藥物性過敏症候群後遺症的白斑症病例

摘 要：

藥物誘導的過敏綜合徵（DIHS）是一種嚴重的藥物不良反應，其發病率和死亡率較高。已有報告指出，DIHS患者隨後可能發展為自身免疫性疾病，並可能隨之發生器官功能失調。我們報告了一名47歲的女性，服用了雪諾昔布和磺胺沙拉嗪1個月後出現發燒、全身性斑丘疹、面部水腫和嗜酸性白血球增多，伴有肝功能受損。患者被診斷為明確的DIHS。由於反復發作，患者接受了約1.5年的免疫抑制劑治療，包括全身性類固醇。檢測到人類第六型疱疹病毒的再活化，以及可能的巨細胞病毒再活化。DIHS發病後4個月，發現皮膚全身性色素沉著不足和白髮。組織病理學檢查確診了白斑症的診斷。在DIHS發病後4年，觀察到一些自發性色素重塑，未進行特定治療。進一步的免疫血清學研究顯示，我們的患者血漿中C-X-C模鍵結蛋白10水平升高，與白斑症活動有關。DIHS後廣泛性白斑症的發生是一種極為罕見的情況。該病例提醒醫師在DIHS後監測此類嚴重併發症的重要性。

Drug-induced hypersensitivity syndrome (DIHS) is a type of severe drug adverse reaction with high morbidity and mortality. DIHS patients have been reported to subsequently develop autoimmune disease, which may be followed by end-organ decompensation. We report a 47-year-old woman who presented with fever, generalized maculopapular eruption, facial edema and eosinophilia with liver function impairment after taking celecoxib and sulfasalazine for 1 month. The patient was diagnosed with definite DIHS. The patient was treated with immunosuppressants including systemic corticosteroid for approximately 1.5 years due to recurrent episodes. Reactivation of human herpesvirus 6 and possible reactivation of cytomegalovirus were detected. Generalized hypopigmentation of the skin and leukotrichia were noted 4 months after the onset of DIHS. Histopathological examination confirmed the diagnosis of vitiligo. Some spontaneous repigmentation was noted 4 years after DIHS without specific treatment. Further immunoserology study showed elevated plasma C-X-C motif chemokine 10 level, which is related to vitiligo activity, in our patient. The occurrence of widespread vitiligo after DIHS is an extremely rare condition. This case provides an important reminder for physicians to monitor such severe complications after DIHS.