Levin Laura E, Magro Cynthia, Horowitz James, Harp Joanna
Department of Dermatology, Weill Cornell Medical College, New York, New York, USA.
Department of Pathology, Weill Cornell Medical College, New York, New York, USA.
Cutis. 2017 May;99(5):E25-E29.
Hydralazine-induced small vessel vasculitis is a rare entity with a limited number of cases reported in the dermatologic literature. A characteristic pattern of acral pseudoembolic vesiculopustules with necrosis and ulceration has been suggested along with involvement of the aerodigestive tract, indicating mucosal involvement is an important feature of this disease. We report the case of a patient with hydralazine-induced vasculitis who exemplified this characteristic presentation associated with severe involvement of the aerodigestive tract and gastrointestinal tract bleeding. In addition to the distinctive clinical presentation, the patient's workup revealed the characteristic antineutrophil cytoplasmic antibody (ANCA)-positive serologic profile. Increased recognition of the clinical and serological features of hydralazine-induced small vessel vasculitis may lead to earlier recognition of this disease and decreased time to discontinuation of hydralazine when appropriate. Drug withdrawal is the cornerstone of therapy, and depending on the severity of symptoms, additional immunosuppressive treatment such as corticosteroids may be necessary.
肼屈嗪诱发的小血管血管炎是一种罕见疾病,皮肤病学文献中报道的病例数量有限。已提出一种伴有坏死和溃疡的肢端假栓塞性水疱脓疱的特征性模式,同时累及气道消化道,表明黏膜受累是该疾病的一个重要特征。我们报告了一例肼屈嗪诱发血管炎的患者,该患者体现了这种与气道消化道严重受累及胃肠道出血相关的特征性表现。除了独特的临床表现外,患者的检查还显示出特征性的抗中性粒细胞胞浆抗体(ANCA)阳性血清学特征。对肼屈嗪诱发的小血管血管炎的临床和血清学特征的认识增加,可能会导致更早地识别该疾病,并在适当的时候缩短停用肼屈嗪的时间。停药是治疗的基石,根据症状的严重程度,可能需要额外的免疫抑制治疗,如使用皮质类固醇。