habitual VA was 20/100 (mean [SD], 0.67 [0.36] logMAR). At least 2-line improvement was observed in 493 patients (27.8%), and any VA improvement was seen in 1023 patients (57.7%). At least 2-line improvement was observed in 54 patients (34.8%) with corneal disorders and was more likely seen among patients aged 40 to <65 years compared with those aged 20 to <40 years (odds ratio [OR], 1.57; 95% CI, 1.02-2.41), African American patients compared with white patients (OR, 1.41; 95% CI, 1.08-1.85), or patients with moderate VI compared with mild VI (OR, 1.36; 95% CI, 1.07-1.72).Conclusions and relevanceThe findings suggest that URE is prevalent among patients with ocular disease and accessing LVR and that refractive evaluation should be considered for patients with ocular disease and reduced VA, especially working-age adults aged 40 to <65 years, African American patients, and those with moderate VI.

Dataset Information

The Visual Acuity Outcome and Relevant Factors Affecting Visual Improvement in Pediatric Sporadic Chiasmatic-Hypothalamic Glioma Patients Who Received Surgery.

Publications

The Visual Acuity Outcome and Relevant Factors Affecting Visual Improvement in Pediatric Sporadic Chiasmatic-Hypothalamic Glioma Patients Who Received Surgery.

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