A Japanese Woman with Stevens-Johnson Syndrome without Skin Lesions Induced by an Initial Dosage of Lamotrigine

Abstract

Atypical Stevens-Johnson syndrome (SJS) which is characterized by little or no skin involment, is reported to be triggered by less medications than infection. There are few reports with atypical SJS induced by medications. This is a case report of atypical SJS induced by Lamotrigine (LTG). A 31-year-old Japanese woman, who was bipolar disorder and prescribed an initial dosage of LTG 25mg/day ten days ago, presented with high fever and throat pain for two days. At first she was suspected of pharyngitis and administered antibiotics. She was hospitalized on the first visit day, but she was getting worse and fell into shock over several days. Furthermore she presented with erosions and bleeding of her lips and oral mucosa on day6, and so she was diagnosed with SJS, but her skin lesions were mild even at the extreme of the clinical course. She gradually developed acute pulmonary failure, liver dysfunction, coma and Disseminated Intravascular Coagulation (DIC). But she was treated with intensive care consisting of steroid pulse followed by high dose corticosteroids, and so she gradually recovered from SJS and corticosteroids could be tapered. At last she was discharged on day50 and had no sequelae with SJS. It was extremely difficult to diagnose this case as atypical SJS because of the unremarkable skin lesions and similarity to pharyngitis. It was also characteristic that she was already in severe status when it satisfied the diagnostic criteria for SJS. But physicians should not miss from the perspective of the fatality and social problem. It should be noted that atypical SJS shows mild or little cutaneous lesions.