{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,31]],"date-time":"2026-07-31T03:20:36Z","timestamp":1785468036372,"version":"3.56.0"},"reference-count":24,"publisher":"S. Karger AG","issue":"5","license":[{"start":{"date-parts":[[2022,1,1]],"date-time":"2022-01-01T00:00:00Z","timestamp":1640995200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/www.karger.com\/Services\/SiteLicenses"},{"start":{"date-parts":[[2022,1,1]],"date-time":"2022-01-01T00:00:00Z","timestamp":1640995200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.karger.com\/Services\/SiteLicenses"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Neuroepidemiology"],"published-print":{"date-parts":[[2022]]},"abstract":"<b><i>Background:<\/i><\/b> Essential tremor (ET) is a highly prevalent neurological disease that frequently runs in families. A recent and controversial proposal is to separate ET patients into two distinct groups \u2013 ET versus ET-plus. If this were a valid construct, one would expect in familial aggregation studies to observe that ET-plus would cluster in some families yet be absent in others, rather than being randomly distributed across families. We examined whether there is evidence of familial aggregation of ET-plus. <b><i>Methods:<\/i><\/b> Probands (<i>n<\/i> = 84 [56 ET-plus and 28 ET]) and their first- and second-degree relatives (<i>n<\/i> = 182 and 48) enrolled in a genetics study. \u03c7<sup>2<\/sup> and generalized estimating equations (GEE) tested associations between probands\u2019 ET-plus status and the ET-plus status of their relatives. <b><i>Results:<\/i><\/b> \u03c7<sup>2<\/sup> analyses revealed that ET-plus was no more prevalent in relatives of probands diagnosed with ET-plus than in relatives of probands diagnosed with ET, <i>p<\/i> &#x3e; 0.05. Restricting relatives to first-degree relatives similarly did not detect a significant association (<i>p<\/i> = 0.88). GEE yielded similar results (respective <i>p<\/i>\u2019s = 0.39 and 0.81). <b><i>Conclusion:<\/i><\/b> The data demonstrate that ET-plus does not seem to aggregate in families. As such, they do not lend support to the notion that ET-plus is a valid biological construct. <\/jats:p>","DOI":"10.1159\/000526278","type":"journal-article","created":{"date-parts":[[2022,8,8]],"date-time":"2022-08-08T21:00:32Z","timestamp":1659992432000},"page":"373-379","source":"Crossref","is-referenced-by-count":5,"title":["Lack of Familial Aggregation of the \u201cEssential Tremor-Plus\u201d Phenotype in Familial Essential Tremor"],"prefix":"10.1159","volume":"56","author":[{"given":"Diane S.","family":"Berry","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7038-1492","authenticated-orcid":false,"given":"Nora","family":"Hernandez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lorraine N.","family":"Clark","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elan D.","family":"Louis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"127","published-online":{"date-parts":[[2022,8,8]]},"reference":[{"key":"ref1"},{"key":"ref2"},{"key":"ref3"},{"key":"ref4"},{"key":"ref5"},{"key":"ref6"},{"key":"ref7"},{"key":"ref8"},{"key":"ref9"},{"key":"ref10"},{"key":"ref11"},{"key":"ref12"},{"key":"ref13"},{"key":"ref14"},{"key":"ref15"},{"key":"ref16"},{"key":"ref17"},{"key":"ref18"},{"key":"ref19"},{"key":"ref20"},{"key":"ref21"},{"key":"ref22"},{"key":"ref23"},{"key":"ref24"}],"container-title":["Neuroepidemiology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.karger.com\/Article\/Pdf\/526278","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.karger.com\/Article\/Pdf\/526278","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,23]],"date-time":"2025-04-23T15:14:19Z","timestamp":1745421259000},"score":1,"resource":{"primary":{"URL":"https:\/\/karger.com\/article\/doi\/10.1159\/000526278"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022]]},"references-count":24,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2022,11,3]]},"published-print":{"date-parts":[[2022,11,8]]}},"URL":"https:\/\/doi.org\/10.1159\/000526278","archive":["Portico"],"relation":{},"ISSN":["0251-5350","1423-0208"],"issn-type":[{"value":"0251-5350","type":"print"},{"value":"1423-0208","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022]]}}}