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1.
Neurol Clin ; 42(2): 433-471, 2024 May.
Article in English | MEDLINE | ID: mdl-38575259

ABSTRACT

Pseudotumor cerebri syndrome is a syndrome of increased cerebrospinal fluid pressure without ventriculomegaly, mass lesion, or meningeal abnormality. It is either primary (idiopathic intracranial hypertension, IIH) or secondary. A secondary cause is unlikely when adhering to the diagnostic criteria. Permanent visual loss occurs if undetected or untreated, and the associated headaches may be debilitating. Fulminant disease may result in blindness despite aggressive treatment. This study addresses the diagnosis and management of IIH including new insights into the pathobiology of IIH, updates in therapeutics and causes of overdiagnosis.


Subject(s)
Intracranial Hypertension , Papilledema , Pseudotumor Cerebri , Humans , Pseudotumor Cerebri/diagnosis , Pseudotumor Cerebri/etiology , Pseudotumor Cerebri/therapy , Intracranial Hypertension/complications , Headache/diagnosis , Headache/etiology , Headache/therapy , Vision Disorders/therapy , Syndrome , Papilledema/complications , Papilledema/diagnosis
2.
Am J Case Rep ; 25: e942833, 2024 Mar 20.
Article in English | MEDLINE | ID: mdl-38504435

ABSTRACT

BACKGROUND Carotid-cavernous fistula (CCF) is a rare, atypical vascular shunt between the carotid arterial system and the venous channels of the cavernous sinus, classified according to the shunt's anatomy, by etiology (resulting from trauma or occurring spontaneously), or by hemodynamic characteristics (such as low- or high-flow fistulas). CASE REPORT A 62-year-old female patient with poorly controlled arterial hypertension presented with bilateral periorbital edema, conjunctival chemosis, ophthalmoplegia, diplopia, and diminished visual acuity. On magnetic resonance angiography (MRA), abnormal arterial flow along the cavernous sinuses was noted, suggestive of bilateral CCF. The diagnosis of indirect dural low-flow CCF (Barrow Type D) was later confirmed by digital subtraction angiography, with feeding arteries from intracavernous internal carotid artery branches, and meningeal branches of the external carotid artery, draining bilaterally to ophthalmic veins, the intracavernous sinus, and the inferior petrosal sinus. The patient was successfully treated with endovascular embolization. At 7-month follow-up, no residual arteriovenous shunting was detected. This case highlights the importance of non-invasive radiological methods for CCF, and presents rarely published radiological findings of bilateral Type-D dural CCFs on 3-dimensional time-of-flight MRA with post-treatment MRA follow-up. CONCLUSIONS Regardless of the patient's history of possible trauma, a patient presenting with bilateral periorbital edema, conjunctival chemosis, ophthalmoplegia, diplopia, and diminished visual acuity should have a spontaneous bilateral CCF investigated to prevent delayed treatment. Experienced neuroradiologists are needed to accurately detect indirect CCF, since this condition often does not demonstrate classic symptoms.


Subject(s)
Carotid-Cavernous Sinus Fistula , Embolization, Therapeutic , Fistula , Ophthalmoplegia , Female , Humans , Middle Aged , Magnetic Resonance Angiography/adverse effects , Diplopia/etiology , Carotid-Cavernous Sinus Fistula/diagnostic imaging , Carotid-Cavernous Sinus Fistula/therapy , Fistula/therapy , Embolization, Therapeutic/methods , Vision Disorders/therapy , Edema
3.
BMJ Open Ophthalmol ; 9(1)2024 Feb 23.
Article in English | MEDLINE | ID: mdl-38395461

ABSTRACT

OBJECTIVES: To assess needs and views regarding eye health and empowerment from craftswomen's perspectives to develop a theory of change (ToC) for a women-targeted eyecare programme. MATERIAL AND METHODS: Eighteen stakeholders participated in a 2-day consultation workshop in Zanzibar. The composition was (1) 15 women and 3 men; (2) Unguja (n=8), Pemba (n=6) and Tanzania mainland (n=4) and (3) craftswomen (n=14) and governmental stakeholders (n=4). Thematic analysis determined the craftswomen's needs and views regarding eye health and empowerment and subsequently inputs, activities, outputs, outcomes and impact to develop the programme's initial ToC. In refining the initial ToC, we used insights from a qualitative study suggesting that improved near vision is perceived by craftswomen as a potential source of empowerment across economic, psychological, social, political and educational dimensions. RESULTS: The eye conditions experienced by the craftswomen were eye irritation caused by foreign bodies, the need for near spectacles and other eye morbidities. They were advised by the cooperatives to visit eye health centres for treatment. The main barriers to accessing services were inaccessibility and unaffordability of eye services and a lack of eye health knowledge and practices. Nineteen subthemes on women empowerment (economic n=4, social n=4, psychological n=6, education n=2 and political n=3) were obtained. We created a ToC on how investing in improving craftswomen near vision could achieve empowerment. CONCLUSION: The participants provided insights into their needs and how they would like the eyecare programme to be implemented and how they see they could be empowered in the process.


Subject(s)
Empowerment , Vision Disorders , Male , Humans , Female , Tanzania , Vision Disorders/therapy , Vision, Ocular , Qualitative Research
4.
Pharmacoeconomics ; 42(3): 275-299, 2024 Mar.
Article in English | MEDLINE | ID: mdl-37971639

ABSTRACT

BACKGROUND: Evidence on the socioeconomic burden associated with childhood visual impairment, severe visual impairment and blindness (VI/SVI/BL) is needed to inform economic evaluations of existing and emerging interventions aimed at protecting or improving vision. This study aimed to evaluate the quantity and quality of literature on resource use and/or costs associated with childhood VI/SVI/BL disorders. METHODS: PubMed, Web of Science (Ovid), the National Health Service (NHS) Economic Evaluation Database and grey literature were searched in November 2020. The PubMed search was rerun in February 2022. Original articles reporting unique estimates of resource use or cost data on conditions resulting in bilateral VI/SVI/BL were eligible for data extraction. Quality assessment (QA) was undertaken using the Drummond checklist adapted for cost-of-illness (COI) studies. RESULTS: We identified 31 eligible articles, 27 from the peer-reviewed literature and four from the grey literature. Two reported on resource use, and 29 reported on costs. Cerebral visual impairment and optic nerve disorders were not examined in any included studies, whereas retinopathy of prematurity was the most frequently examined condition. The quality of studies varied, with economic evaluations having higher mean QA scores (82%) compared to COI studies (77%). Deficiencies in reporting were seen, particularly in the clinical definitions of conditions in economic evaluations and a lack of discounting and sensitivity analyses in COI studies. CONCLUSIONS: There is sparse literature on resource use or costs associated with childhood visual impairment disorders. The first step in addressing this important evidence gap is to ensure core visual impairment outcomes are measured in future randomised control trials of interventions as well as cohort studies and are reported as a discrete health outcome.


Subject(s)
Cost of Illness , State Medicine , Infant, Newborn , Humans , Child , Infant, Premature , Cost-Benefit Analysis , Vision Disorders/therapy
5.
Curr Pain Headache Rep ; 28(2): 55-63, 2024 Feb.
Article in English | MEDLINE | ID: mdl-38079073

ABSTRACT

PURPOSE OF REVIEW: Visual snow (VS) involves visualization of innumerable dots throughout the visual field, sometimes resembling "TV static." Patients who experience this symptom may also have additional visual symptoms (e.g., photophobia, palinopsia, floaters, and nyctalopia) with a pattern now defined as visual snow syndrome (VSS). This manuscript describes both VS and VSS in detail and provides an updated review on the clinical features, pathophysiology, and optimal management strategies for these symptoms. RECENT FINDINGS: VS/VSS may be primary or secondary to a variety of etiologies, including ophthalmologic or brain disorders, systemic disease, and medication/hallucinogen exposure. Evaluation involves ruling out secondary causes and mimics of VS. Increasing evidence suggests that VSS is a widespread process extending beyond the visual system. Pathophysiology may involve cortical hyperexcitability or dysfunctional connectivity of thalamocortical or attention/salience networks. VSS is typically a benign, non-progressive syndrome and can be managed with non-medicine strategies. Though no medication provides complete relief, some may provide partial improvement in severity of VS.


Subject(s)
Migraine with Aura , Perceptual Disorders , Humans , Migraine with Aura/diagnosis , Vision Disorders/diagnosis , Vision Disorders/therapy , Perceptual Disorders/complications , Visual Fields
6.
Am J Ophthalmol ; 257: 113-128, 2024 Jan.
Article in English | MEDLINE | ID: mdl-37716450

ABSTRACT

PURPOSE: To assess longitudinal relationships among visual function and anatomical measures of gene therapy in G11778A Leber hereditary optic neuropathy (LHON). DESIGN: Phase 1 clinical trial. METHODS: This was a single-institution study of patients with G11778A LHON. Patients with chronic bilateral visual loss >12 months (group 1, n = 11), acute bilateral visual loss <12 months (group 2, n = 9), or unilateral visual loss (group 3, n = 8) were administered unilateral intravitreal AAV2(Y444,500,730F)-P1ND4v2 injection with low, medium, high, and higher doses to worse eye for groups 1 and 2 and better eye for group 3. Oucome measures were best-corrected visual acuity (BCVA), visual field mean deviation (VF MD), steady-state pattern electroretinogram (SS-PERG), optical coherence tomography (OCT) retinal nerve fiber layer (RNFL) thickness and ganglion cell+inner plexiform layer (GCIPL) thickness, and National Eye Institute Visual Function Questionnaire (NEI-VFQ-25) scores. Mean follow-up was 33.6 months (range = 18-36 months). RESULTS: Baseline SS-PERG amplitude was much reduced in both eyes of all groups including asymptomatic eyes of group 3, and showed no appreciable changes irrespective of disease stage and treatment. Significant and progressive GCIPL and RNFL thinning occurred in all eyes; BCVA and VF MD fluctuated in treated and fellow eyes, with some eyes having modest improvement that may be related to natural history or to gene therapy. Mean NEI-VFQ-25 scores declined in group 3 subjects (P = .023), CONCLUSION: Asymptomatic eyes in LHON patients with unilateral visual loss may be beyond the window of effective neuroprotection given reduced GCIPL and SS-PERG. Randomization of patients to an untreated control group would help to assess treatment effect by accounting for variable natural history. NOTE: Publication of this article is sponsored by the American Ophthalmological Society.


Subject(s)
Optic Atrophy, Hereditary, Leber , Humans , Genetic Therapy , Optic Atrophy, Hereditary, Leber/genetics , Optic Atrophy, Hereditary, Leber/therapy , Retinal Ganglion Cells/physiology , Tomography, Optical Coherence/methods , Vision Disorders/therapy , Visual Acuity , Visual Fields
7.
Pediatr Neurol ; 151: 131-137, 2024 Feb.
Article in English | MEDLINE | ID: mdl-38157718

ABSTRACT

BACKGROUND: The majority of studies have investigated neurodevelopmental outcomes, whereas visual impairment is less explored in children with a history of neonatal (hypoxic-ischemic) encephalopathy. Our aim was to perform a detailed neurological and visual assessment and also to investigate the presence of cerebral visual impairment in infants and toddlers with neonatal encephalopathy. METHODS: Thirty participants with a history of neonatal encephalopathy, who had been hospitalized for therapeutic hypothermia, underwent a detailed neurological examination at age five to 36 months. Age-matched, 30 healthy children were also enrolled as a control group. All children in the study and control groups received neurological and a comprehensive ophthalmologic examination, including visual field and visual acuity. Presence of cerebral visual impairment was also evaluated clinically. RESULTS: Rates of cerebral palsy, severe motor impairment, cognitive impairment, epilepsy, and cerebral visual impairment were found to be 20%, 10%, 15.3%, 10%, and 20%, respectively. When compared with healthy controls, oculomotor functions, pupillary light response, refractive parameters, anterior/posterior segment examinations, ocular visual impairment rates, and last, visual acuities were found similar. However, we found a statistically significant increase in visual field defects in our study group. CONCLUSIONS: It could be better to perform a comprehensive ophthalmologic examination including visual field, visual acuity, and oculomotor functions by a pediatric ophthalmologist to accurately diagnose neurovisual deficits in infants following therapeutic hypothermia. Early identification and rehabilitation of the visual deficits might improve the neurodevelopment in these children.


Subject(s)
Cerebral Palsy , Hypothermia, Induced , Hypoxia-Ischemia, Brain , Infant, Newborn, Diseases , Infant, Newborn , Infant , Humans , Child, Preschool , Child , Hypoxia-Ischemia, Brain/complications , Hypoxia-Ischemia, Brain/therapy , Infant, Newborn, Diseases/therapy , Cerebral Palsy/therapy , Vision Disorders/etiology , Vision Disorders/therapy
8.
Curr Biol ; 33(19): R1019-R1036, 2023 10 09.
Article in English | MEDLINE | ID: mdl-37816323

ABSTRACT

The retina is a part of the brain that sits at the back of the eye, looking out onto the world. The first neurons of the retina are the rod and cone photoreceptors, which convert changes in photon flux into electrical signals that are the basis of vision. Rods and cones are frequent targets of heritable neurodegenerative diseases that cause visual impairment, including blindness, in millions of people worldwide. This review summarizes the diverse genetic causes of inherited retinal degenerations (IRDs) and their convergence onto common pathogenic mechanisms of vision loss. Currently, there are few effective treatments for IRDs, but recent advances in disparate areas of biology and technology (e.g., genome editing, viral engineering, 3D organoids, optogenetics, semiconductor arrays) discussed here enable promising efforts to preserve and restore vision in IRD patients with implications for neurodegeneration in less approachable brain areas.


Subject(s)
Retina , Retinal Degeneration , Humans , Retina/physiology , Vision, Ocular , Retinal Cone Photoreceptor Cells/physiology , Retinal Degeneration/genetics , Retinal Degeneration/therapy , Retinal Degeneration/pathology , Vision Disorders/therapy , Vision Disorders/pathology , Blindness/genetics , Blindness/therapy
9.
Dtsch Arztebl Int ; 120(44): 747-753, 2023 Nov 03.
Article in English | MEDLINE | ID: mdl-37656479

ABSTRACT

BACKGROUND: In this systematic review, we address the question whether children and adolescents with developmental visual disorders benefit from computer-assisted visual training. METHODS: Systematic literature searches were carried out in three bibliographic databases (initial search in October 2021) and trial registries. Included were randomized controlled trials that evaluated the efficacy of computer-assisted visual training in children and adolescents with developmental visual disorders in comparison to no training, sham training, or conservative treatment. RESULTS: The inclusion criteria were met by 17 trials (with a total of 1323 children and adolescents) focusing on binocular or monocular computer-assisted visual training for the treatment of amblyopia. In these trials, visual training was carried out for 2 to 24 weeks, either as "stand alone" therapy or in addition to occlusion therapy. Six trials showed a statistically significant difference in favor of the visual training for the outcome "best corrected visual acuity of the amblyopic eye." However, this difference was small and mostly below the threshold of clinical relevance of -0.05 logMAR (equivalent to an improvement of 0.5 lines on the eye chart, or 2.5 letters per line). Only few data were available for the outcomes "binocular vision" and "adverse events"; the differences between the groups were similarly small. CONCLUSION: The currently available data do not permit any firm conclusions regarding the efficacy of visual training in children and adolescents with amblyopia. Moreover, treatment adherence was often insufficient and the treatment durations in the trials was relatively short. No results from randomized trials have yet been published with respect to other developmental visual disorders (refractive errors, strabismus).


Subject(s)
Amblyopia , Refractive Errors , Child , Humans , Adolescent , Amblyopia/therapy , Visual Acuity , Vision Disorders/diagnosis , Vision Disorders/therapy , Computers , Randomized Controlled Trials as Topic
10.
Patient Educ Couns ; 116: 107932, 2023 11.
Article in English | MEDLINE | ID: mdl-37566948

ABSTRACT

OBJECTIVES: Patient education and health literacy aim to enhance understanding of health and self-care for optimal health outcomes. However, the attention towards populations with visual impairments requiring specialized accommodations for improved health literacy and healthcare access appears to be insufficient METHODS: In an effort to bridge this gap, we conducted a scoping review focusing on health literacy studies undertaken specifically for individuals with visual impairments. RESULTS: We encapsulate the main findings and constraints of preceding studies and deliberate on the influence of health literacy research for individuals with visual impairments on healthcare inequalities and health disparities. CONCLUSIONS: A health-literate approach, combined with an accessible healthcare environment, can serve as a catalyst to motivate individuals with visual impairments to actively engage in their self-care practices. PRACTICE IMPLICATIONS: It is of utmost urgency to develop and validate a health literacy assessment tool for visually impaired individuals, and to utilize it for providing healthcare interventions as well as health education.


Subject(s)
Health Literacy , Humans , Health Services Accessibility , Healthcare Disparities , Health Education , Vision Disorders/therapy
11.
Neurol Neurochir Pol ; 57(4): 392-396, 2023.
Article in English | MEDLINE | ID: mdl-37501541

ABSTRACT

AIM OF THE STUDY: Ophthalmological symptoms are common in patients with Parkinson's Disease (PD) and can be evaluated by the Visual Impairment in Parkinson's Disease Questionnaire (VIPD-Q). This study aimed to assess the prevalence of ophthalmological symptoms in PD depending on the type of treatment used i.e. pharmacological or subthalamic nucleus deep brain stimulation (STN-DBS). MATERIAL AND METHODS: We performed a cross-sectional study. The data was gathered from a VIPD-Q and from medical records. Patients with PD were divided into two groups based on the type of treatment - pharmacological (control group, CG) (39 patients) or STN-DBS (40 patients). RESULTS: The great majority of patients - 72 (91.1%) - experienced an ophthalmological symptom. The prevalence of three symptoms differed significantly between the groups. A burning sensation or a gritty feeling in the eyes occurred more often in patients in the STN-DBS group (40.0% vs. 15.4%; p = 0.015). On the other hand, the inability to read plain text on a coloured or grey background and problems with rapid changes of light intensity were more common in the CG group (38.5% vs. 15.0%, p = 0.018 and 28.2% vs. 10.0%, p = 0.039, respectively). CONCLUSIONS AND CLINICAL IMPLICATIONS: The prevalence of ophthalmological symptoms in PD is high. Despite significant differences in the three symptoms, the overall prevalence of ophthalmological clinical features was similar in the evaluated groups.


Subject(s)
Deep Brain Stimulation , Parkinson Disease , Subthalamic Nucleus , Humans , Parkinson Disease/therapy , Parkinson Disease/drug therapy , Cross-Sectional Studies , Subthalamic Nucleus/physiology , Vision Disorders/epidemiology , Vision Disorders/etiology , Vision Disorders/therapy , Treatment Outcome
12.
West J Emerg Med ; 24(3): 377-383, 2023 May 03.
Article in English | MEDLINE | ID: mdl-37278797

ABSTRACT

INTRODUCTION: The emergency department (ED) is a critical service area for patients living with disabilities in the United States. Despite this, there is limited research on best practices from the patient experience regarding accommodation and accessibility for those with disabilities. In this study we investigate the ED experience from the perspective of patients living with physical and cognitive disability, as well as visual impairment and blindness, to better understand the barriers to accessibility in the ED for these populations. METHODS: Twelve individuals with either physical or cognitive disabilities, visual impairments or blindness were interviewed regarding their ED experiences, particularly related to accessibility. Interviews were transcribed and coded for qualitative analysis with generation of significant themes relating to accessibility in the ED. RESULTS: Major themes from coded analysis were as follows: 1) inadequate communication between staff and patients with visual impairments and physical disabilities; 2) the need for electronic delivery for after-visit summaries for individuals with cognitive and visual disabilities; 3) the importance of mindful listening and patience by healthcare staff; 4) the role of increased hospital support including greeters and volunteers; and 5) comprehensive training with both prehospital and hospital staff around assistive devices and services. CONCLUSION: This study serves as an important first step toward improving the ED environment to ensure accessibility and inclusivity for patients presenting with various types of disabilities. Implementing specific training, policies, and infrastructure changes may improve the experiences and healthcare of this population.


Subject(s)
Disabled Persons , Humans , United States , Disabled Persons/education , Qualitative Research , Emergency Service, Hospital , Vision Disorders/therapy , Blindness , Health Services Accessibility
13.
Am J Ophthalmol ; 254: 54-61, 2023 10.
Article in English | MEDLINE | ID: mdl-37336384

ABSTRACT

PURPOSE: Compared with the general population in North America, Native American/American Indian and Alaska Native (AI/AN) populations experience a disparate prevalence of eye diseases. Visual impairment is a barrier to communication, interferes with academic and social success, and decreases overall quality of life. The prevalence of ocular pathology could serve as an indicator of health and social disparities. Therefore, the objective of this research was to perform a thorough review comparing the prevalence of common ophthalmological pathologies between AI/AN and non-AI/AN individuals in North America. DESIGN: Retrospective, cross-sectional study. METHODS: A total of 57 articles were retrieved and reviewed, and 14 met the criteria outlined for inclusion. These articles were retrieved from PubMed, MEDLINE, and ISI Web of Knowledge. Only studies that were peer reviewed in the last 25 years and reported on the prevalence of eye diseases in AI/AN compared with a non-AI/AN population met criteria. RESULTS: Rates of retinopathy, cataracts, visual impairment, and blindness were clearly higher for AI/AN compared with non-AI/AN counterparts. Although rates of macular degeneration and glaucoma were similar between AI/AN and non-AI/AN populations, the treatment rates were lower and associated with poorer outcomes in AI/AN individuals. CONCLUSIONS: There are considerable inequities in the prevalence and treatment rates of ophthalmologic conditions in AI/AN individuals. A likely explanation is the barrier of lack of access to adequate health and eye care. Because of substantial underinsurance and geographic variability, attention needs to be brought to expanding eye care access to AI/AN communities. The results are subject to the availability of appropriate technology, health literacy, and language.


Subject(s)
American Indian or Alaska Native , Health Status Disparities , Healthcare Disparities , Vision Disorders , Humans , Cross-Sectional Studies , Indians, North American , Prevalence , Quality of Life , Retrospective Studies , United States/epidemiology , Vision Disorders/epidemiology , Vision Disorders/ethnology , Vision Disorders/therapy , Healthcare Disparities/ethnology
14.
Occup Ther Health Care ; 37(3): 326-356, 2023.
Article in English | MEDLINE | ID: mdl-37139689

ABSTRACT

Cerebral visual impairment is the leading cause of low vision in children, and functional gains can be achieved with intervention. To date there exists no evidence-based intervention protocol to guide rehabilitation therapists. This scoping review was conducted to synthesize the evidence currently available and explore current interventions in order to guide future research. This review identified five types of interventions for cerebral visual impairment; habilitation, visual stimulation, video game, color tent, and medical and also evidenced the need for standardized, objective measures of function for this population.


Subject(s)
Occupational Therapy , Vision, Low , Child , Humans , Occupational Therapists , Vision Disorders/therapy
16.
Eye (Lond) ; 37(16): 3412-3416, 2023 11.
Article in English | MEDLINE | ID: mdl-37076690

ABSTRACT

BACKGROUND: In the UK, the Certificate of Vision Impairment (CVI) certifies a person as sight impaired (partially sighted) or severely sight impaired (blind). This is completed by ophthalmologists and passed with the patient's consent to their GP, their local authority, and The Royal College of Ophthalmologists Certifications office. Once a person is certified, they can be registered by their local authority which is voluntary but enables the person to access rehabilitation or habitation services, financial concessions, welfare benefits and other services provided by local authorities. METHODS: We conducted semi-structured individual interviews with 17 patients with a diagnosed eye condition, 4 Eye Clinic Liaison Officers (ECLO) and 4 referring optometrists around their experiences around CVI and registration processes. Analysis of themes was conducted with results synthesised in a narrative analysis. RESULTS: Patients reported lack of clarity around the processes of certification and registration, benefits of certification and what happens beyond certification, the type of support that they are entitled to, delays in accessing support. Optometrists appear not to engage with the process much, especially if the patient is being treated by the hospital eye service. CONCLUSION: Vision loss can be a devastating experience for the patient. There is a lack of information and confusion around the process. The lack of a joined-up process between certification and registration needs to be addressed if we are to provide the support that patients deserve in order to improve their quality of life and wellbeing.


Subject(s)
Optometrists , Quality of Life , Humans , Vision Disorders/diagnosis , Vision Disorders/therapy , Certification , Visual Acuity
17.
Am J Phys Med Rehabil ; 102(9): 780-786, 2023 09 01.
Article in English | MEDLINE | ID: mdl-36753447

ABSTRACT

OBJECTIVE: The aim of the study was to measure the short- and long-term impact of a virtual disability education curriculum associated with a 2-wk mandatory physical medicine and rehabilitation clerkship for fourth-year medical students. DESIGN: A prospective pre-post intervention survey-based study measuring the impact of a virtual disability education series at 1-wk and 6-mo time points after a mandatory physical medicine and rehabilitation clerkship including 8 hrs of virtual didactics with an emphasis on physical disabilities. The surveys assessed the overall virtual curriculum, perceived benefit of a virtual encounter, and the long-term applicability of the information learned from the clerkship. RESULTS: The physical medicine and rehabilitation clerkship was effective in improving medical students' perceived comfort and clinical knowledge regarding treatment of persons with disabilities ( P < 0.01). There were no statistical differences at the 6-mo time point, indicating sustained benefit and integration of knowledge in the long term ( P > 0.05). In addition, 84% of students reported using the information in clinical experiences at 6 mos. CONCLUSIONS: The physical medicine and rehabilitation clerkship including a virtual disability education curriculum improved long-term perceived medical student comfort and knowledge of treating persons with disabilities, with a focus on those with physical disabilities. Virtual encounters with persons with disabilities are viable and impactful avenues to provide this education.


Subject(s)
Physical and Rehabilitation Medicine , Vision Disorders , Vision Disorders/therapy , Students, Medical , Education, Medical , Humans , Male , Female , Prospective Studies
18.
JAMA Ophthalmol ; 141(3): 275-283, 2023 03 01.
Article in English | MEDLINE | ID: mdl-36757689

ABSTRACT

Importance: X-linked retinitis pigmentosa (XLRP) is a severe cause of early-onset RP in male individuals, characterized by degeneration of photoreceptors, an extinguished electroretinogram, and vision loss. Objective: To assess the duration of improvements in retinal sensitivity associated with a single, subretinal injection of cotoretigene toliparvovec (BIIB112/AAV8-RPGR) gene therapy after vitrectomy surgery in the dosed eye over 12 months in part 1 of the Clinical Trial of Retinal Gene Therapy for X-linked Retinitis Pigmentosa Using BIIB112 (XIRIUS) study, compared with untreated fellow eyes and eyes from the untreated subgroup from the Natural History of the Progression of X-Linked Retinitis Pigmentosa (XOLARIS) study. Design, Setting, and Participants: This was a post hoc analysis of the XIRIUS and XOLARIS studies. Part 1 of the XIRIUS study was a phase 1, dose-escalation study of 18 male participants 18 years or older enrolled between March 8, 2017, and October 16, 2018, with genetically confirmed RPGR-variant XLRP with active disease and best-corrected visual acuity better than or equal to light perception (cohort 1), 34 to 73 letters (20/40 to 20/200 Snellen equivalent; cohorts 2-3), or greater than or equal to 34 letters (better than or equal to 20/200 Snellen equivalent; cohorts 4-6). Participants from the noninterventional, multicenter, global, prospective XOLARIS clinical study who met the inclusion and exclusion criteria of part 1 of XIRIUS were included as a comparator group (n = 103). Safety assessments included all XIRIUS participants; post hoc associations of retinal sensitivity assessments in XIRIUS only included the 12 participants receiving the 4 highest doses of cotoretigene toliparvovec. Data were analyzed on June 30, 2021. Main Outcomes and Measures: Incidence of dose-limiting toxicities (DLTs), treatment-emergent adverse events, changes from baseline in retinal sensitivity (as assessed by macular integrity assessment microperimetry), retinal sensitivity response (achievement of ≥7-dB improvement from baseline at ≥5 of 16 central loci), and low-luminance visual acuity were assessed over 24 months. Results: A total of 18 participants (mean [SD] age, 31.9 [9.4] years; male, 100%) were enrolled and completed the XIRIUS study. A subgroup of 103 participants (mean [SD] age, 30.8 [11.4] years; male, 100%) from the XOLARIS study was included. Administration of the 4 highest doses of cotoretigene toliparvovec (n = 12) among the 18 XIRIUS participants was associated with early improvements in retinal sensitivity. One of 103 untreated participants (1%) in the XOLARIS subgroup achieved improved retinal sensitivity at month 12. No DLTs were noted at any dose, and serious adverse events of reduced visual acuity (n = 2) and noninfective retinitis (n = 1) occurred. Conclusions and Relevance: Results suggest that early and sustained improvements in retinal sensitivity and low-luminance visual acuity in some participants through 12 months support consideration of additional clinical trials. Trial Registration: ClinicalTrials.gov Identifier: XIRIUS: NCT03116113; XOLARIS: NCT04926129.


Subject(s)
Retina , Retinitis Pigmentosa , Adult , Humans , Male , Eye Proteins/genetics , Genetic Therapy/methods , Prospective Studies , Retinitis Pigmentosa/diagnosis , Retinitis Pigmentosa/genetics , Retinitis Pigmentosa/therapy , Vision Disorders/therapy , Visual Acuity
19.
J Pak Med Assoc ; 73(Suppl 1)(2): S79-S88, 2023 Feb.
Article in English | MEDLINE | ID: mdl-36788396

ABSTRACT

Visual impairment adversely impacts quality of life and affects more than 295 million individuals globally. Currently, there is no cure or tissue regenerative approaches in clinical practice for vision loss caused by corneal disease, glaucoma, cataracts, macular degeneration, diabetic retinopathy, and inherited retinal disease. Stem cells-based therapeutic approaches to diseases causing moderate to severe visual impairment have shown encouraging outcomes in animal models and in vitro studies. The goal of this narrative review is to describe and evaluate the potential of stem cell-based treatment, and their advantages and safety concerns in treating conditions causing vision loss.


Subject(s)
Cataract , Diabetic Retinopathy , Glaucoma , Animals , Quality of Life , Vision Disorders/etiology , Vision Disorders/therapy , Cataract/complications , Glaucoma/therapy , Cell- and Tissue-Based Therapy
20.
Am J Ophthalmol ; 252: 213-224, 2023 08.
Article in English | MEDLINE | ID: mdl-36822570

ABSTRACT

PURPOSE: To evaluate the effectiveness of plasma exchange (PLEX) for optic neuritis (ON). METHODS: We conducted an international multicenter retrospective study evaluating the outcomes of ON following PLEX. Outcomes were compared to raw data from the Optic Neuritis Treatment Trial (ONTT) using a matched subset. RESULTS: A total of 395 ON attack treated with PLEX from 317 patients were evaluated. The median age was 37 years (range 9-75), and 71% were female. Causes of ON included multiple sclerosis (108), myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) (92), aquaporin-4-IgG-positive neuromyelitis optica spectrum disorder (AQP4+NMOSD) (75), seronegative-NMOSD (34), idiopathic (83), and other (3). Median time from onset of vision loss to PLEX was 2.6 weeks (interquartile range [IQR], 1.4-4.0). Median visual acuity (VA) at the time of PLEX was count fingers (IQR, 20/200-hand motion), and median final VA was 20/25 (IQR, 20/20-20/60) with no differences among etiologies except MOGAD-ON, which had better outcomes. In 81 (20.5%) ON attacks, the final VA was 20/200 or worse. Patients with poor outcomes were older (P = .002), had worse VA at the time of PLEX (P < .001), and longer delay to PLEX (P < .001). In comparison with the ONTT subset with severe corticosteroid-unresponsive ON, a final VA of worse than 20/40 occurred in 6 of 50 (12%) PLEX-treated ON vs 7 of 19 (37%) from the ONTT treated with intravenous methylprednisolone without PLEX (P = .04). CONCLUSION: Most ON attacks improved with PLEX, and outcomes were better than attacks with similar severity in the ONTT. The presence of severe vision loss at nadir, older age, and longer delay to PLEX predicted a worse outcome whereas MOGAD-ON had a more favorable prognosis. NOTE: Publication of this article is sponsored by the American Ophthalmological Society.


Subject(s)
Neuromyelitis Optica , Optic Neuritis , Humans , Female , Male , Plasma Exchange , Retrospective Studies , Myelin-Oligodendrocyte Glycoprotein , Optic Neuritis/therapy , Vision Disorders/therapy , Autoantibodies
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