Literature DB >> 32062004

Self-reported baseline phenotypes from the International Fibrodysplasia Ossificans Progressiva (FOP) Association Global Registry.

Robert J Pignolo1, Kin Cheung2, Sammi Kile3, Mary Anne Fitzpatrick4, Carmen De Cunto5, Mona Al Mukaddam6, Edward C Hsiao7, Genevieve Baujat8, Patricia Delai9, Elisabeth M W Eekhoff10, Maja Di Rocco11, Zvi Grunwald12, Nobuhiko Haga13, Richard Keen14, Benjamin Levi15, Rolf Morhart16, Christiaan Scott17, Adam Sherman18, Keqin Zhang19, Fredrick S Kaplan20.   

Abstract

A global, patient-reported registry has been established to characterize the course of disease and track clinical outcomes in patients with fibrodysplasia ossificans progressiva (FOP), an ultra-rare genetic condition of progressive heterotopic ossification (HO) that results in ankylosis of joints and renders most affected individuals immobile by the second decade of life. Here, we present baseline phenotypes on 299 patients (median age 21 years; range 0.1 to 78 years) from 54 countries based on aggregate data from the International FOP Association (IFOPA) Global Registry (the "FOP Registry"). The mean current age of the patients is 23.7 years (range, 0.1 to 78 years). Baseline characteristics are presented for FOP diagnosis, HO, flare-ups and precedent events, system-based prevalent symptomatology, encounters with medical and dental care providers, Patient Reported Outcomes Measurement Information System (PROMIS) Global Health Scale scores, physical function, as well as the use of aids, assistive devices, and adaptations. Correlations of PROMIS Global Health scores with HO burden and physical function are calculated. Associations of joint mobility with PROMIS Global Health scores, physical function, and use of aids, assistive devices, and adaptations are summarized. Overall, the FOP Registry database contains a broad sample of the global FOP patient population, providing a useful tool for expanding knowledge of FOP, designing clinical trials and facilitating evidence-based decisions about the optimal monitoring and management of affected individuals.
Copyright © 2020 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Fibrodysplasia ossificans progressiva; Heterotopic ossification; Patient registry; Physical functioning; Quality of life

Mesh:

Year:  2020        PMID: 32062004     DOI: 10.1016/j.bone.2020.115274

Source DB:  PubMed          Journal:  Bone        ISSN: 1873-2763            Impact factor:   4.398


  6 in total

1.  Suppressed prefrontal cortex oscillations associate with clinical pain in fibrodysplasia ossificans progressiva.

Authors:  Ke Peng; Keerthana Deepti Karunakaran; Robert Labadie; Miranda Veliu; Chandler Cheung; Arielle Lee; Paul B Yu; Jaymin Upadhyay
Journal:  Orphanet J Rare Dis       Date:  2021-01-30       Impact factor: 4.123

Review 2.  Genomic Context and Mechanisms of the ACVR1 Mutation in Fibrodysplasia Ossificans Progressiva.

Authors:  Roberto Ravazzolo; Renata Bocciardi
Journal:  Biomedicines       Date:  2021-02-05

Review 3.  Fibrodysplasia Ossificans Progressiva: What Have We Achieved and Where Are We Now? Follow-up to the 2015 Lorentz Workshop.

Authors:  Ruben D de Ruiter; Bernard J Smilde; Gerard Pals; Nathalie Bravenboer; Petra Knaus; Ton Schoenmaker; Esmée Botman; Gonzalo Sánchez-Duffhues; Maurizio Pacifici; Robert J Pignolo; Eileen M Shore; Marjolein van Egmond; Hans Van Oosterwyck; Frederick S Kaplan; Edward C Hsiao; Paul B Yu; Renata Bocciardi; Carmen Laura De Cunto; Patricia Longo Ribeiro Delai; Teun J de Vries; Susanne Hilderbrandt; Richard T Jaspers; Richard Keen; Peter Koolwijk; Rolf Morhart; Jan C Netelenbos; Thomas Rustemeyer; Christiaan Scott; Clemens Stockklausner; Peter Ten Dijke; James Triffit; Francesc Ventura; Roberto Ravazzolo; Dimitra Micha; Elisabeth M W Eekhoff
Journal:  Front Endocrinol (Lausanne)       Date:  2021-11-10       Impact factor: 6.055

4.  Social and clinical impact of COVID-19 on patients with fibrodysplasia ossificans progressiva.

Authors:  Samuel Kou; Sammi Kile; Sai Samhith Kambampati; Evelyn C Brady; Hayley Wallace; Carlos M De Sousa; Kin Cheung; Lauren Dickey; Kelly L Wentworth; Edward C Hsiao
Journal:  Orphanet J Rare Dis       Date:  2022-03-04       Impact factor: 4.123

Review 5.  Current challenges and opportunities in the care of patients with fibrodysplasia ossificans progressiva (FOP): an international, multi-stakeholder perspective.

Authors:  Robert J Pignolo; Christopher Bedford-Gay; Amanda Cali; Michelle Davis; Patricia L R Delai; Kristi Gonzales; Candace Hixson; Alastair Kent; Hope Newport; Manuel Robert; Christiaan Scott; Frederick S Kaplan
Journal:  Orphanet J Rare Dis       Date:  2022-04-18       Impact factor: 4.303

Review 6.  The Survey of Cells Responsible for Heterotopic Ossification Development in Skeletal Muscles-Human and Mouse Models.

Authors:  Łukasz Pulik; Bartosz Mierzejewski; Maria A Ciemerych; Edyta Brzóska; Paweł Łęgosz
Journal:  Cells       Date:  2020-05-26       Impact factor: 6.600

  6 in total

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