{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T02:18:45Z","timestamp":1787624325755,"version":"build-2736575974"},"reference-count":46,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2026,7,14]],"date-time":"2026-07-14T00:00:00Z","timestamp":1783987200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,7,14]],"date-time":"2026-07-14T00:00:00Z","timestamp":1783987200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/501100000265","name":"Medical Research Council","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000265","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000866","name":"Cardiff University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000866","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Genes Brain and Behavior"],"published-print":{"date-parts":[[2026,8]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Steroid sulfatase (STS) cleaves sulphate groups from steroid hormones. In humans, STS deficiency is associated with X\u2010linked ichthyosis, an increased predisposition to neurodevelopmental and mood conditions (including Attention Deficit Hyperactivity Disorder, autism, depression and anxiety), and cardiac arrhythmia risk. Until recently, no single\u2010gene \u2018knockout\u2019 mammalian model existed; previous work in such a model is limited to skin phenotypes. We generated a novel C57BL\/6J mouse model with a deletion in exon 2 of\n                    <jats:italic>Sts<\/jats:italic>\n                    . We examined gene expression and enzyme activity in liver and brain samples of homozygous mice, and assessed the breeding performance and health of male and female deletion\u2010carriers. Subsequently, we compared performance across a range of behavioural paradigms in wildtype and homozygous male and female mice: elevated plus maze, open field, rotarod, spontaneous alternation, and acoustic startle\/prepulse inhibition. We also investigated serum steroid hormone levels by liquid chromatography\u2010mass spectrometry and measured heart weights and two morphological indices (bodyweight\/tibia length)\n                    <jats:italic>post mortem<\/jats:italic>\n                    . Homozygous mice almost completely lacked STS expression\/activity. Genetically\u2010altered mice exhibited grossly\u2010normal breeding performance, health, and endocrinology. Homozygous mice were more active and had higher normalised heart weights than wildtype mice. We also found significant genotype\u2009\u00d7\u2009sex interactions on bodyweight and on two behavioural measures (potentially reflecting lower anxiety in homozygous males and heightened anxiety in homozygous females). The \u2018\n                    <jats:italic>Sts<\/jats:italic>\n                    \u2010deletion\u2019 mouse represents an experimentally\u2010tractable model in which to identify and characterise phenotypes associated with STS deficiency. The mechanistic basis of the associations described here requires further investigation, and whether these translate to humans remains to be tested.\n                  <\/jats:p>","DOI":"10.1111\/gbb.70061","type":"journal-article","created":{"date-parts":[[2026,7,15]],"date-time":"2026-07-15T04:30:07Z","timestamp":1784089807000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Physiological and Behavioural Characterisation of a Novel Steroid Sulfatase\u2010Deficient Mouse"],"prefix":"10.1111","volume":"25","author":[{"given":"Trevor","family":"Humby","sequence":"first","affiliation":[{"name":"School of Psychology, Cardiff University  Cardiff UK"},{"name":"Neuroscience and Mental Health Innovation Institute Cardiff University  Cardiff UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Freya R.","family":"Shepherd","sequence":"additional","affiliation":[{"name":"Neuroscience and Mental Health Innovation Institute Cardiff University  Cardiff UK"},{"name":"Division of Psychological Medicine and Clinical Neurosciences and Centre for Neuropsychiatric Genetics and Genomics School of Medicine, Cardiff University  Cardiff UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Talia","family":"Elgie","sequence":"additional","affiliation":[{"name":"School of Psychology, Cardiff University  Cardiff UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Libby","family":"Anderson\u2010Watkins","sequence":"additional","affiliation":[{"name":"School of Biosciences, Cardiff University  Cardiff UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lucy I.","family":"Beevors","sequence":"additional","affiliation":[{"name":"Department of Metabolism and Systems Science College of Medicine and Health, University of Birmingham  Birmingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5835-5643","authenticated-orcid":false,"given":"Angela E.","family":"Taylor","sequence":"additional","affiliation":[{"name":"Department of Metabolism and Systems Science College of Medicine and Health, University of Birmingham  Birmingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7190-1592","authenticated-orcid":false,"given":"Paul A.","family":"Foster","sequence":"additional","affiliation":[{"name":"Department of Metabolism and Systems Science College of Medicine and Health, University of Birmingham  Birmingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7714-2440","authenticated-orcid":false,"given":"William","family":"Davies","sequence":"additional","affiliation":[{"name":"School of Psychology, Cardiff University  Cardiff UK"},{"name":"Neuroscience and Mental Health Innovation Institute Cardiff University  Cardiff UK"},{"name":"Division of Psychological Medicine and Clinical Neurosciences and Centre for Neuropsychiatric Genetics and Genomics School of Medicine, Cardiff University  Cardiff UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,7,14]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1210\/er.2015\u20101036"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejmg.2021.104169"},{"key":"e_1_2_11_4_1","unstructured":"GTEx Portal \u201cGene Expression \u201d2026 https:\/\/www.gtexportal.org\/home\/gene\/STS."},{"key":"e_1_2_11_5_1","unstructured":"Mouse Genome Informatics \u201cGenes and Markers \u201daccessed March 27 2026 https:\/\/www.informatics.jax.org\/marker\/key\/13669."},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaad.2009.04.028"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/ski2.179"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet\u20102019\u2010106676"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/gbb.12893"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/ced.15116"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg\u20102022\u2010108862"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1042\/EBC20230098"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1093\/ced\/llaf221"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.biopsych.2009.01.001"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.psyneuen.2011.06.006"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1038\/npp.2011.314"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.psyneuen.2012.12.002"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1038\/npp.2014.115"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1101\/2022.03.26.485930"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/s0006\u20108993(97)00867\u20106"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467\u2010020\u201020269\u2010y"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11033\u2010023\u201009066\u20100"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0596\u201083"},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbadis.2023.167004"},{"key":"e_1_2_11_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbamcr.2025.119898"},{"key":"e_1_2_11_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ymeth.2017.03.016"},{"key":"e_1_2_11_27_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2016\u20103716"},{"key":"e_1_2_11_28_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jchromb.2022.123413"},{"key":"e_1_2_11_29_1","doi-asserted-by":"publisher","DOI":"10.1016\/s0006\u20108993(01)03171\u20107"},{"key":"e_1_2_11_30_1","doi-asserted-by":"publisher","DOI":"10.1161\/HYPERTENSIONAHA.110.166157"},{"key":"e_1_2_11_31_1","doi-asserted-by":"publisher","DOI":"10.1186\/1471\u20102156\u20109\u201083"},{"key":"e_1_2_11_32_1","unstructured":"Mouse Genome Informatics \u201cInbred Strains of Mice: C57BL \u201d2026 https:\/\/www.informatics.jax.org\/inbred_strains\/mouse\/docs\/C57BL.shtml."},{"key":"e_1_2_11_33_1","unstructured":"The Jackson Laboratory \u201cC57BL\/6J \u201d2026 https:\/\/www.jax.org\/strain\/000664."},{"key":"e_1_2_11_34_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijms242316822"},{"key":"e_1_2_11_35_1","doi-asserted-by":"publisher","DOI":"10.1111\/cei.13437"},{"key":"e_1_2_11_36_1","first-page":"782","article-title":"Incidence Rates of Spontaneous Disease in Laboratory Mice Used at a Large Biomedical Research Institution","volume":"52","author":"Marx J. O.","year":"2013","journal-title":"Journal of the American Association for Laboratory Animal Science"},{"key":"e_1_2_11_37_1","first-page":"611","volume-title":"Small Animal Critical Care Medicine","author":"Kutzler M. A.","year":"2008"},{"key":"e_1_2_11_38_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0212330"},{"key":"e_1_2_11_39_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0164417"},{"key":"e_1_2_11_40_1","doi-asserted-by":"publisher","DOI":"10.1111\/acps.12845"},{"key":"e_1_2_11_41_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12888\u2010020\u201002707\u20109"},{"key":"e_1_2_11_42_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12915\u2010017\u20100463\u20106"},{"key":"e_1_2_11_43_1","doi-asserted-by":"publisher","DOI":"10.1016\/bs.vh.2025.08.002"},{"key":"e_1_2_11_44_1","doi-asserted-by":"publisher","DOI":"10.1186\/2040\u20102392\u20105\u201021"},{"key":"e_1_2_11_45_1","doi-asserted-by":"publisher","DOI":"10.1194\/jlr.R300005\u2010JLR200"},{"key":"e_1_2_11_46_1","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCEP.125.014195"},{"key":"e_1_2_11_47_1","doi-asserted-by":"publisher","DOI":"10.1101\/2024.06.03.597045"}],"container-title":["Genes, Brain and Behavior"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/gbb.70061","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/gbb.70061","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/gbb.70061","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T01:21:12Z","timestamp":1787620872000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/gbb.70061"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,7,14]]},"references-count":46,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2026,8]]}},"alternative-id":["10.1111\/gbb.70061"],"URL":"https:\/\/doi.org\/10.1111\/gbb.70061","archive":["Portico"],"relation":{},"ISSN":["1601-1848","1601-183X"],"issn-type":[{"value":"1601-1848","type":"print"},{"value":"1601-183X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,7,14]]},"assertion":[{"value":"2026-03-27","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-01","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-14","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70061"}}