{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T15:13:32Z","timestamp":1791213212321,"version":"4.1.0"},"reference-count":23,"publisher":"Wiley","issue":"11","license":[{"start":{"date-parts":[[2021,10,18]],"date-time":"2021-10-18T00:00:00Z","timestamp":1634515200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clin Experimental Allergy"],"published-print":{"date-parts":[[2021,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Palmar hyperlinearity is a feature of ichthyosis vulgaris, the monogenic skin disorder caused by\n                      <jats:italic>FLG<\/jats:italic>\n                      loss\u2010of\u2010function mutations.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To investigate how well the presence or absence of hyperlinear palms (HLP) detect FLG genotype in children.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>STARD criteria are used to report this diagnostic accuracy study. Phenotype and genotype data (four most prevalent FLG null mutations) were obtained from a total of 3656 children in three studies: the UK CLOTHES trial (children 1\u20135\u00a0years with moderate\u2013severe atopic eczema); UK BEEP trial (2\u00a0year olds at high risk of developing atopic eczema); UK\u2010Irish eczema case collection (0\u201316\u00a0year olds with atopic eczema). All participants included in analyses of HLP as the index test and FLG genotype as the reference were of white European ancestry.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Thirty\u2010two percent of participants (1159\/3656) had FLG null mutation(s) and 37% (1347\/3656) had HLP. In 13% (464\/3656), HLP was recorded as \u2018unsure\u2019 or not recorded. The sensitivity and specificity of HLP for detecting FLG mutations in each of the studies was: 67% (95% CI 55\u201378%) and 75% (67\u201382%) in CLOTHES; 46% (36\u201355%) and 89% (86\u201391%) in BEEP; 72% (68\u201375%) and 60% (57\u201362%) in the UK\u2010Irish case collection. Positive and negative likelihood ratios were: 2.73 (1.95\u20133.81) and 0.44 (0.31\u20130.62) in CLOTHES; 4.02 (2.99\u20135.40) and 0.61 (0.52\u20130.73) in BEEP; 1.79 (1.66\u20131.93) and 0.47 (0.42\u20130.53) in the UK\u2010Irish collection.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>Trained observers were able to define palmar hyperlinearity in the majority (3191\/3656, 87%) of cases. The presence of HLP is not a reliable sign to detect FLG mutations, but the absence of HLP excludes FLG null genotype with a reasonable degree of certainty.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/cea.14025","type":"journal-article","created":{"date-parts":[[2021,10,6]],"date-time":"2021-10-06T04:12:08Z","timestamp":1633493528000},"page":"1421-1428","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Clinical examination for hyperlinear palms to determine filaggrin genotype: A diagnostic test accuracy study"],"prefix":"10.1111","volume":"51","author":[{"given":"Lucy E.","family":"Bradshaw","sequence":"first","affiliation":[{"name":"Nottingham Clinical Trials Unit University of Nottingham Nottingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel H.","family":"Haines","sequence":"additional","affiliation":[{"name":"Nottingham Clinical Trials Unit University of Nottingham Nottingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kim S.","family":"Thomas","sequence":"additional","affiliation":[{"name":"Centre of Evidence Based Dermatology University of Nottingham Nottingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joanne R.","family":"Chalmers","sequence":"additional","affiliation":[{"name":"Centre of Evidence Based Dermatology University of Nottingham Nottingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9048-2044","authenticated-orcid":false,"given":"Alan D.","family":"Irvine","sequence":"additional","affiliation":[{"name":"Clinical Medicine Trinity College Dublin Dublin Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hywel C.","family":"Williams","sequence":"additional","affiliation":[{"name":"Centre of Evidence Based Dermatology University of Nottingham Nottingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3232-5251","authenticated-orcid":false,"given":"Sara J.","family":"Brown","sequence":"additional","affiliation":[{"name":"Centre for Genomic and Experimental Medicine Institute of Genetics and Cancer University of Edinburgh Edinburgh UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2021,10,18]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng1743"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2133.2009.09339.x"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng1767"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.jid.5700587"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2011.01.031"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2008.01.026"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.b2433"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1038\/jid.2011.393"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/jdv.15786"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjd.15191"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pmed.1002280"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(19)32984-8"},{"issue":"170","key":"e_1_2_11_14_1","first-page":"e1","article-title":"Chromosome 11q13.5 variant associated with childhood eczema: an effect supplementary to filaggrin mutations","volume":"125","author":"O'Regan GM","year":"2010","journal-title":"J Allergy Clin Immunol"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.3424"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng2020"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmjopen-2016-012799"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2008.01.013"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2014.08.005"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2020.01.033"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2133.1996.d01-925.x"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1600-0625.2001.100102.x"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1001\/archderm.140.12.1513"},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2133.2000.03300.x"}],"container-title":["Clinical &amp; 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