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万古霉素诱导的线状IgA疾病,表现为大疱性多形红斑。

Vancomycin-induced linear IgA disease manifesting as bullous erythema multiforme.

作者信息

Armstrong April Wang, Fazeli Amin, Yeh Shih Wei, Mackool Bonnie T, Liu Vincent

机构信息

Pasteur/Doris Duke Clinical Research Fellowship, Harvard Medical School, Boston, MA, USA.

出版信息

J Cutan Pathol. 2004 May;31(5):393-7. doi: 10.1111/j.0303-6987.2004.00190.x.

Abstract

BACKGROUND

Vancomycin-induced linear immunoglobulin A (IgA) disease, an autoimmune, blistering disease in response to vancomycin administration, is characterized by a subepidermal, vesiculobullous eruption and linear IgA deposition along the basement membrane zone on direct immunofluorescence.

CASE REPORT

We report the case of an 81-year-old man treated with vancomycin who developed diffuse erythema multiforme and tense bullae involving the palmoplantar surfaces. Discontinuation of vancomycin therapy resulted in complete resolution of this patient's cutaneous eruption.

RESULTS

Biopsy of a representative skin lesion demonstrated lichenoid interface dermatitis with focal subepidermal clefting, dyskeratosis, and prominent eosinophils. Direct immunofluorescence showed linear basement membrane staining with immunoreactants to IgA; indirect immunofluorescence demonstrated the presence of circulating IgG antibodies binding in an intercellular pattern. Immunoprecipitation studies using the patient's serum revealed 210, 130, and 83 kDa target antigens.

CONCLUSIONS

Presenting with an initial clinical picture suggestive of bullous erythema multiforme, this patient's subsequent clinical course and direct immunofluorescence confirm the diagnosis of linear IgA bullous disease (LABD). His indirect immunofluorescence findings and immunoprecipitation results suggest that circulating non-IgA antibodies may represent a newly recognized immunopathologic feature of vancomycin-induced linear IgA disease, underscoring the variable and unpredictable manifestations of this drug-induced cutaneous disease.

摘要

背景

万古霉素诱导的线性免疫球蛋白A(IgA)病是一种因使用万古霉素而引发的自身免疫性水疱病,其特征为表皮下水疱大疱性皮疹,直接免疫荧光检查显示沿基底膜带呈线性IgA沉积。

病例报告

我们报告一例81岁男性患者,接受万古霉素治疗后出现累及掌跖面的弥漫性多形红斑和紧张性大疱。停用万古霉素治疗后,该患者的皮肤皮疹完全消退。

结果

对一个具有代表性的皮肤病变进行活检,显示苔藓样界面性皮炎,伴有局灶性表皮下裂隙、角化不良和显著的嗜酸性粒细胞。直接免疫荧光显示基底膜呈线性染色,对IgA有免疫反应物;间接免疫荧光显示存在以细胞间模式结合的循环IgG抗体。使用患者血清进行的免疫沉淀研究显示有210、130和8 kDa的靶抗原。

结论

该患者最初的临床表现提示大疱性多形红斑,随后的临床病程和直接免疫荧光检查证实为线性IgA大疱病(LABD)。其间接免疫荧光结果和免疫沉淀结果表明,循环非IgA抗体可能代表万古霉素诱导的线性IgA病新发现的免疫病理特征,强调了这种药物性皮肤病表现的多样性和不可预测性。

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