Community Consensus Guidelines to Support FAIR Data Standards in Clinical Research Studies in Primary Mitochondrial Disease.

FAIR standards clinical trials data sharing primary mitochondrial disease therapeutic developments

Journal

Advanced genetics (Hoboken, N.J.)
ISSN: 2641-6573
Titre abrégé: Adv Genet (Hoboken)
Pays: United States
ID NLM: 101774320

Informations de publication

Date de publication:
Mar 2022
Historique:
entrez: 23 3 2022
pubmed: 24 3 2022
medline: 24 3 2022
Statut: ppublish

Résumé

Primary mitochondrial diseases (PMD) are genetic disorders with extensive clinical and molecular heterogeneity where therapeutic development efforts have faced multiple challenges. Clinical trial design, outcome measure selection, lack of reliable biomarkers, and deficiencies in long-term natural history data sets remain substantial challenges in the increasingly active PMD therapeutic development space. Developing "FAIR" (findable, accessible, interoperable, reusable) data standards to make data sharable and building a more transparent community data sharing paradigm to access clinical research metadata are the first steps to address these challenges. This collaborative community effort describes the current landscape of PMD clinical research data resources available for sharing, obstacles, and opportunities, including ways to incentivize and encourage data sharing among diverse stakeholders. This work highlights the importance of, and challenges to, developing a unified system that enables clinical research structured data sharing and supports harmonized data deposition standards across clinical consortia and research groups. The goal of these efforts is to improve the efficiency and effectiveness of drug development and improve understanding of the natural history of PMD. This initiative aims to maximize the benefit for PMD patients, research, industry, and other stakeholders while acknowledging challenges related to differing needs and international policies on data privacy, security, management, and oversight.

Identifiants

pubmed: 35317023
doi: 10.1002/ggn2.202100047
pmc: PMC8936395
mid: NIHMS1771325
pii:
doi:

Types de publication

Journal Article

Langues

eng

Subventions

Organisme : NICHD NIH HHS
ID : U24 HD093483
Pays : United States
Organisme : NINDS NIH HHS
ID : U54 NS078059
Pays : United States

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Auteurs

Amel Karaa (A)

Department of Genetics, Massachusetts General Hospital, Harvard Medical School, Boston, MA 02114, USA.

Laura E MacMullen (LE)

Mitochondrial Medicine Frontier Program, Division of Human Genetics, Department of Pediatrics, Children's Hospital of Philadelphia, Philadelphia, PA 19104, USA.

John C Campbell (JC)

Minovia Therapeutics, Cambridge, MA 02138, USA.

John Christodoulou (J)

Murdoch Children's Research Institute and Department of Paediatrics, University of Melbourne, Melbourne, Victoria 3052, Australia.

Bruce H Cohen (BH)

Department of Pediatrics and the Rebecca D. Considine Research Institute, Akron Children's Hospital, Akron, OH 44308, USA.

Thomas Klopstock (T)

Friedrich-Baur Institute, Department of Neurology, University Hospital, LMU, Munich 80336, Germany; German Center for Neurodegenerative Diseases (DZNE), Munich 80336, Germany; Munich Cluster for Systems Neurology (SyNergy), Munich 80336, Germany; German Network for Mitochondrial Disorders (mitoNET), Munich 80336, Germany.

Yasutoshi Koga (Y)

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume 830-0011, Japan.

Costanza Lamperti (C)

UO Genetics and Neurogenetics, Fondazione IRCCS Instituto Neurologico C. Besta, Milan 20126, Italy.

Rob van Maanen (R)

Khondrion BV., Nijmegen 6525 EX, The Netherlands.

Robert McFarland (R)

WCMR Newcastle University, Newcastle NE1 7RU, UK.

Sumit Parikh (S)

Cleveland Clinic Foundation, Cleveland, OH 44195, USA.

Shamima Rahman (S)

UCL Great Ormond Street Institute of Child Health and Great Ormond, Street Hospital for Children NHS Foundation Trust, London WC1N 1EH, UK.

Fernando Scaglia (F)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA; Texas Children's Hospital, Houston, TX 77030, USA; Joint BCM-CUHK Center of Medical Genetics, Prince of Wales Hospital, Hong Kong SAR NT, China.

Alexander V Sherman (AV)

Department of Neurology, Massachusetts General Hospital, Harvard Medical School, Boston, MA 02114, USA.

Philip Yeske (P)

United Mitochondrial Disease Foundation, Pittsburgh, PA 15239, USA.

Marni J Falk (MJ)

Mitochondrial Medicine Frontier Program, Division of Human Genetics, Department of Pediatrics, Children's Hospital of Philadelphia, Philadelphia, PA 19104, USA; Department of Pediatrics, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA 19104, USA.

Classifications MeSH