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Effects of COVID -19 pandemic and lockdown on people with multiple system atrophy participating in a therapeutic education program

2021; Elsevier BV; Volume: 86; Linguagem: Inglês

10.1016/j.parkreldis.2021.03.021

1873-5126

Ana Cámara, Yaroslau Compta, Alexandra Pérez‐Soriano, Núria Montagut, M. Baixauli, L. Maragall, E. Ludeña, Juan Carlos Lopez de los Reyes, L. Peri-Cusí, Noemí Fernández, S. Villote, Maribel Ahuir, Anna Grau, Núria Caballol, Maríateresa Buongiorno, Claustre Pont‐Sunyer, Víctor Puente, Darly Milena Giraldo, Oriol de Fábregues, Alícia Garrido, Judith Navarro‐Otano, Cèlia Painous, Almudena Sánchez‐Gómez, Esteban Muñoz, I. Zaro, Donina Obiang, Francesc Valldeoriola, María Lombraña, María‐José Martí,

Cerebral Palsy and Movement Disorders

COVID-19 has been widely studied in people with Parkinson's disease (PwPD) [[1]Santos-García D. Oreiro M. Pérez P. Fanjul G. Paz González J.M. Feal Painceiras M.J. Cores Bartolomé C. Valdés Aymerich L. García Sancho C. Castellanos Rodrigo M. del M. Impact of coronavirus disease 2019 pandemic on Parkinson's disease: a cross-sectional survey of 568 Spanish patients.Mov. Disord. 2020; 35: 1712-1716https://doi.org/10.1002/mds.28261Crossref PubMed Scopus (46) Google Scholar], but besides a study including only nine people with multiple system atrophy (PwMSA) in a cohort of mostly vascular parkinsonism cases [[2]Vignatelli L. Zenesini C. Belotti L.M.B. Baldin E. Bonavina G. Calandra‐Buonaura G. Cortelli P. Descovich C. Fabbri G. Giannini G. Guarino M. Pantieri R. Samoggia G. Scaglione C. Trombetti S. D'Alessandro R. Nonino F. Risk of hospitalization and death for COVID ‐19 in people with Parkinson's disease or parkinsonism.Mov. Disord. 2020; (mds): 28408https://doi.org/10.1002/mds.28408Crossref Scopus (34) Google Scholar], information is lacking on COVID-19 outcomes in PwMSA. In this descriptive multiple-centre study, we included 32 participants in a therapeutic education program during December 2019–February 2020, consisting of 16 PwMSA fulfilling the diagnostic criteria of probable MSA and their respective 16 main caregivers. We classified PwMSA with the disability UMSARS sub-scale as early-intermediate vs. advanced. In July 2020, after the first pandemic wave, we reviewed the clinical charts and administered a semi-structured phone-questionnaire focusing on infection, hospital-admission, symptoms worsening, and perceived impact of the lockdown (Suppl-Table 1). The ethics committee approved the study. All participants signed the informed consent. Data are expressed as absolute and relative frequencies or median and interquartile range (IQR) and compared by Fisher's or Mann-Whitney's U test as appropriate. Statistical significance was p ≤ 0.05. Baseline features in PwMSA were: women = nine (56%); median age-at-onset = 55 (IQR = 48–59); median age-at-inclusion = 60 (IQR = 56–66); median disease-duration = 5 (IQR = 4–7) years; median UMSARS-2 = 24 (IQR = 20–38). Eight (50%) had parkinsonian MSA variant (MSA-P). Eight were in early-intermediate stage (50%). Eleven (68.7%) were following physical and/or speech therapy sessions weekly. Baseline features among caregivers were: women = seven (44%), median age-at-inclusion = 57 (IQR = 40–59) years (no significant differences with PwMSA). Fig. 1-A shows the study flow-chart and Suppl Fig 1-A-D data of infection, admissions and fatality in PwMSA. Frequency of symptomatic COVID-19 was 6% (a respiratory infection case requiring admission, with good recovery). The other three participants who underwent PCR due to respiratory symptoms were negative (75%). Five (31%) were admitted: two due to respiratory infection, two urinary infection and one sudden respiratory failure. Median admission duration was 9 days (IQR = 2–23). Admitted cases had significantly higher UMSARS-2 scores (median = 38.00; IQR = 29.50–43.00) than the rest (median = 22.5; IQR = 17.25–25.25, p = 0.013). Fatality was 25% (N = 4). No autopsies were performed. Three died during the first wave. The cause of death was sepsis (N = 1), sudden death (N = 2, one of whom had been just diagnosed with locally disseminated endometrial cancer after a respiratory infection with negative SARS-CoV-2 PCR), and hyperacute respiratory syndrome (N = 1, a patient who died within 24 hours of admission at a stage when PCR for SARS-COV-2 was not widely applied yet in our country, not allowing to rule out COVID-19). One was early-intermediate (12.5%), and three were advanced (37.5%; p = 0.57). Two had MSA-P (50%). Median age-at-onset, age-at-inclusion, disease duration and UMSARS-2 did not differ between participants who died and survivors (data not shown). No caregivers had symptoms, tested positive, were admitted, nor died. Among the 13 PwMSA surviving the first wave, symptoms that worsened most were gait (100%), speech (69.2%), psychiatric (61.5%), dysautonomic (46.1%), dyskinesia (30.7%), and sleep (23%) features (Fig. 1-B,C,D,E). Among caregivers, 46% reported anxiety. Of these 13 PwMSA, 31% considered that they worsened due to stopping physiotherapy, 23% due to interruption of speech therapy and 15% due to other (social and family) problems (Fig. 1-F). To the best of our knowledge, besides a prior survey of different parkinsonisms [[2]Vignatelli L. Zenesini C. Belotti L.M.B. Baldin E. Bonavina G. Calandra‐Buonaura G. Cortelli P. Descovich C. Fabbri G. Giannini G. Guarino M. Pantieri R. Samoggia G. Scaglione C. Trombetti S. D'Alessandro R. Nonino F. Risk of hospitalization and death for COVID ‐19 in people with Parkinson's disease or parkinsonism.Mov. Disord. 2020; (mds): 28408https://doi.org/10.1002/mds.28408Crossref Scopus (34) Google Scholar], this is the first specific study of PwMSA during the COVID-19 pandemic. The frequency of COVID-19 was 6%, similar to PwPD [[1]Santos-García D. Oreiro M. Pérez P. Fanjul G. Paz González J.M. Feal Painceiras M.J. Cores Bartolomé C. Valdés Aymerich L. García Sancho C. Castellanos Rodrigo M. del M. Impact of coronavirus disease 2019 pandemic on Parkinson's disease: a cross-sectional survey of 568 Spanish patients.Mov. Disord. 2020; 35: 1712-1716https://doi.org/10.1002/mds.28261Crossref PubMed Scopus (46) Google Scholar]. This low figure could be due to important motor disability and strict lockdown, as suggested for advanced PD [[1]Santos-García D. Oreiro M. Pérez P. Fanjul G. Paz González J.M. Feal Painceiras M.J. Cores Bartolomé C. Valdés Aymerich L. García Sancho C. Castellanos Rodrigo M. del M. Impact of coronavirus disease 2019 pandemic on Parkinson's disease: a cross-sectional survey of 568 Spanish patients.Mov. Disord. 2020; 35: 1712-1716https://doi.org/10.1002/mds.28261Crossref PubMed Scopus (46) Google Scholar]. Fatality in about six months was 25%, higher than the ~10% of the six-month follow-up periods in the European and American MSA natural history studies, and median disease-duration of deceased participants was 6.5 years in contrast to 9.8 years in the aforementioned studies [[3]Wenning G.K. Geser F. Krismer F. Seppi K. Duerr S. Boesch S. Köllensperger M. Goebel G. Pfeiffer K.P. Barone P. Pellecchia M.T. Quinn N.P. Koukouni V. Fowler C.J. Schrag A. Mathias C.J. Giladi N. Gurevich T. Dupont E. Ostergaard K. Nilsson C.F. Widner H. Oertel W. Eggert K.M. Albanese A. Del Sorbo F. Tolosa E. Cardozo A. Deuschl G. Hellriegel H. Klockgether T. Dodel R. Sampaio C. Coelho M. Djaldetti R. Melamed E. Gasser T. Kamm C. Meco G. Colosimo C. Rascol O. Meissner W.G. Tison F. Poewe W. The natural history of multiple system atrophy: a prospective European cohort study.Lancet Neurol. 2013; 12: 264-274https://doi.org/10.1016/S1474-4422(12)70327-7Abstract Full Text Full Text PDF PubMed Scopus (386) Google Scholar,[4]Low P.A. Reich S.G. Jankovic J. Shults C.W. Stern M.B. Novak P. Tanner C.M. Gilman S. Marshall F.J. Wooten F. Racette B. Chelimsky T. Singer W. Sletten D.M. Sandroni P. Mandrekar J. Natural history of multiple system atrophy in the USA: a prospective cohort study.Lancet Neurol. 2015; 14: 710-719https://doi.org/10.1016/S1474-4422(15)00058-7Abstract Full Text Full Text PDF PubMed Scopus (230) Google Scholar]. These differences might be related to the pandemic and lockdown setting, or, alternatively, could be merely stochastic in the setting of small sample. Frequent worsening of gait, speech and dysautonomia are in line with MSA natural history. Additionally, low mood, irritability, anxiety and insomnia are not surprising in the pandemic and lockdown context. Anxiety was also frequent among caregivers, probably due to the burden of caregiving and social isolation. The fact that more than 50% of PwMSA perceived that the lockdown was harmful due to interruption of physiotherapy and speech therapy is in line with a recently published study where physiotherapy was helpful in MSA [[5]Raccagni C. Goebel G. Gaßner H. Granata R. Ndayisaba J.P. Seebacher B. Schoenherr G. Mitterhuber J. Hendriks P. Kaindlstorfer C. Eschlboeck S. Fanciulli A. Krismer F. Seppi K. Poewe W. Bloem B.R. Klucken J. Wenning G.K. Physiotherapy improves motor function in patients with the Parkinson variant of multiple system atrophy: a prospective trial.Park. Relat. Disord. 2019; 67: 60-65https://doi.org/10.1016/j.parkreldis.2019.09.026Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar]. The main limitation of this study is the small sample inevitable in a rare disease such as MSA. Also, only PwMSA with respiratory symptoms were PCR-tested for SARS-CoV-2, since at the time of the first pandemic wave in our country PCR was only applied to people with symptoms; hence we cannot rule out additional cases of asymptomatic COVID-19 in our cohort. This limitation is important when interpreting fatality, as only one of the four deceased cases had a negative PCR for SARS-CoV-2. Due to the lockdown restrictions, no autopsies were performed and we used an "ad-hoc" short semi-structured questionnaire instead of published and validated questionnaires. As strengths this is the first study specifically describing MSA outcomes during the pandemic and lockdown, and the semi-structured questionnaire asked open questions to avoid inducing replies. In conclusion, we recorded a single case of COVID-19 infection, yet the mortality was 25%. Gait, speech and psychiatric features were the symptoms that worsened most. Impairment during the lockdown was related to interruption of physical and speech therapies according to the perception of PwMSA. Data can be shared with a qualified researcher upon reasonable request, following an approval by the responsible ethical committee. The authors have no conflicts of interest relevant to this particular study. We are most grateful to all the participants, as without their generosity none of this research would have been possible. This study is part of a project funded by the PERIS (Pla Estratègic de Recerca i Innovació per la Salut) 2019–2020 from Departament de Salut de la Generalitat de Catalunya (PI: A Cámara; SLT008/18/00191). The co-authors from Parkinson's Diseagse & Movement Disorders Unit, Hospital Clinic are members of the European Reference Network for Rare Neurological Diseases – Project ID No 739510. Our institution is supported by the CERCA programme of the Generalitat de Catalunya. The following are the Supplementary data to this article: Download .docx (.02 MB) Help with docx files Multimedia component 1 Download .jpg (.52 MB) Help with files Supplementary Fig. 1Panel A: Pie diagram of the COVID-19 involvement in our case-series. Panel B: Pie diagram of PCR testing for SARS-CoV-2 in our sample. Panel C: Bar diagram of the hospital admissions and their reasons. Panel D: Ring diagram of fatality.