{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,18]],"date-time":"2026-02-18T08:21:09Z","timestamp":1771402869494,"version":"3.50.1"},"reference-count":21,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2024,3,22]],"date-time":"2024-03-22T00:00:00Z","timestamp":1711065600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2024,3,22]],"date-time":"2024-03-22T00:00:00Z","timestamp":1711065600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2024,8]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>\n                    Primrose syndrome (PS) is a rare genetic disease characterized by developmental delay, intellectual disability, sensorineural hearing loss, and dysmorphic features. PS is caused by de novo pathogenic variants in the\n                    <jats:italic>ZBTB20<\/jats:italic>\n                    gene, which encodes a transcription factor modulating neurogenesis. We describe resolution with sertraline of neurobehavioral difficulties in a 17\u2010year\u2010old Hispanic male with PS with de novo heterozygous c.1916G\u2009&gt;\u2009A (p.C639Y) variant of\n                    <jats:italic>ZBTB20<\/jats:italic>\n                    . Neurobehavioral difficulties included aggression towards self and others, irritability, tearfulness, and mood liability that did not respond to behavioral interventions or aripiprazole. Treatment with sertraline, a medication indicated for psychiatric disorders including anxiety and depression, led to the resolution of neurobehavioral difficulties after 2\u2009weeks of initiation of medication. The treatment course suggests that selective serotonin reuptake inhibitors, such as sertraline, may be a useful tool for neurobehavioral difficulties in PS over antipsychotics that are accompanied by complex side effect profiles, and suggest that anxiety is the primary cause of the neurobehavioral difficulties in this patient.\n                  <\/jats:p>","DOI":"10.1002\/ajmg.a.63610","type":"journal-article","created":{"date-parts":[[2024,3,22]],"date-time":"2024-03-22T08:44:14Z","timestamp":1711097054000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Resolution of severe neurobehavioral difficulties in an individual with Primrose syndrome with sertraline"],"prefix":"10.1002","volume":"194","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-9196-0441","authenticated-orcid":false,"given":"Young Min","family":"Moon","sequence":"first","affiliation":[{"name":"Department of Psychiatry School of Medicine, Johns Hopkins University  Baltimore Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sa Eun","family":"Park","sequence":"additional","affiliation":[{"name":"Department of Psychiatry School of Medicine, Johns Hopkins University  Baltimore Maryland USA"},{"name":"Department of Psychiatry Kennedy Krieger Institute  Baltimore Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8241-9574","authenticated-orcid":false,"given":"Constance","family":"Smith\u2010Hicks","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medicine, Johns Hopkins Univerisity  Baltimore Maryland USA"},{"name":"Department of Neurology Kennedy Krieger Institute  Baltimore Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aaron","family":"Hauptman","sequence":"additional","affiliation":[{"name":"Department of Psychiatry School of Medicine, Johns Hopkins University  Baltimore Maryland USA"},{"name":"Department of Psychiatry Kennedy Krieger Institute  Baltimore Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2024,3,22]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1749-6632.2003.tb07095.x"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejmg.2020.103967"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/mp.2016.262"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.61024"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.rasd.2012.08.001"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.3035"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1186\/gb-2014-15-4-r56"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1176\/appi.ajp.2009.08111747"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0203760"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1097\/CHI.0b013e3181b76658"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.13749"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1038\/mp.2011.88"},{"key":"e_1_2_8_14_1","first-page":"101","article-title":"A slowly progressive degenerative condition characterized by mental deficiency, wasting of limb musculature and bone abnormalities, including ossification of the pinnae","volume":"26","author":"Primrose D. 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