DRESS/DiHS syndrome induced by Propylthiouracil: a case report

Abstract

Background: Drug reaction with eosinophilia and systemic symptoms (DRESS), also known as Drug-induced hypersensitivity syndrome (DiHS), is a severe adverse drug reaction. 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.

Case presentation: A 38-year-old female was treated with methimazole for hyperthyroidism at first. 4 weeks later, the patient developed elevated liver transaminase so methimazole was stopped. After liver function improved in 2 weeks, medication was switched to propylthiouracil therapy. 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 month 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 As a result, propylthiouracil was immediately withdrawn.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 weeks later.

Conclusions: 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.

Keywords: Drug reaction with eosinophilia and systemic symptoms (DRESS); Drug-induced hypersensitivity syndrome (DiHS); Propylthiouracil.

Fig. 1

The diagnosis and treatment timeline of the case

Fig. 2

The detection of liver function and eosinophils throughout the course of disease. Liver function throughout the course of disease was shown in (A) and the absolute counts of eosinophils were shown in (B), respectively

Fig. 3

The detection of CD4 + T and CD8 + T cells in peripheral blood of the patient throughout the course of disease. The percentages of total CD3 + T cells (A), CD3 + CD4 + T cells (B), CD3 + CD8 + T cells (C) were detected in day 2, day 12 and discharge day, respectively. The ratio of CD8+/CD4 + T cells was shown in (D). Day 2 was the day after the first injection of adalimumab (80 mg), day 12 was the day before plasmapheresis