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Propylthiouracil, a member of thiouracils group, is widely used in medical treatment of hyperthyroidism. Propylthiouracil is associated with multiple adverse effects such as rash, agranulocytosis hepatitis and antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis, but rarely triggers DRESS\/DiHS syndrome. Here, we describe a severe case of propylthiouracil-induced DRESS\/DiHS syndrome.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>A 38-year-old female was treated with methimazole for hyperthyroidism at first. 4\u2009weeks later, the patient developed elevated liver transaminase so methimazole was stopped. After liver function improved in 2\u2009weeks, medication was switched to propylthiouracil\u00a0therapy. The patient subsequently developed nausea and rash followed by a high fever, acute toxic hepatitis and multiple organ dysfunction (liver, lung and heart), which lasted for 1\u2009month after propylthiouracil was started. According to the diagnostic criteria, the patient was diagnosed of DRESS\/DiHS syndrome which was induced by propylthiouracil. As a result, propylthiouracil was immediately withdrawn. And patient was then treated with adalimumab, systematic corticosteroids and plasmapheresis in sequence. Symptoms were finally resolved 4\u2009weeks later.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>Propylthiouracil is a rare cause of the DRESS\/DiHS syndrome, which typically consists of severe dermatitis and various degrees of internal organ involvement. We want to emphasize through this severe case that DRESS\/DiHS syndrome should be promptly recognized to hasten recovery.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12902-023-01273-x","type":"journal-article","created":{"date-parts":[[2023,1,23]],"date-time":"2023-01-23T11:03:03Z","timestamp":1674471783000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["DRESS\/DiHS syndrome induced by Propylthiouracil: a case report"],"prefix":"10.1186","volume":"23","author":[{"given":"Qiong","family":"Shen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qingyao","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Huanhuan","family":"Zang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ling","family":"Yu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiangguo","family":"Cong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3929-2314","authenticated-orcid":false,"given":"Xinxin","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lei","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2023,1,23]]},"reference":[{"issue":"10","key":"1273_CR1","doi-asserted-by":"publisher","first-page":"1343","DOI":"10.1089\/thy.2016.0229","volume":"26","author":"DS Ross","year":"2016","unstructured":"Ross DS, Burch HB, Cooper DS, et al. 2016 American Thyroid Association Guidelines for Diagnosis and Management of Hyperthyroidism and Other Causes of Thyrotoxicosis. 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