Affective and anxiety disorders in patients with different rare chronic diseases: a systematic review and meta-analysis

Uhlenbusch, Natalie; Swaydan, J; Höller, Alexandra; Löwe, Bernd

doi:10.1017/s0033291721003792

Cited by 20 publications

(14 citation statements)

References 52 publications

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“…Our findings demonstrate that rare disease has a substantial negative impact on mental health for both patients and carers. This reflects existing literature on unmet needs [ 3 , 5 , 28 ] and is consistent with findings from condition-specific studies which used diagnostic tools to measure anxiety and/or depression in rare disease patients and/or carers and impact on quality of life [ 8 , 10 , 29 – 32 ].…”

Section: Discussionsupporting

confidence: 87%

“…Australian and American rare disease patient populations have been better studied and findings illustrate clear psychosocial and health-related quality of life impacts [ 5 – 9 ]. Recently a systematic review and meta-analysis of affective and anxiety disorders in adult patients with rare conditions reported prevalence estimates higher than the general population [ 10 ].…”

Section: Introductionmentioning

confidence: 99%

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Mental health care for rare disease in the UK – recommendations from a quantitative survey and multi-stakeholder workshop

Spencer-Tansley

Meade

Ali

et al. 2022

BMC Health Serv Res

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Background Rare disease patients and carers report significant impacts on mental health but studies on UK populations have focussed on relatively few, specific conditions. Collectively rare conditions represent a substantial health burden, with an estimated 3.5 million affected individuals in the UK. Method We explored the impact on mental health of living with a rare condition, and experiences of health service support, through an online survey. The survey assessed the impact of specific experiences commonly reported by those affected by a rare condition through multiple choice questions and Likert scale items, and open text question boxes. Through a multi-stakeholder workshop that involved facilitated discussion of our findings with patients/carers, clinicians and a government advisor, we developed recommendations for policy and practice toward a more person-centred and integrated approach. Results Eligible responses came from 1231 patients and 564 carers. Due to their rare condition, the majority of respondents (> 90%) had felt worried/anxious; stressed; and /or low/depressed. Thirty-six percent of patients and 19% of carers had had suicidal thoughts. Challenges that are particular to rare conditions and which negatively affect mental health included limited knowledge of the condition amongst healthcare professionals (88%), and not being believed or taken seriously by them. Only 23% of respondents felt healthcare professionals considered mental and physical health as equally important. Almost half reported never having been asked about mental health by healthcare professionals. Our findings indicate that access to, and appropriateness of, professional psychological support needs to be improved. Peer group support is important but signposting is inadequate. Our recommendations are for healthcare professionals to be supported to effectively and sensitively recognise and address patients’ and carers’ mental health needs; and for service level coordination of care to integrate professional psychological support with rare disease services. Conclusion Living with a rare disease substantially impacts mental health. Many of the drivers of poor mental health reflect issues specific to managing rare conditions. To meet UK government commitments, there should be a focus on empowering healthcare professionals who treat rare disease patients and on integration of mental health support with rare disease services.

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Section: Discussionsupporting

confidence: 87%

Section: Introductionmentioning

confidence: 99%

Mental health care for rare disease in the UK – recommendations from a quantitative survey and multi-stakeholder workshop

Spencer-Tansley

Meade

Ali

et al. 2022

BMC Health Serv Res

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“…This is further exacerbated by the often-chronic characteristics of these diseases and social, economic, and psychological impacts inherent to rare diseases ( Alonso et al, 2004 ). With regards to mental health for instance, anxiety and depression are often reported amongst people living with a rare disease in addition to condition-specific symptoms ( Winter et al, 2010 ; Uhlenbusch et al, 2019b ; Uhlenbusch et al, 2021 ). Similar conclusions can be drawn for diagnosis, as people living with a rare disease often wait on average 5 years before receiving a correct diagnosis, thus contributing to the psychological impact of rare diseases in addition to costs incurred during the diagnostic odyssey ( European Brain Council, 2017 ; Eurordis, 2017 ; World Economic Forum, 2020 ).…”

Section: Discussionmentioning

confidence: 99%

Valuing the “Burden” and Impact of Rare Diseases: A Scoping Review

2022

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Introduction: Rare diseases (RDs) are a severe, chronic, degenerative and often life-threatening group of conditions affecting more than 30 million people in Europe. Their impact is often underreported and ranges from psychological and physical symptoms seriously compromising quality of life. There is then a need to consolidate knowledge on the economic, social, and quality of life impacts of rare diseases.Methods: This scoping review is the result of 9 qualitative interviews with experts and a literature search on Cost-of-Illness (COI) studies and quality of life (QoL) studies following the PRISMA methodology. Grey literature was also included to complement findings. Results. 63 COI studies were retrieved, covering 42 diseases and a vast majority of them using a prevalence-based approach (94%). All studies included medical costs, while 60% included non-medical costs, 68% productivity losses and 43% informal care costs. 56 studies on QoL were retrieved, mostly from Europe, with 30 different measurement tools. Grey literature included surveys from the pharmaceutical industry and patient organisations.Discussion: The majority of studies evaluating the impact of RDs on the individual and society use the COI approach, mostly from a societal perspective. Studies often vary in scope, making them difficult to consolidate or compare results. While medical costs and productivity losses are consistently included, QoL aspects are rarely considered in COI and are usually measured through generic tools.Conclusion: A comprehensive study on impact of rare disease across countries in Europe is lacking. Existing studies are heterogeneous in their scope and methodology and often lack a holistic picture of the impact of rare. Consensus on standards and methodology across countries and diseases is then needed. Studies that consider a holistic approach are often conducted by pharmaceutical companies and patient organisations exploring a specific disease area but are not necessarily visible in the literature and could benefit from the sharing of standards and best practices.

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“…Unfortunately, the reality is so far from this ambitious goal that, actually, achieving a diagnosis is a road riddled with dark sides and pitfalls. Alongside the consolidated literature about the economic burden and the impact on the quality of life of patients affected with a chronic illness [6][7][8][9], the ethical, legal, social, and psychological implications (ELSI and ELPAG) of RD are now a growing issue in medicine and genomic research, as evidenced by the widespread initiatives on ELSI and ELPAG research supported by human genetics scientific societies [10][11][12] and other international research programs, such as the ELSI Research Program at the National Human Genome Research Institute [13]. Numerous initiatives are available with strong educational components to provide genomic education to healthcare professionals and empower patients and associations.…”

Section: Introductionmentioning

confidence: 99%

The Right to Ask, the Need to Answer—When Patients Meet Research: How to Cope with Time

Priolo

Tartaglia

2023

IJERPH

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Reaching a diagnosis and its communication are two of the most meaningful events in the physician–patient relationship. When facing a disease, most of the patients’ expectations rely on the hope that their clinicians would be able to understand the cause of their illness and eventually end it. Rare diseases are a peculiar subset of conditions in which the search for a diagnosis might reveal a long and painful journey scattered by doubts and requiring, in most cases, a long waiting time. For many individuals affected by a rare disease, turning to research might represent their last chance to obtain an answer to their questions. Time is the worst enemy, threatening to disrupt the fragile balance among affected individuals, their referring physicians, and researchers. It is consuming at all levels, draining economic, emotional, and social resources, and triggering unpredictable reactions in each stakeholder group. Managing waiting time is one of the most burdensome tasks for all the parties playing a role in the search for a diagnosis: the patients and their referring physicians urge to obtain a diagnosis in order to know the condition they are dealing with and establish proper management, respectively. On the other hand, researchers need to be objective and scientifically act to give a rigorous answer to their demands. While moving towards the same goal, patients, clinicians, and researchers might have different expectations and perceive the same waiting time as differently hard or tolerable. The lack of information on mutual needs and the absence of effective communication among the parties are the most common mechanisms of the failure of the therapeutic alliance that risk compromising the common goal of a proper diagnosis. In the landscape of modern medicine that goes faster and claims high standards of cure, rare diseases represent an exception where physicians and researchers should learn to cope with time in order to care for patients.

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Affective and anxiety disorders in patients with different rare chronic diseases: a systematic review and meta-analysis

Cited by 20 publications

References 52 publications

Mental health care for rare disease in the UK – recommendations from a quantitative survey and multi-stakeholder workshop

Mental health care for rare disease in the UK – recommendations from a quantitative survey and multi-stakeholder workshop

Valuing the “Burden” and Impact of Rare Diseases: A Scoping Review

The Right to Ask, the Need to Answer—When Patients Meet Research: How to Cope with Time

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