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

BACKGROUND: COVID-19, caused by the SARS-CoV-2 virus, is a severe inflammatory condition. Patients with pre-existing conditions including diabetes, hypertension, and cardiovascular disease are at particularly high risk of complications. Fibrodysplasia ossificans progressiva (FOP) is an ultra-rare an...

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Autores principales: Kou, Samuel, Kile, Sammi, Kambampati, Sai Samhith, Brady, Evelyn C., Wallace, Hayley, De Sousa, Carlos M., Cheung, Kin, Dickey, Lauren, Wentworth, Kelly L., Hsiao, Edward C.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8894823/
https://www.ncbi.nlm.nih.gov/pubmed/35246171
http://dx.doi.org/10.1186/s13023-022-02246-4
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author Kou, Samuel
Kile, Sammi
Kambampati, Sai Samhith
Brady, Evelyn C.
Wallace, Hayley
De Sousa, Carlos M.
Cheung, Kin
Dickey, Lauren
Wentworth, Kelly L.
Hsiao, Edward C.
author_facet Kou, Samuel
Kile, Sammi
Kambampati, Sai Samhith
Brady, Evelyn C.
Wallace, Hayley
De Sousa, Carlos M.
Cheung, Kin
Dickey, Lauren
Wentworth, Kelly L.
Hsiao, Edward C.
author_sort Kou, Samuel
collection PubMed
description BACKGROUND: COVID-19, caused by the SARS-CoV-2 virus, is a severe inflammatory condition. Patients with pre-existing conditions including diabetes, hypertension, and cardiovascular disease are at particularly high risk of complications. Fibrodysplasia ossificans progressiva (FOP) is an ultra-rare and debilitating genetic disorder that is characterized by a pro-inflammatory state, which leads to progressive heterotopic ossification and complications after trauma, including intramuscular vaccinations. To better understand the impact of COVID-19 on patients with FOP, we first examined the social impact of the pandemic using data from the FOP Registry managed by the International FOP Association. We also identified patients with FOP who were exposed to or contracted the SARS-CoV-2 virus, or who received a COVID-19 vaccine, to investigate if patients with FOP were at increased risks of complications from SARS-CoV2 exposure or vaccination. RESULTS: Data from 326 individuals in 69 countries in the International FOP Association FOP Connection Registry were examined using patient-reported outcomes measurement information system (PROMIS) global health scale scores. Twenty-six (28.9%) participants aged ≥ 15 years old rated their satisfaction with their social activities and relationships as poor in 2020, which was an increase from 18 (18.9%) in 2019, prior to the SARS-CoV-2 outbreak. Similar trends were noted for physical and mental health in the pediatric population. Frequency of physician visits was not changed, but a larger portion of patients reported missing dental visits in 2020 compared with 2019 (31.5% vs. 41.7%). A second cohort with 32 subjects was tracked after SARS-CoV-2 exposure or vaccination. Ten subjects were positively diagnosed with COVID-19, 15 received a COVID-19 vaccine, and seven had high-risk SARS-CoV-2 exposure but either did not have a confirmed clinical diagnosis or tested negative. Subjects who tested positive for the virus showed no major complications or increased FOP disease activity, though our sample size is very limited. Among the 15 subjects who received a COVID-19 vaccine, using the International Clinical Council on FOP guidelines for prophylaxis with ibuprofen or acetaminophen, only one person experienced flare-like activity at the injection site. CONCLUSIONS: Patients with FOP showed a significant decrease in social activities that was reflective of the isolation and mobility changes in this debilitated population. In our limited cohort, the majority of the patients with FOP who tested positive for COVID-19 showed no major complications. Also, although limited in sample size, the majority of patients who received a COVID-19 vaccination and followed guidelines from the FOP International Clinical Council tolerated vaccination well. Only one person experiencing flare activity following their injection. Thus, the risks and benefits of COVID-19 vaccination needs to be discussed carefully so as to support informed decisions.
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spelling pubmed-88948232022-03-04 Social and clinical impact of COVID-19 on patients with fibrodysplasia ossificans progressiva Kou, Samuel Kile, Sammi Kambampati, Sai Samhith Brady, Evelyn C. Wallace, Hayley De Sousa, Carlos M. Cheung, Kin Dickey, Lauren Wentworth, Kelly L. Hsiao, Edward C. Orphanet J Rare Dis Research BACKGROUND: COVID-19, caused by the SARS-CoV-2 virus, is a severe inflammatory condition. Patients with pre-existing conditions including diabetes, hypertension, and cardiovascular disease are at particularly high risk of complications. Fibrodysplasia ossificans progressiva (FOP) is an ultra-rare and debilitating genetic disorder that is characterized by a pro-inflammatory state, which leads to progressive heterotopic ossification and complications after trauma, including intramuscular vaccinations. To better understand the impact of COVID-19 on patients with FOP, we first examined the social impact of the pandemic using data from the FOP Registry managed by the International FOP Association. We also identified patients with FOP who were exposed to or contracted the SARS-CoV-2 virus, or who received a COVID-19 vaccine, to investigate if patients with FOP were at increased risks of complications from SARS-CoV2 exposure or vaccination. RESULTS: Data from 326 individuals in 69 countries in the International FOP Association FOP Connection Registry were examined using patient-reported outcomes measurement information system (PROMIS) global health scale scores. Twenty-six (28.9%) participants aged ≥ 15 years old rated their satisfaction with their social activities and relationships as poor in 2020, which was an increase from 18 (18.9%) in 2019, prior to the SARS-CoV-2 outbreak. Similar trends were noted for physical and mental health in the pediatric population. Frequency of physician visits was not changed, but a larger portion of patients reported missing dental visits in 2020 compared with 2019 (31.5% vs. 41.7%). A second cohort with 32 subjects was tracked after SARS-CoV-2 exposure or vaccination. Ten subjects were positively diagnosed with COVID-19, 15 received a COVID-19 vaccine, and seven had high-risk SARS-CoV-2 exposure but either did not have a confirmed clinical diagnosis or tested negative. Subjects who tested positive for the virus showed no major complications or increased FOP disease activity, though our sample size is very limited. Among the 15 subjects who received a COVID-19 vaccine, using the International Clinical Council on FOP guidelines for prophylaxis with ibuprofen or acetaminophen, only one person experienced flare-like activity at the injection site. CONCLUSIONS: Patients with FOP showed a significant decrease in social activities that was reflective of the isolation and mobility changes in this debilitated population. In our limited cohort, the majority of the patients with FOP who tested positive for COVID-19 showed no major complications. Also, although limited in sample size, the majority of patients who received a COVID-19 vaccination and followed guidelines from the FOP International Clinical Council tolerated vaccination well. Only one person experiencing flare activity following their injection. Thus, the risks and benefits of COVID-19 vaccination needs to be discussed carefully so as to support informed decisions. BioMed Central 2022-03-04 /pmc/articles/PMC8894823/ /pubmed/35246171 http://dx.doi.org/10.1186/s13023-022-02246-4 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Research
Kou, Samuel
Kile, Sammi
Kambampati, Sai Samhith
Brady, Evelyn C.
Wallace, Hayley
De Sousa, Carlos M.
Cheung, Kin
Dickey, Lauren
Wentworth, Kelly L.
Hsiao, Edward C.
Social and clinical impact of COVID-19 on patients with fibrodysplasia ossificans progressiva
title Social and clinical impact of COVID-19 on patients with fibrodysplasia ossificans progressiva
title_full Social and clinical impact of COVID-19 on patients with fibrodysplasia ossificans progressiva
title_fullStr Social and clinical impact of COVID-19 on patients with fibrodysplasia ossificans progressiva
title_full_unstemmed Social and clinical impact of COVID-19 on patients with fibrodysplasia ossificans progressiva
title_short Social and clinical impact of COVID-19 on patients with fibrodysplasia ossificans progressiva
title_sort social and clinical impact of covid-19 on patients with fibrodysplasia ossificans progressiva
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8894823/
https://www.ncbi.nlm.nih.gov/pubmed/35246171
http://dx.doi.org/10.1186/s13023-022-02246-4
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