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一名男性青少年中与新型NLRP12突变相关的自身炎症性复发性心包炎

Autoinflammatory Recurrent Pericarditis Associated with a New NLRP12 Mutation in a Male Adolescent.

作者信息

Cinteza Eliza, Stefan Dan, Iancu Mihaela Adela, Ioan Andreea, Vasile Corina Maria, Vatasescu Radu, Cochino Alexis

机构信息

Department of Pediatrics, "Carol Davila" University of Medicine and Pharmacy, 020021 Bucharest, Romania.

Department of Pediatric Cardiology, "Marie Curie" Emergency Children's Hospital, 41451 Bucharest, Romania.

出版信息

Life (Basel). 2023 Oct 28;13(11):2131. doi: 10.3390/life13112131.

Abstract

Idiopathic recurrent pericarditis (IRP) can be the hallmark of an autoinflammatory syndrome with recurrent attacks of chest pain and symptom-free intervals following an acute episode. The recurrence rate may be 35% in the pediatric population, frequently with less severe manifestations than at the first episode. Pericarditis can be the sole clinical manifestation or may be part of a systemic autoinflammatory disease (SAID), especially in the case of a recurrence. Familial Mediterranean Fever (FMF), Tumor Necrosis Factor Receptor-Associated Periodic Syndrome (TRAPS), Mevalonate-Kinase Deficiency (MKD), nucleotide-binding oligomerization domain 2 (NOD2)-associated autoinflammatory syndrome, and others are closely related to IRP based on similar clinical manifestations and treatment responses to anti-interleukin 1 (IL-1) agents, such as anakinra, and should therefore be excluded in patients with IRP. A newly described SAID, an autosomal dominant disorder known as NLRP12-AID (nucleotide-binding leucine-rich repeat-containing receptor 12-related autoinflammatory disease) is caused by heterozygous mutations in the NLRP12 gene and most commonly affects children. Fewer than 40 pediatric patients with NLRP12-AID have been described in the medical literature, with none presenting with RP. We report a case of relapsing pericarditis responsive to anti-IL-1 therapy in a male adolescent who carried a missense mutation in the NLRP12 gene potentially causative of the excessive activation of inflammatory pathways. This is a unique case in the medical literature that associates recurrent pericarditis in an adolescent presumed to be related to the missense mutation in the NLRP12 gene. The role of the NLRP12 inflammasome in generating and maintaining recurrent pericardial inflammation should be considered.

摘要

特发性复发性心包炎(IRP)可能是一种自身炎症综合征的标志,其特征为胸痛反复发作,急性发作后有症状缓解期。儿科人群的复发率可能为35%,通常表现不如首次发作时严重。心包炎可能是唯一的临床表现,也可能是系统性自身炎症性疾病(SAID)的一部分,尤其是在复发的情况下。家族性地中海热(FMF)、肿瘤坏死因子受体相关周期性综合征(TRAPS)、甲羟戊酸激酶缺乏症(MKD)、核苷酸结合寡聚化结构域2(NOD2)相关自身炎症综合征等,基于类似的临床表现和对抗白细胞介素1(IL-1)药物(如阿那白滞素)的治疗反应,与IRP密切相关,因此IRP患者应排除这些疾病。一种新描述的SAID,一种常染色体显性疾病,称为NLRP12-AID(核苷酸结合富含亮氨酸重复序列受体12相关自身炎症性疾病),由NLRP12基因的杂合突变引起,最常影响儿童。医学文献中描述的患有NLRP12-AID的儿科患者不到40例,无一例表现为复发性心包炎(RP)。我们报告了一例对IL-1治疗有反应的复发性心包炎男性青少年病例,该青少年携带NLRP12基因错义突变,可能导致炎症途径过度激活。这是医学文献中一个独特的病例,将青少年复发性心包炎与NLRP12基因错义突变相关联。应考虑NLRP12炎性小体在引发和维持复发性心包炎症中的作用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/830a/10672620/71326a36dd18/life-13-02131-g001.jpg

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