Impact of Retinitis Pigmentosa on Quality of Life, Mental Health, and Employment Among Young Adults

Chaumet-Riffaud, Anne Elisabeth; Chaumet-Riffaud, Philippe; Cariou, Anaelle; Devisme, Céline; Audo, Isabelle; Sahel, José‐Alain; Mohand‐Saïd, Saddek

doi:10.1016/j.ajo.2017.02.016

Cited by 63 publications

(80 citation statements)

References 22 publications

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“…Among this sample of people with advanced retinal degeneration, neither VA nor visual fields were independently associated with quality of life or any other psychosocial measure. This is in contrast to previous reports of a strong relationship between visual function (particularly visual field) and quality of life . This may be because many participants in this study had late‐stage vision loss and personalities were heterogeneous, with some participants being much more able to cope with severe vision loss than others.…”

Section: Discussioncontrasting

confidence: 96%

“…This study has shown there are a number of validated questionnaires that can be of use in assessing psychosocial characteristics of persons with advanced retinal degeneration. In particular, depression and vision‐specific coping are known to be associated with quality of life among people with RP, and have again been shown to be correlated in the current study. However, whether or not a person might be interested in participating in a retinal prosthesis clinical trial varies considerably and is not clearly predicted by psychosocial measures, or measures of residual vision.…”

Section: Discussionsupporting

confidence: 63%

“…RP is a genetic disease affecting over 1.6 million people worldwide . It has previously been reported that people with RP exhibit a higher prevalence of depression and anxiety than people with normal vision, as is the case among people with vision impairment caused by other eye diseases, and that mental health is a determinant of quality of life in this population . Other prior studies of psychosocial factors in people with RP have investigated adaptation to vision loss, social participation, and emotional health .…”

mentioning

confidence: 99%

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Psychosocial assessment of potential retinal prosthesis trial participants

Bentley

O’Hare

Murphy

et al. 2019

Clinical and Experimental Optometry

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Background: As the field of retinal prostheses advances, volunteers are required for device trials, and optimal participant recruitment is vital for intervention success. The aims of this study were: (i) to select tests that assess the psychosocial aspects of visual impairment and develop a psychosocial assessment protocol for persons who may be eligible for participation in retinal prostheses trials; (ii) to investigate correlations between these tests; and (iii) to determine associations between psychosocial factors and a person's interest in participating in a retinal prosthesis (bionic eye) trial. Methods: Cross-sectional study of 72 adults with advanced retinal degeneration. Questionnaire assessments included personality, cognitive ability, social-support, self-efficacy, coping, optimism, depression, and quality of life (Impact of Vision Impairment Profile ([IVI], and Vision and Quality of Life Index [VisQoL]). Level of interest in a retinal prosthesis was also evaluated. Results: All questionnaires were completed without floor or ceiling effects and with minimal respondent burden. Depression correlated with decreased quality of life (rho = −0.37 and 0.40, p < 0.001 for IVI and VisQoL, respectively). Together, depression, gender and visionspecific coping explained 35.2 per cent of variance in IVI quality of life (p < 0.001). Forty-nine per cent of participants were interested in a retinal prosthesis now and 77 per cent in the future. Although the personality trait of 'openness' was somewhat predictive of interest in retinal prostheses (odds ratio 0.78, 95% CI 0.62-0.97), neither severity of vision impairment nor any of the psychosocial measures were strong predictors. Conclusions: Several existing psychosocial questionnaires can be used for patients with advanced retinal degeneration and may be useful in exploring suitability for a retinal prosthesis or evaluating outcomes. However, the questionnaires used in this study were not good predictors of whether or not a person might be interested in a retinal prosthesis. Functional vision score, % Median (range) 9 (0-80) Personality -NEO-FFI-3 neuroticism, T-score Mean (SD; range) 47 (11; 24-75) Personality -NEO-FFI-3 extraversion, T-score Mean (SD; range) 51 (9; 27-74) Personality -NEO-FFI-3 openness, T-score Mean (SD; range) 51 (9; 35-72) Personality -NEO-FFI-3 agreeableness, T-score Mean (SD; range) 53 (11; 24-76) Personality -NEO-FFI-3 conscientiousness, T-score Mean (SD; range) 54 (10; 24-76) Cognitive ability -WAIS-IV verbal similarities, raw score Mean (SD; range) 18 (4; 7-27) Cognitive ability -WAIS-IV verbal comprehension, raw score Mean (SD; range) 21 (6; 6-31) Social support -MOS-SSS, score Mean (SD; range) 80 (19; 21-100) Self-efficacy -GSE, score Mean (SD; range) 33 (5; 14-40) Coping -Vis-OPS selective primary control, score Mean (SD; range) 23 (2; 17-47) Coping -Vis-OPS compensatory primary control, score Mean (SD; range) 30 (5; 17-36)Coping -Vis-OPS selective secondary control, score Mean (SD; range) 31 (5; 16-40)Coping -Vis-OPS compensatory seconda...

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Section: Discussioncontrasting

confidence: 96%

Section: Discussionsupporting

confidence: 63%

mentioning

confidence: 99%

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Psychosocial assessment of potential retinal prosthesis trial participants

Bentley

O’Hare

Murphy

et al. 2019

Clinical and Experimental Optometry

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“…Children affected by visual impairment are more likely to experience social and economic disadvantage [7], and the impacts of reduced mobility and independence become an increasing problem as young adults progress into further and higher education. Severe visual impairment is associated with an increased risk of depression, anxiety and sleep disorders [8][9][10][11][12][13], and studies have also reported an increased risk of depression among caregivers of those with visual impairment [14,15]. It is estimated that the full lifetime cost of blindness in individuals with biallelic RPE65-mediated IRD in the UK is between £1.6 and £1.8 million.…”

Section: Disease Backgroundmentioning

confidence: 99%

An Economic Evaluation of Voretigene Neparvovec for the Treatment of Biallelic RPE65-Mediated Inherited Retinal Dystrophies in the UK

et al. 2020

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Introduction: Voretigene neparvovec (VN) is a gene therapy and the first approved pharmacological treatment for biallelic RPE65-mediated inherited retinal dystrophies (IRD), a rare condition that starts in early life and causes vision to progressively deteriorate towards complete blindness. In a phase III trial, treatment with VN significantly improved functional vision and visual function, and in October 2019 the National Institute for Health and Care Excellence (NICE) Highly Specialised Technologies Enhanced Digital Features To view enhanced digital features for this article go to https://doi.org/10.6084/ m9.figshare.11637108.

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“…A report on rare diseases in Europe indicated a large percentage of patients and carers reported difficulty completing daily tasks (80%). 28 Regarding care, 60% of patients and carers found it difficult to manage care, and 70% found it time-consuming. 28 It was also reported that health, social, and local services communicated poorly with each other by 67% of respondents.…”

Section: Discussionmentioning

confidence: 99%

<p>The Impact of Inherited Retinal Diseases in the Republic of Ireland (ROI) and the United Kingdom (UK) from a Cost-of-Illness Perspective</p>

Galvin¹,

Chi

Brady

et al. 2020

OPTH

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To date, there has been a global lack of data regarding the prevalence of conditions falling under the Inherited Retinal Diseases (IRD) classification, the impact on the individuals and families affected, and the cost burden to economies. The absence of an international patient registry, and equitable access to genetic testing, compounds this matter. The resulting incomplete knowledge of the impact of IRDs hinders the development and commissioning of clinical services, provision of treatments, and planning and implementation of clinical trials. Thus, there is a need for stronger evidence to support value for money to regulatory bodies for treatments approved, and progressing through clinical trials. To ensure a strategic approach to future research and service provision, it is necessary to learn more about the IRD landscape. This review highlights two recent cost-of-illness reports on the socioeconomic impact of 10 IRDs in the Republic of Ireland (ROI) and the United Kingdom (UK), which demonstrate the comprehensive impact of IRDs on individuals affected, their families, friends and society. Total costs attributable to IRDs in the ROI were estimated to be £42.6 million in 2019, comprising economic (£28.8 million) and wellbeing costs (£13.8 million). Wellbeing costs were estimated using the World Health Organization (WHO) burden of disease methodology, a non-financial approach, where pain, suffering and premature mortality are measured in terms of disability-adjusted-lifeyears (DALYs). In the UK, wellbeing costs attributable to IRDs were £196.1 million, and economic costs were £327.2 million amounting to £523.3 million total costs in 2019. Accounting for over one-third of total costs, the wellbeing burden of persons affected by IRDs should be emphasized and factored into reimbursement processes for therapies and care pathways. This targeted review presents the most current and relevant data on IRD prevalence in the ROI and the UK, and the impacts (financial and non-financial) of IRDs in terms of diagnosis, wellbeing, employment, formal and informal care, health system costs, deadweight losses and issues surrounding payers and reimbursement. This review demonstrates IRD patients and their families have common issues including, the need for timely equitable access to genetic testing and counselling, equality in accessing employment, and a revision of the assessment process for reimbursement of therapies currently focused on the cost-of-illness to the healthcare system. This review reveals that IRD patients do not frequently engage the healthcare system and as such suggests a cost-of-illness model from a societal perspective may be a better format.

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Impact of Retinitis Pigmentosa on Quality of Life, Mental Health, and Employment Among Young Adults

Cited by 63 publications

References 22 publications

Psychosocial assessment of potential retinal prosthesis trial participants

Psychosocial assessment of potential retinal prosthesis trial participants

An Economic Evaluation of Voretigene Neparvovec for the Treatment of Biallelic RPE65-Mediated Inherited Retinal Dystrophies in the UK

<p>The Impact of Inherited Retinal Diseases in the Republic of Ireland (ROI) and the United Kingdom (UK) from a Cost-of-Illness Perspective</p>

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