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      A Multifactorial Case of Stevens-Johnson Syndrome With Atypical Features

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      Author(s): 1 , , 2 , 1 , 3
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      Publication date (Electronic, pub):
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      Journal: Cureus
      Publisher: Cureus
      Keywords: stevens-johnson syndrome (sjs), hypercoagulability, vancomycin infusion, toxic epidermal necrolysis (ten), nikolsky sign

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          Abstract

          Stevens-Johnson syndrome (SJS) is a severe mucocutaneous reaction that has a broad spectrum of causes and risk factors that include medications and other infectious causes such as Mycoplasma. In this case report, a patient with multiple comorbidities that confounded the presentation of Stevens-Johnson syndrome is observed. The patient was a 29-year-old female with a past medical history of recurrent cerebrovascular accident (CVA) who presented for an evaluation of chest pain. After empiric vancomycin was started for suspicion of endocarditis, our patient developed altered mental status, mucositis, and a painful erythematous erosion on her chest concerning for vasculitis, but after treatment and pathological review, it was found to be Stevens-Johnson syndrome. It is important to not forget the wide variety of risk factors for developing Stevens-Johnson syndrome and some of its unique associated presenting symptoms.

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          Current Perspectives on Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis.

          Marianne Lerch, Carlo Mainetti, Benedetta Terziroli Beretta-Piccoli … (2018)
          Stevens-Johnson syndrome (SJS) and toxic epidermal necrolysis (TEN) are considered a delayed-type hypersensitivity reaction to drugs. They represent true medical emergencies and an early recognition and appropriate management is decisive for the survival. SJS/TEN manifest with an "influenza-like" prodromal phase (malaise, fever), followed by painful cutaneous and mucous membrane (ocular, oral, and genital) lesions, and other systemic symptoms. The difference between SJS, SJS/TEN overlap, and TEN is defined by the degree of skin detachment: SJS is defined as skin involvement of  30%, and SJS/TEN overlap as 10-30% skin involvement. The diagnosis of different degrees of epidermal necrolysis is based on the clinical assessment in conjunction with the corresponding histopathology. The mortality rates for SJS and TEN have decreased in the last decades. Today, the severity-of-illness score for toxic epidermal necrolysis (SCORTEN) is available for SJS/TEN severity assessment. Drugs with a high risk of causing SJS/TEN are anti-infective sulfonamides, anti-epileptic drugs, non-steroidal anti-inflammatory drugs of the oxicam type, allopurinol, nevirapine, and chlormezanone. Besides conventional drugs, herbal remedies and new biologicals should be considered as causative agents. The increased risk of hypersensitivity reactions to certain drugs may be linked to specific HLA antigens. Our understanding of the pathogenesis of SJS/TEN has improved: drug-specific T cell-mediated cytotoxicity, genetic linkage with HLA- and non-HLA-genes, TCR restriction, and cytotoxicity mechanisms were clarified. However, many factors contributing to epidermal necrolysis still have to be identified, especially in virus-induced and autoimmune forms of epidermal necrolysis not related to drugs. In SJS/TEN, the most common complications are ocular, cutaneous, or renal. Nasopharyngeal, esophageal, and genital mucosal involvement with blisters, erosions as well as secondary development of strictures also play a role. However, in the acute phase, septicemia is a leading cause of morbidity and fatality. Pulmonary and hepatic involvement is frequent. The acute management of SJS/TEN requires a multidisciplinary approach. Immediate withdrawal of potentially causative drugs is mandatory. Prompt referral to an appropriate medical center for specific supportive treatment is of utmost importance. The most frequently used treatments for SJS/TEN are systemic corticosteroids, immunoglobulins, and cyclosporine A.
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            Recent advances in managing and understanding Stevens-Johnson syndrome and toxic epidermal necrolysis

            Akito Hasegawa, Riichiro Abe (2020)
            Stevens-Johnson syndrome (SJS) and toxic epidermal necrolysis (TEN) are life-threatening diseases characterized by detachment of the epidermis and mucous membrane. SJS/TEN are considered to be on the same spectrum of diseases with different severities. They are classified by the percentage of skin detachment area. SJS/TEN can also cause several complications in the liver, kidneys, and respiratory tract. The pathogenesis of SJS/TEN is still unclear. Although it is difficult to diagnose early stage SJS/TEN, biomarkers for diagnosis or severity prediction have not been well established. Furthermore, optimal therapeutic options for SJS/TEN are still controversial. Several drugs, such as carbamazepine and allopurinol, are reported to have a strong relationship with a specific human leukocyte antigen (HLA) type. This relationship differs between different ethnicities. Recently, the usefulness of HLA screening before administering specific drugs to decrease the incidence of SJS/TEN has been investigated. Skin detachment in SJS/TEN skin lesions is caused by extensive epidermal cell death, which has been considered to be apoptosis via the Fas-FasL pathway or perforin/granzyme pathway. We reported that necroptosis, i.e. programmed necrosis, also contributes to epidermal cell death. Annexin A1, released from monocytes, and its interaction with the formyl peptide receptor 1 induce necroptosis. Several diagnostic or prognostic biomarkers for SJS/TEN have been reported, such as CCL-27, IL-15, galectin-7, and RIP3. Supportive care is recommended for the treatment of SJS/TEN. However, optimal therapeutic options such as systemic corticosteroids, intravenous immunoglobulin, cyclosporine, and TNF-α antagonists are still controversial. Recently, the beneficial effects of cyclosporine and TNF-α antagonists have been explored. In this review, we discuss recent advances in the pathophysiology and management of SJS/TEN.
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              Stevens–Johnson Syndrome and Toxic Epidermal Necrolysis: A Review of Diagnosis and Management

              Robert Frantz, Simo Huang, Abhirup Are … (2021)
              Stevens–Johnson Syndrome (SJS) and Toxic Epidermal Necrolysis (TEN) are rare diseases that are characterized by widespread epidermal necrosis and sloughing of skin. They are associated with significant morbidity and mortality, and early diagnosis and treatment is critical in achieving favorable outcomes for patients. In this scoping review, Excerpta Medica dataBASE and PubMed were searched for publications that addressed recent advances in the diagnosis and management of the disease. Multiple proteins (galectin 7 and RIP3) were identified that are promising potential biomarkers for SJS/TEN, although both are still in early phases of research. Regarding treatment, cyclosporine is the most effective therapy for the treatment of SJS, and a combination of intravenous immunoglobulin (IVIg) and corticosteroids is most effective for SJS/TEN overlap and TEN. Due to the rare nature of the disease, there is a lack of prospective, randomized controlled trials and conducting these in the future would provide valuable insights into the management of this disease.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Cureus
                Journal ID (iso-abbrev): Cureus
                Journal ID (issn): 2168-8184
                Title: Cureus
                Publisher: Cureus (Palo Alto (CA) )
                ISSN (Electronic): 2168-8184
                Publication date (Electronic, pub): 23 November 2023
                Publication date (Electronic, collection): November 2023
                Volume: 15
                Issue: 11
                Electronic Location Identifier: e49289
                Affiliations
                [1 ] Family Medicine, Hospital Corporation of America (HCA) Florida St. Petersburg Hospital, St. Petersburg, USA
                [2 ] Internal Medicine, Nova Southeastern University Dr. Kiran C. Patel College of Osteopathic Medicine, Clearwater, USA
                [3 ] Critical Care, Hospital Corporation of America (HCA) Florida St. Petersburg Hospital, St. Petersburg, USA
                Author notes
                Article
                DOI: 10.7759/cureus.49289
                PMC ID: 10748208
                PubMed ID: 38143660
                SO-VID: e8c2e8d9-ad74-4341-baec-c85c1989218b
                Copyright © Copyright © 2023, Paterno et al.
                License:

                This is an open access article distributed under the terms of the Creative Commons Attribution License CC-BY 4.0., which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

                History
                Date accepted : 22 November 2023
                Categories
                Subject: Internal Medicine
                Subject: Dermatology
                Subject: Infectious Disease

                Keywords: stevens-johnson syndrome (sjs),hypercoagulability,vancomycin infusion,toxic epidermal necrolysis (ten),nikolsky sign
                Data availability:
                Keywords: stevens-johnson syndrome (sjs), hypercoagulability, vancomycin infusion, toxic epidermal necrolysis (ten), nikolsky sign

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