{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,24]],"date-time":"2026-02-24T11:58:46Z","timestamp":1771934326778,"version":"3.50.1"},"reference-count":21,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2020,11,17]],"date-time":"2020-11-17T00:00:00Z","timestamp":1605571200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100011773","name":"Conseil r\u00e9gional de Bourgogne-Franche-Comt\u00e9","doi-asserted-by":"publisher","award":["STIC11"],"award-info":[{"award-number":["STIC11"]}],"id":[{"id":"10.13039\/501100011773","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Special Care in Dentistry"],"published-print":{"date-parts":[[2021,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Aims<\/jats:title><jats:p>Cohen syndrome (CS) is an uncommon autosomal recessive disorder due to mutations in vacuolar protein sorting 13B, with an intermittent presence of neutropenia. Contrary to other clinical phenotypic features, oral health has been little investigated in CS. We described oral health and dental hygiene in a cohort of CS patients.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods and Results<\/jats:title><jats:p>Twelve CS patients with neutropenia (&lt;1500\/mm<jats:sup>3<\/jats:sup>) were recruited in the dental department of Dijon University Hospital (France). Patients underwent oral examination, and blood samples were collected. Oral health markers were described and compared between patients with moderate and severe neutropenia (&lt;500\/mm<jats:sup>3<\/jats:sup>). In 12 patients (mean age = 21.1 years, SD = 13.7, six females), 45.5% brushed at least twice daily their teeth, and the same percentage annually visited a dentist. Dental plaque index was high (mean = 1.7, SD = 1.4). So was the number of lost teeth per patient, notably among adults (mean = 13.8, SD = 9.8). Elevated markers of periodontitis were noted as percentage of bleeding dental sites (mean = 70.2%, SD = 45.2%) or Gingival Index (mean = 2.2, SD = 1.0). The severity of neutropenia was correlated to the level of tooth\u2010loss (<jats:italic>P<\/jats:italic> = .03).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>This study highlighted in CS patients worrisome oral health and dental follow\u2010up in the context of intellectual disability with behavioural anomalies. More attention is needed by care\u2010givers on oral condition in CS.<\/jats:p><\/jats:sec>","DOI":"10.1111\/scd.12544","type":"journal-article","created":{"date-parts":[[2020,11,17]],"date-time":"2020-11-17T19:27:04Z","timestamp":1605641224000},"page":"118-124","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Periodontal disorders in a cohort of patients with Cohen syndrome"],"prefix":"10.1111","volume":"41","author":[{"given":"Arnaud","family":"Lafon","sequence":"first","affiliation":[{"name":"Facult\u00e9 d'odontologie Universit\u00e9 de Lyon Universit\u00e9 Lyon 1  Lyon France"},{"name":"Hospices Civils de Lyon Service de Consultations et de Traitements Dentaires  Lyon France"},{"name":"Enseignant chercheur du Laboratoire Parcours de Sant\u00e9 Syst\u00e9mique EA 4129\u2010Ecole Doctorale EDISS Universit\u00e9 Claude Bernard Lyon 1  Lyon 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France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Brigitte","family":"Grogogeat","sequence":"additional","affiliation":[{"name":"Facult\u00e9 d'odontologie Universit\u00e9 de Lyon Universit\u00e9 Lyon 1  Lyon France"},{"name":"Hospices Civils de Lyon Service de Consultations et de Traitements Dentaires  Lyon France"},{"name":"Universit\u00e9 de Lyon LMI UMR CNRS 5615  Lyon France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anahide","family":"Marcelet","sequence":"additional","affiliation":[{"name":"Liberal Dental Surgeon"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3694-7699","authenticated-orcid":false,"given":"Laurent","family":"Laforest","sequence":"additional","affiliation":[{"name":"Facult\u00e9 d'odontologie Universit\u00e9 de Lyon Universit\u00e9 Lyon 1  Lyon 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