{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,20]],"date-time":"2026-10-20T05:31:51Z","timestamp":1792474311462,"version":"4.5.0"},"reference-count":48,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2022,7,2]],"date-time":"2022-07-02T00:00:00Z","timestamp":1656720000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,1,5]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Population-based data on the epidemiology of progressive multifocal leukoencephalopathy, its predisposing conditions and mortality rate are lacking, although such data are crucial to raise awareness among clinicians and to lay foundations for future therapeutic trials in immunomodulating therapies.<\/jats:p>\n                  <jats:p>In our study, patients were identified by interrogating the French national healthcare reimbursement database from 1 January 2008 to 31 December 2017, using progressive multifocal leukoencephalopathy International Classification of Diseases code and a patient\u2019s selection algorithm. Overall incidence rate, 1-year all-cause mortality rate and survival patterns were calculated, and factors associated with death were identified using a multivariate Cox proportional hazards regression model.<\/jats:p>\n                  <jats:p>Our cohort is the largest to date, comprising 584 patients with incident progressive multifocal leukoencephalopathy. The overall incidence in France from 2010 to 2017 was stable during the study period at 0.11 per 100 000 person-years, 95% confidence interval [0.10\u20130.12]. Predisposing diseases were HIV infection (43.7%), followed by haematological malignancies (21.9%), chronic inflammatory diseases (20.2%), solid organ transplantation (4.3%), solid neoplasm (4.1%) and primary immune deficiency (1.5%). The 1-year mortality rate was 38.2%, with a 95% confidence interval (34.2\u201342.2). In multivariate analysis, factors independently associated with death were older age [adjusted hazard ratio 0.33 (0.20\u20130.53) for patients aged 20 to 40 compared with patients aged over 60], male gender [adjusted hazard ratio 0.73 (0.54\u20130.99) for females compared with males] and predisposing immunosuppressive disease, with the highest risk for solid neoplasms [adjusted hazard ratio 4.34 (2.25\u20138.37)], followed by haematological malignancies [adjusted hazard ratio 3.13 (1.85\u20135.30)] and HIV infection [adjusted hazard ratio 1.83 (1.12\u20133.00)], compared with chronic inflammatory diseases. Immune reconstitution inflammatory syndrome was notified in 7.0% of patients.<\/jats:p>\n                  <jats:p>In conclusion, incidence of progressive multifocal leukoencephalopathy is stable in France, and HIV infection remains the main predisposing disease. This large-size cohort uncovers a higher risk of mortality for male patients compared to females, and the worst prognosis for patients with solid neoplasm, while prognosis in patients with haematological malignancies appeared less dismal than in previous studies.<\/jats:p>","DOI":"10.1093\/brain\/awac237","type":"journal-article","created":{"date-parts":[[2022,7,2]],"date-time":"2022-07-02T13:36:46Z","timestamp":1656769006000},"page":"349-358","source":"Crossref","is-referenced-by-count":58,"title":["Progressive multifocal leukoencephalopathy: epidemiology and spectrum of predisposing conditions"],"prefix":"10.1093","volume":"146","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0191-2584","authenticated-orcid":false,"given":"Marine","family":"Joly","sequence":"first","affiliation":[{"name":"Department of Infectious and Tropical Diseases, Toulouse University Hospital Center , Toulouse 31300 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C\u00e9cile","family":"Conte","sequence":"additional","affiliation":[{"name":"Department of Medical Pharmacology, CIC 1436, Toulouse University Hospital Center , Toulouse 31300 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charles","family":"Cazanave","sequence":"additional","affiliation":[{"name":"Department of Infectious and Tropical Diseases, Bordeaux University Hospital Center , Bordeaux 33300 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vincent","family":"Le Moing","sequence":"additional","affiliation":[{"name":"Department of Infectious and Tropical Diseases, Montpellier University Hospital Center , Montpellier 34295 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3617-5411","authenticated-orcid":false,"given":"Pierre","family":"Tattevin","sequence":"additional","affiliation":[{"name":"Department of Medical Intensive Care and Infectious Diseases, Rennes University Hospital Center , Rennes 35000 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2874-3581","authenticated-orcid":false,"given":"Pierre","family":"Delobel","sequence":"additional","affiliation":[{"name":"Department of Infectious and Tropical Diseases, Toulouse University Hospital Center , Toulouse 31300 , France"},{"name":"Institut Toulousain des Maladies Infectieuses et Inflammatoires (Infinity) INSERM UMR1291, CNRS UMR5051, University of Toulouse III , Toulouse 31300 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Agne\u0300s","family":"Sommet","sequence":"additional","affiliation":[{"name":"Department of Medical Pharmacology, CIC 1436, Toulouse University Hospital Center , Toulouse 31300 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guillaume","family":"Martin-Blondel","sequence":"additional","affiliation":[{"name":"Department of Infectious and Tropical Diseases, Toulouse University Hospital Center , Toulouse 31300 , France"},{"name":"Institut Toulousain des Maladies Infectieuses et Inflammatoires (Infinity) INSERM UMR1291, CNRS UMR5051, University of Toulouse III , Toulouse 31300 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2022,7,2]]},"reference":[{"key":"2023010803504138800_awac237-B1","doi-asserted-by":"crossref","first-page":"37","DOI":"10.1038\/s41582-020-00427-y","article-title":"Progressive multifocal leukoencephalopathy and the spectrum of JC virus-related disease","volume":"17","author":"Cortese","year":"2021","journal-title":"Nat Rev Neurol"},{"issue":"Pt 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