关键词: Burkholderia adult-onset immunodeficiency due to anti-interferon-gamma antibodies anti-IFN-γ autoantibodies drug rash with eosinophilia and systemic symptoms infection sulfamethoxazole

Mesh : Adult Anti-Bacterial Agents / therapeutic use Autoantibodies Drug Hypersensitivity Syndrome / complications etiology Eosinophilia / chemically induced complications diagnosis Exanthema Female Humans Immunologic Deficiency Syndromes Interferon-gamma Middle Aged Mycobacterium Infections, Nontuberculous Trimethoprim, Sulfamethoxazole Drug Combination / adverse effects

来  源:   DOI:10.3389/fimmu.2022.969912   PDF(Pubmed)

Abstract:
A 56-year-old Chinese woman with previous disseminated mycobacterium avium complex infection and recurrent cervical abscesses from Burkholderia cepacia complex visited our hospital. She was diagnosed with adult-onset immunodeficiency (AOID) and tested positive for interferon-γ-neutralizing autoantibody. Ceftazidime was administered as the initial antimicrobial treatment, which was later combined with sulfamethoxazole-trimethoprim (SMZ-TMP). She developed drug rash with eosinophilia and systemic symptoms (DRESS) syndrome after SMZ-TMP administration and improved after withdrawal of the culprit antibiotic and systemic glucocorticoids treatment. Her cervical infection was eventually cured after combined therapy of long-term antibiotics and anti-IFN-γ autoantibodies (AIGA) titer-lowering treatments including glucocorticoids, rituximab, and plasmapheresis. This is the first case of DRESS syndrome in the setting of AIGA-induced AOID and is worthy of notice.
摘要:
一名56岁的中国妇女曾传播过鸟分枝杆菌复合感染,并患有洋葱伯克霍尔德氏菌并发的宫颈脓肿。她被诊断出患有成人发作性免疫缺陷(AOID),并且干扰素-γ中和自身抗体检测呈阳性。头孢他啶作为初始抗菌治疗,后来与磺胺甲恶唑-甲氧苄啶(SMZ-TMP)合用。SMZ-TMP给药后,她出现了伴嗜酸性粒细胞增多和全身症状(DRESS)综合征的皮疹,并在停用罪魁祸首抗生素和全身糖皮质激素治疗后有所改善。在长期抗生素和抗IFN-γ自身抗体(AIGA)降滴度治疗(包括糖皮质激素)的联合治疗后,她的宫颈感染最终治愈。利妥昔单抗,和血浆置换.这是AIGA诱导的AOID背景下DRESS综合征的第一例,值得注意。
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