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非典型溶血性尿毒症综合征中与抗补体因子H自身抗体相关的肺炎支原体感染

Mycoplasma pneumoniae Infection Associated with Anti-Factor H Autoantibodies in Atypical Hemolytic Uremic Syndrome.

作者信息

Valoti Elisabetta, Piras Rossella, Mele Caterina, Alberti Marta, Liguori Lucia, Breno Matteo, Bertulli Cristina, Bresin Elena, Donadelli Roberta

机构信息

Department of Rare Diseases, Istituto di Ricerche Farmacologiche Mario Negri - IRCCS, Bergamo, Italy.

Department of Pediatrics, Azienda Ospedaliero-Universitaria di Bologna, Bologna, Italy.

出版信息

Nephron. 2022;146(6):593-598. doi: 10.1159/000523998. Epub 2022 Apr 11.

Abstract

Hemolytic uremic syndrome (HUS) is a rare disease characterized by hemolytic anemia, thrombocytopenia, and renal impairment mostly triggered by strains of Shiga-like toxin-producing Escherichia coli (STEC-HUS). A rarer form of HUS, defined as atypical HUS (aHUS), is associated with genetic or acquired dysregulation of the alternative pathway of the complement system and presents a poorer prognosis than STEC-HUS. Factor H autoantibodies (anti-FHs) have been reported in aHUS in 5-11% of cases and are strongly associated with the homozygous deletion of CFHR3-CFHR1 genes. In the large majority of patients, anti-FH-associated aHUS is commonly preceded by gastrointestinal or respiratory tract infections. Here, we described the clinical case of a 3-year-old boy who was hospitalized for aHUS preceded by Mycoplasma pneumoniae (MP) infection. He resulted positive for anti-FHs and carried the homozygous deletion of CFHR3-CFHR1. Of relevance, he also showed a variant of unknown significance in the C5 gene. The patient was successfully treated with eculizumab and achieved hematological and renal remission. The anti-FH titer decreased, became negative after 6 months of mycophenolate mofetil (MMF) treatment, and remained negative for 21-month follow-up indicating that immunosuppression was effective and could prevent the reappearance of anti-FHs. We hypothesized that MP, likely through an evasion strategy of immunosurveillance based on binding of pathogen to FH, triggers anti-FH antibody generation and aHUS in a subject genetically predisposed. In conclusion, to the best of our knowledge, here, we reported the first case of anti-FH-mediated aHUS after an MP infection who benefited from eculizumab and immunosuppressive therapy based on MMF. Hence, monitoring of anti-FHs in patients with post-MP infection glomerulonephritis could be recommended, especially in those with low C3 plasma levels.

摘要

溶血尿毒综合征(HUS)是一种罕见疾病,其特征为溶血性贫血、血小板减少和肾功能损害,主要由产志贺样毒素大肠杆菌菌株(STEC-HUS)引发。一种更罕见的HUS形式,定义为非典型HUS(aHUS),与补体系统替代途径的遗传或获得性失调相关,且预后比STEC-HUS更差。在5%-11%的aHUS病例中报告了H因子自身抗体(抗-FHs),并且与CFHR3-CFHR1基因的纯合缺失密切相关。在绝大多数患者中,抗-FH相关的aHUS通常在胃肠道或呼吸道感染之后出现。在此,我们描述了一名3岁男孩的临床病例,他因肺炎支原体(MP)感染后发生aHUS而住院。他的抗-FHs检测呈阳性,并且携带CFHR3-CFHR1的纯合缺失。值得注意的是,他在C5基因中还表现出一种意义不明的变异。该患者接受依库珠单抗治疗成功,实现了血液学和肾脏缓解。抗-FH滴度下降,在霉酚酸酯(MMF)治疗6个月后变为阴性,并且在21个月的随访中一直保持阴性,表明免疫抑制有效且可防止抗-FHs再次出现。我们推测,MP可能通过基于病原体与H因子结合的免疫监视逃避策略,在遗传易感个体中触发抗-FH抗体产生和aHUS。总之,据我们所知,在此我们报告了首例MP感染后抗-FH介导的aHUS病例,该病例受益于依库珠单抗和基于MMF的免疫抑制治疗。因此,对于MP感染后肾小球肾炎患者,尤其是那些血浆C3水平较低的患者,建议监测抗-FHs。

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