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Edition of 08 October 2021

International News

Evaluation of rare diseases policy performance of OECD countries using health policy indicators

A new study assessing the policy performance of 18 OECD countries on rare diseases has been published in the Health Policy and Technology Journal. Using the EUCERD indicators for rare diseases and MOORA, COPRAS, and TOPSIS techniques, the study reveals that Germany, France and the Netherlands have the best rare diseases policy performance, as opposed to Latvia, Greece and Turkey which rank the lowest. The study also highlights that EU Member States have excellent rare disease policies and health services, boosted by a well-coordinated and strong structure for rare diseases, extensive research on healthcare and education, as well as patient advocacy organisations, all of which are notably true for France Germany and England. Health policy ratings also depend on the budget allocated by governments, the presence of epidemiological studies as well as orphan drug regulations and coverage in health insurance.

The study sheds light on the need to integrate rare diseases into health policies and to further conduct performance evaluation studies for service delivery and policy arrangements in the field of rare diseases.

United States: Launch of a new rare diseases data and analytics platform

C-Path, Nord and the FDA have launched the Rare Disease Cures Accelerator-Data and Analytics Platform (RDCA-DAP®), a new platform that aims to support rare disease treatment innovation by centralizing patient data and standardizing data collection. The RDCA-DAP will serve as a base to host and share de-identified rare diseases data in order to inform and boost new FDA approved drugs and rare disease therapies.

United States: California legislature poised to establish Rare Disease Advisory Council

The US Senate cleared a bill aimed at establishing a Rare Disease Advisory Council within the California Health and Human Services Agencies (CHHSA), before being signed into law by the governor of California. The Council will represent rare disease patients in the state and will act as the advisory body to the Legislature and state departments in the field of rare diseases. It will also be tasked with adopting and implementing regulations, researching and determining the most appropriate methods to collect data on rare diseases, and identifying best practices for rare disease care.

India: Government launched a digital crowdfunding programme for rare diseases

The Indian government has launched a digital platform for voluntary donations dedicated to rare disease treatment. The portal allows corporates and individuals to donate online and provides information relating to the patients, the diseases from which they are suffering, the estimated cost of treatment as well as the details of bank accounts of the treating Institutions.

Australia: Formation of the first Parliamentary Friends of People with Rare and Undiagnosed Diseases in Western Australia

Members of the Legislative Council of Western Australia have formed the first Parliamentary Friends of People with Rare and Undiagnosed Diseases. The non-partisan Parliamentary Friendship Group aims to provide information on opportunities and challenges raised by rare diseases and raise awareness of the role and opportunity of innovation, data, research, policy as well as national and international partnerships in the rare disease field. It will also champion initiatives that seek to deliver improved outcomes for people with rare and undiagnosed diseases, and their families.

New Zealand: Ministry of Health not to proceed with a rare disorder national framework

The New Zealand Ministry of Health dismissed the call made by several organisations
for the development a national framework for rare disorders. The Ministry stated that the current government work program in health includes improving health outcomes for rare disease patients, while a national rare disorders framework would require resourcing that is not available at this time.

Call for a UN Resolution for persons living with a rare disease and their families

The opening of the 76th session of the UN General Assembly on 14 September 2021 offered a unique opportunity for the international rare disease community and the future of rare diseases as a Core Group of Member States- Brazil, Qatar, and Spain – is expected to table a Resolution on Persons Living with a Rare Disease and their Families. The campaign launched by Rare Diseases International using the #Resolution4Rare called all Member States of the UN General Assembly to join the call and adopt the UN Resolution this year. To boost the campaign during this crucial time, Rare Diseases International asked patients and families to share a testimony on their journey and experience through their website.

European News

Agreement on a regulation on the European Cooperation in Health Technology Assessment

On 22 June 2021, Parliament and Commission reached provisional agreement on a regulation on the European Cooperation in Health Technology Assessment (HTA), a research-based tool that supports decision-making in healthcare by assessing the added value of a given health technology compared to others. The regulation, proposed by the Commission in 2018, aims to strengthen EU-level cooperation among Member States and will help provide more robust, transparent, and complete information about new medicines, devices, and diagnostics to patients, including in the field of rare diseases and orphan drugs. The regulation also establishes a new member state coordination group which will include representatives and experts from Member States.

The Committee of the Permanent Representatives (COREPER) confirmed the provisional agreement on 30 June 2021 and the Parliament’s Environment, Public Health & Food Safety committee voted on the provisional agreement on 13 July 2021. The Council and then Parliament are expected to formally adopt it in the coming months.

Ahead of this agreement, the European Network for Health Technology Assessment has published its final white paper on the future model of HTA cooperation across the EU. This white paper defines the scientific and technical principles for a future model of HTA cooperation and recommends the next steps to implement this model. The paper highlights that in order to be successful, the future HTA framework for cooperation needs to provide stability and continuity, allow the cooperation to act as an entity, and have a commissioning and resourcing structure that can guarantee project teams for joint HTA activities.

Rare disease patients' opinion on the EU Cross-Border Healthcare Directive

On the tenth anniversary of the adoption of the EU Directive on Cross-Border Health care and its evaluation by the European Commission, EURORDIS conducted surveys and held extensive consultations with rare diseases patients and advocates to collect their opinion on the Directive’s achievements. The results of these consultations highlight that rare disease patients are facing bureaucratic and financial obstacles to access cross-border health care, including barriers experienced in the healthcare systems of their home countries. Surveyed patients also pointed out the lack of adequate information received on access to healthcare abroad according to their situations. Moreover, the lack of investment received by the European Reference Networks keeps them from delivering high-quality, accessible, and cost-effective health care to rare disease patients.

Recommendations for action in the EU Pharmaceutical strategy

A new paper entitled “Recommendations for Action in the EU Pharmaceutical Strategy” has been published by the European Alliance for Transformative Therapies (TRANSFORM) calling for safe and timely patient access to transformative therapies, such as gene therapies for genetic rare diseases. The series of recommendations include promoting the distribution of these novel therapies through rare disease centres of reference or centres of excellence in European Member States, equipped with the necessary facilities and specialist knowledge on both these innovative therapies and the conditions they intend to treat. TRANSFORM also calls for more cross-border healthcare cooperation in the European Union and between these qualified centres. The paper highlights the need for improved national funding dedicated to the development of innovative therapies in the field of rare diseases.

Launch of a survey on paediatric Advanced Therapy Medicinal Products Expertise

The European Paediatric and Translational Research Infrastructure (EPTRI) and RESTORE have launched a survey on paediatric Advanced Therapy Medicinal Products (ATMPs) dedicated to professionals involved in the development of ATMPs. The aim of this survey is to describe the map of competences available in the framework of the paediatric ATMPs research in pan-European countries in order to have the chance to promote the development of tailored paediatric advanced drugs and facilitate collaborations.

Call for a European Action Plan on Rare Diseases: EURORDIS survey results

The results of the Rare Barometer survey launched by EURORDIS on the opinion of people living with a rare disease on policies that may impact their lives over the next ten years have been published. This survey was carried out as part of the EURORDIS campaign calling for a European Action Plan on rare diseases.

Surveyed respondents ranked access to diagnosis and treatments that do not yet exist as the highest priority to improve their care, followed by improving coordination between all healthcare professionals involved as well as having consultations with healthcare professionals specialised in their disease. Respondents also highlighted how a more holistic approach, including receiving more emotional support, better managing symptoms and facilitating access to employment, could improve their lives in the next decade. The survey also reveals that 95% of the respondents support screening for rare conditions at birth, despite the lack of a harmonised approach across the EU.

These results highlight the need for an overarching strategy on rare diseases across member states, a European Action Plan for rare diseases, which considers patients’ preferences while making significant progress in the fields of health innovation, precision medicine, genomic health, and digital health innovation.

Health Economics

The impact of health-care utilisation by rare disease patients in the United States

A new study has been published in the Genetics in Medicine journal to measure the impact of rare disease patients within the health-care systems of the United States. By analysing their overall cost and utilization of healthcare using the Healthcare Cost and Utilization Project (HCUP) database, the study reveals that children and adults suffering from rare conditions have a substantially higher health-care utilization than patients with common conditions, accounting for nearly half of the US national bill. In light of these findings, the study highlights the need to incorporate cost-saving measures and improved health-care access for those affected by rare diseases.

Parents’ experiences with the U.S health insurance system for children with rare diseases

A study has been published in the Orphanet Journal of Rare Diseases to reflect parents’ perceptions when navigating the complex United States health insurance system for their children with rare diseases. During the interviews, parents expressed the barriers they face with insurance companies that are often ill-equipped to provide clear answers for rare diseases due to lack of knowledge on the conditions, forcing them to track insurance benefits and interactions to balance medical needs and financial stability. These setbacks are costly and disruptive to the health of the child.

Economic burden and health related quality of life of ultra-rare Gaucher disease in China

A new study on the economic burden of the Gaucher disease on Chinese patients as well as the consequences of the disease on their quality of life and well-being has been published in the Orphanet Journal of Rare Diseases. The study reveals that Gaucher disease patients are often faced with a high misdiagnosis rate in China, coupled with extremely high disease-related expenses, especially related to costly pharmaceutical treatments and restrictive reimbursement caps. Lack of social support for patients suffering from Gaucher disease often resulted in the deterioration of their health-related quality of life and other major health issues both of which deserve more attention.

Estimating the broader fiscal consequences of acute hepatic porphyria in Belgium

A study has been published in the Orphanet Journal of Rare Diseases on the fiscal consequences of acute hepatic porphyria (AHP) with recurrent attacks in Belgium. AHP is characterized by potentially life-threatening attacks often resulting in chronic symptoms. The study reveals that this condition causes significant public costs, beyond just health service costs. Lifetime taxes paid are also reduced as these attacks occur during peak earning and working years. In those patients, reducing AHP attacks can confer significant fiscal benefits for government, including reduced healthcare costs, reduced disability payments and improved tax revenue.

Economic burden associated with tenosynovial giant-cell tumour

A study has been published in the Orphanet Journal of Rare Diseases to define the economic burden associated with tenosynovial giant-cell tumour (TGCT) patients and their health-related quality of life in six European countries. The study highlights that TGCT not only places a heavy economic burden on patients due to healthcare resources required and associated costs, but also throughout European societies and healthcare systems.

Direct and indirect costs and cost-driving factors of Tuberous sclerosis complex

Two separate studies have been published in the Orphanet Journal of Rare Diseases on the direct and indirect costs of the Tuberous sclerosis complex in Germany, in children, adolescents and caregivers as well as in adults. The studies found that the rare disease drives substantial direct costs, particularly for medication and inpatient stays, but also indirect costs, including loss of productivity, taking days off, quitting work and reduction in working hours. For children, Tuberous sclerosis complex also induced considerable substantial nursing costs.

Orphan Drugs

R&D and market size: Who benefits from orphan drug legislation?

A new article has been published in the Journal of Health Economics describing the beneficiaries of the orphan drug legislation when looking at R&D. The study reveals that the current incentives included in the orphan drug regulations favour R&D for orphan diseases with high prevalence. This finding highlights the fact that a great gap remains between orphan drug development, which depends on the market size, and the greatest unmet medical needs in the field of rare diseases. The study thus questions the type of incentives adopted under the orphan drug legislation and suggests an overall revision of the current system.

Essential list of medicinal products for rare diseases

A new study published in the Orphanet Journal of Rare Diseases provides a list of medicinal products for rare diseases. The IRDiRC Rare Disease Treatment Access Working Group used the FDA, EMA and Chinese NMPA databases to extract approved drugs and create the first list of 204 essential medicinal products for rare diseases. The main objective of this list is to promote access to treatment for rare disease patients and encourage health care providers and decision-makers to improve standards for care. To this end, it was divided into seven disease categories, excluding rare cancers and rare infectious diseases which should benefit from a separate list due to their large number.

The impact of different country processes for appraising rare disease treatments

A new study on the impact of different country processes for appraising rare disease treatments has been published in the International Journal of Technology Assessment in Health Care. The study is based on reports from Belgium, England, France, Germany, Italy, the Netherlands, Norway, Scotland, Sweden, and the United States on the nusinersen and voretigene neparvovec treatments. From these case-studies, it appears that having a separate assessment process for rare disease treatment results in a more consistent approach for managing rare disease treatment specificities.

Reauthorisation of the Prescription Drug User Fee Act (2023-2027): impact on rare diseases

The FDA has published its plan for revising and renewing its fee program for drugs and biologics, the Prescription Drug User Fee Act (PDUFA). The renewal of the PDUFA VII covers the years 2023 to 2027 and is intended for Congress approval next year. Under the new framework, the FDA will create a Split Real Time Application Review (STAR) pilot program to streamline the review process to allow earlier patient access to therapies that address an unmet medical need. Moreover, the FDA aims at establishing a Rare Disease Endpoint Advancement (RDEA) Pilot Program that provides a mechanism for sponsors to collaborate with the agency on endpoints when developing a program for drugs that would treat rare diseases.

European stakeholder perspectives on challenges to rare disease drug development

A new study has been published in the Expert Opinion on Orphan Drugs journal on European stakeholder’s perspectives on challenges to rare disease drug development. The study is based on interviews conducted with a range of stakeholders in order to identify these challenges and areas of improvement. The study highlights the need to engage clinicians, academics and pharmaceutical companies in order to overcome the lack of awareness surrounding the rare disease field and to boost scientific research. The study also reveals the crucial role of patient groups in rare diseases drug development.

A systematic review of moral reasons on orphan drug reimbursement

A study has been published in the Orphanet Journal of Rare Diseases on the reimbursement of orphan drugs. The study aims to analyse the moral reasons in support of or against granting orphan drugs a special status for their reimbursement. The study highlights the fact that this binary perspective tends to oversimplify the issue and that the debate should rather focus on how the specificities of orphan drugs could be addressed and implemented in Health Technology Assessments.

Trends in rare disease drug development

A study on the trends in rare disease drug development has been published in the Orphanet Journal of Rare Diseases. The study analyses the last four decades of FDA orphan drug designations to identify patterns using data made available between 1983-2019. The study reveals that while orphan designations continue to rise in general, there has been a particular increase in designations for orphan drugs related to rare oncologic, neurologic, and paediatric-onset diseases. The rise in orphan drug designations observed in this study over the past four decades highlights the strong likelihood of increased orphan drug market approvals in the coming years.

The impact of rarity in NICE’s health technology appraisals

A study has been published in the Orphanet Journal of Rare Diseases comparing orphan and non-orphan medicine appraisal by the National Institute for Health and Care Excellence (NICE) through the Single Technology Appraisal (STA) process in the United Kingdom. The study reveals that orphan medicine take longer in the appraisal process than non-orphan drugs, while having a lower proportion of recommended outcomes. A better understanding of the challenges related to orphan drug development and clearer instructions for decision-maker in the health technology appraisal process would allow easier access to orphan medicines.

Screening and prenatal diagnosis

New Australian national pilot program for Congenital Adrenal Hyperplasia newborn screening

The Government of Victoria has announced a new pilot screening program for Congenital Adrenal Hyperplasia under the national Newborn Bloodspot Screening Program. The program will benefit from $1 million in funding to develop its operational capacity and capability.

Ethical Legal and Social

Social and medical needs of rare metabolic patients in Europe

A survey conducted by the European Reference Network for Hereditary Metabolic Disorders (MetabERN) on the social and medical needs of rare metabolic patients and families in Europe has been published in the Orphanet Journal of Rare Diseases. The survey reveals a lack of psychological assistance for rare metabolic patients and families in Europe, as well as the sense of isolation they feel due to their condition. The survey also sheds light on the high disease-related costs patients are faced with and the lack of services aimed at promoting their autonomy.

The parental perspective of thalassaemia in Bangladesh

A new study has been published in the Orphanet Journal of Rare Diseases on the perspectives of parents of thalassaemic children in Bangladesh. The study aims to understand parent’s knowledge of the disease, the challenges they face when caring for their children as well as their opinion on prenatal screening. Through interviews conducted with parents, the study highlights the crucial need for prevention regarding thalassaemia in Bangladesh, the poor transfusion services available to patients as well as the low prenatal screening rate.

Perspectives of people and parents of children with mitochondrial conditions after diagnosis

A study on the experience of people and parents of children with a mitochondrial respiratory chain disorder after diagnosis has been published in the Orphanet Journal of Rare Diseases. The study focuses on Australia and reveals that parents and patients are faced with several challenges regarding access to appropriate advice and services following diagnosis of a mitochondrial condition, notably in the fields of mental health and day-to-day assistance to access social services.

Patient preferences for gene therapy in haemophilia

A new study has been published in the Official Journal of the World Federation of Haemophilia on patient with haemophilia preferences between prophylactic factor replacement therapy (PFRT) and gene therapy. The study is based on the Patient preferences to Assess Value IN Gene therapies (PAVING) survey conducted in Belgium and reveals that preferences for gene therapy in haemophilia are heterogenous and mainly influenced by patient’s comprehension and education on novel therapies

Quality of life

The status quo of rare disease patients and care-givers in China

A new study on the current status of rare disease patients and care-givers in China has been published in the Orphanet Journal of Rare Diseases. The study reveals a need for implementation of awareness measures on rare diseases in China, as well as legislative measures to boost the healthcare, treatment, well-being and social services provided to patients and care-givers.

Quality of life of transplanted children and their parents: a cross-sectional study

A study on the quality of life of transplanted children and their parents compared to children with other chronic diseases has been published in the Orphanet Journal of Rare Diseases. The study highlights the fact that transplanted children and their parents have shown a fairly preserved quality of life that therapeutic management could help increase.

Factors for perceived barriers to employment success for Neurofibromatosis Type 1 patients

A study has been published in the Orphanet Journal of Rare Diseases comparing the impact of quality of life and depression and anxiety on barriers to successful employment for people diagnosed with Neurofibromatosis Type 1. The study reveals that quality of life of patients suffering from Neurofibromatosis Type 1 is affected by barriers to successful employment, and that these barriers affect both mental health and quality of life.

The impact of rare chronic neurological disease in childhood on the quality of life of families

A study on the impact of severe rare chronic neurological disease in childhood on the quality of life of families has been published in the Orphanet Journal of Rare Diseases. The study focuses on MLD and PCH2 and highlights that these conditions constitute a heavy burden on mothers in particular, but also the entire family. Counselling, professional support as well as increased awareness are recommended to improve the quality of life of these families.

The psychological burden of Val50Met hereditary transthyretin amyloidosis

A study on the psychosocial burden of Val50Met hereditary transthyretin amyloidosis has been published in the Orphanet Journal of Rare Diseases. The study sheds light on the impact of the disease on the quality of life and mental health of patients and their families. Lack of information on diagnosis, psychological distress and negative impact on daily life carry a heavy burden on Val50Met hereditary transthyretin amyloidosis patients and relatives.

Clinical, genetic and quality-of-life study of adult patients with tuberous sclerosis

A study on the clinical and genetic characteristics, therapeutic management and quality of life of a cohort of adult patients with tuberous sclerosis has been published in the Orphanet Journal of Rare Diseases. The study reveals that the quality of life of patients with tuberous sclerosis is inferior to the general population, especially in terms of psychiatric and social needs. These findings suggest the need for improved psychosocial counselling.

Health-related quality of life for patients with CLN2 disease

A study on the utility value determination for patients with CLN2 disease to investigate health-related quality of life has been published in Orphanet Journal of Rare Diseases. The study sheds light on the negative impact CLN2 disease has on health-related quality of life of patients. Utility values however show that an early treatment with cerliponase alfa can be beneficial in the case of CLN2.

Anxiety and depression in school-age patients with spinal muscular atrophy in China

A study investigating anxiety and depression in school-age patients with spinal muscular atrophy in China has been published in the Orphanet Journal of Rare Diseases. The study sheds light on the high rates of psychological distress in this population, showing the need for increased professional psychological services, disease awareness and public information.

Quality-of-life impact of ravulizumab and eculizumab in paroxysmal nocturnal hemoglobinuria

A study has been published in the Orphanet Journal of Rare Diseases on the impact of ravulizumab and eculizumab in paroxysmal nocturnal hemoglobinuria on the quality-of-life of patients. The study reveals that people suffering from paroxysmal nocturnal hemoglobinuria who received ravulizumab or eculizumab treatments had a better quality of life levels than the general populations. The study also highlights that the treatments enabled adaptive changes.

Perceived mental health in parents of children with rare congenital surgical diseases

A study has been published in the Orphanet Journal of Rare Diseases on the perceived mental health in parents of children with rare congenital surgical diseases. The study reveals that resources within and outside the family have a mediating role on perceived stress in the mother. The study also highlights the fact that mothers of children with rare congenital surgical diseases reported higher levels of stress, anxiety and depression.

The impacts of having an infant or young child with achondroplasia on parent well-being

A qualitative study on the impacts of having an infant or young child with achondroplasia on parent well-being has been published in the Orphanet Journal of Rare Diseases. The study reveals the several impacts the children condition has on parents well-being, including caretaking responsibilities, impacts on emotional well-being and impacts on daily well-being.

Registries and biobanks

The de novo FAIRification process of a registry for vascular anomalies

A new study describing the development of a process making a rare disease registry for vascular anomalies FAIR from its conception—de novo has been published in the Orphanet Jounral of Rare Diseases. This study highlights how the FAIR process enables data collection across multiple resources allowing the generation of FAIR data that can be used in the field of vascular anomalies.

Lessons learned from haemolytic uremic syndrome registries

A new study assessing haemolytic uremic syndrome registries has been published in the Orphanet Journal of Rare Diseases. The study aims to compare different features of hemolytic uremic syndrome registries and provides recommendations for the development and implementation of future registries.

10 years of the CEMARA database in the AnDDI-Rares network

A new study has been published in the Orphanet Journal of Rare Diseases on the CEMARA registry, a French rare diseases data repository. The study aims to assess the benefits and challenges of the CEMARA database 10 years after its implementation by the French Ministry of Health. The study highlights that the database is a unique resource for therapeutic studies on rare diseases and that the data included in the registry will be available in the French National Rare Disease Registry that is to be deployed throughout the national network of expert centres.

The LORIS MyeliNeuroGene DataBase for Rare Diseases

A new study has been published in the Orphanet Journal of Rare Diseases on the customisation of the LORIS database in order to develop a new online database focusing on disease-causing genes and the natural history of rare disorders of genetic origin. The aim of the online LORIS MyeliNeuroGene DataBase is to increase international collaboration for data collection and dissemination in order to help emerging clinical trials on rare diseases and gene therapies.

Data management

Barriers to data sharing in the EU

The Joint Action Towards the European Health Data Space (TEHDAS) has conducted a literature review to analyse the existing evidence of barriers to health data sharing in Europe. The review shows that the majority of the barriers experiences by researchers and policy-makers are due to the differences in legal interpretation by Member States, including issues related to infrastructure, data management, trust and transparency. According to the review, the most prominent barrier to health data sharing is due to the differences in governance and health data systems in Europe, as well as the different derogations made possible under the General Data Protection Regulation and the lack of a common European interpretation of what constitutes ‘sufficient anonymisation’ to transform personal data to non-personal data.

TEHDAS highlights need to review European policies on health data access

The Joint Action Towards the European Health Data Space (TEHDAS) has published its second report examining potential health data governance in the EU. The report reveals that regulations regarding access, sharing and use of health data are currently specific to each Member State, while national public health institutes are the ones responsible for the collection of health data. The creation of a European Health Data Space as proposed by the European Commission in its priorities for 2019-2025 would promote better cross-border exchange and access to health data not only to support healthcare but also health research and health policy making. While the European Health Data Space will constitute the legal basis for a single health data market, the report describes key elements of existing and proposed mechanisms to protect health data at the EU level.

Bioinformatics and biotechnology

The VGG-16 genetic syndromes screening model

A new study has been published in the Orphanet Journal of Rare Diseases on a new facial recognition model for the identification of children with genetic syndromes, the VGG-16. The study reveals that genetic syndromes could be identified through facial recognition technology and could be used in clinical practice.

Cybernic treatment with a wearable cyborg for rare neuromuscular disease patients

A study evaluating the efficacy of cybernic treatment with a wearable cyborg for rare neuromuscular disease patients has been published in the Orphanet Journal of Rare Diseases. The study reveals that the wearable cyborg Hybrid Assistive Limb has improved the ambulatory function in patients with rare neuromuscular diseases, as observed in gait function and total manual muscle testing score.

Improving early diagnosis of rare diseases using natural language processing

A new study has been published in the Orphanet Journal of Rare Diseases on the diagnosis of the Dravet Syndrome using natural language processing in unstructured medical records. The study highlights that the Dravet Syndrome could be diagnosed sooner in patients when medical reports are analysed by software exploiting Natural Language Processing, which is able to detect clinical terms that are more used in the electronic narrative medical reports of individuals with the disease before the age of 2 years.

Summary of the edition of 08 October 2021

Editorial
Political news
Scientific news
And also...