Kato Seiichi, Gold Benjamin D, Kato Ayumu
Kato Children's Clinic, Natori, Japan.
GI Care for Kids, Children's Center for Digestive Healthcare, LLC, Atlanta, GA, United States.
Front Pediatr. 2024 Oct 21;12:1459394. doi: 10.3389/fped.2024.1459394. eCollection 2024.
Immunoglobulin A vasculitis (IgAV), previously known as Henoch-Schönlein purpura, is the most common form of systemic vasculitis in childhood. The primary organs involved are the skin, gastrointestinal (GI) tract, joints, and kidneys. The spectrum of GI involvement in IgAV ranges from being mild and self-limited to severe manifestations often requiring surgical intervention. Galactose-deficient IgA1 on the immunoglobulin hinge region and its immune complexes are thought to play a central pathogenetic role in IgAV, however, an association between such molecules and specific GI mucosal damage remains unclear. GI endoscopy (both upper and lower) shows a variety of mucosal findings, many of which are not specific for IgAV. In upper GI endoscopy, however, the mucosal features can be diagnostic when found localized in the more distal part of upper GI tract (second and/or third parts of the duodenum). Abdominal computed tomography and capsule endoscopy have demonstrated that the small intestine is most commonly involved in IgAV. The GI mucosal involvement when evaluated microscopically shows IgA deposition which is histologically diagnostic. Conversely, leukocytoclastic vasculitis is less useful. Since the 1960s, cases of duodenojejunitis, in which IgAV was suspected but evident purpura was not dermatologically present, have often been labeled as "idiopathic". In a pediatric case series, IgA enteropathy, without dermatological manifestations (i.e., purpura), was reported to have similar symptoms, as well as endoscopic characteristics and immunohistological findings as in IgAV. Subsequently, several case reports provide additional supportive evidence that IgA enteropathy must be a variant of IgAV. Thus, the immunologically driven auto-immune vasculitis results in the symptom complex dependent on the organ system involved, and the subsequent clinical features which are manifested. Present classification criteria are useful and universally available for diagnosing IgAV. However, based upon current knowledge including IgA enteropathy, minor modification of the IgAV criteria is proposed in the review.
免疫球蛋白A血管炎(IgAV),以前称为过敏性紫癜,是儿童期最常见的系统性血管炎形式。主要受累器官为皮肤、胃肠道(GI)、关节和肾脏。IgAV胃肠道受累的范围从轻度自限性到严重表现,常需手术干预。免疫球蛋白铰链区的半乳糖缺乏型IgA1及其免疫复合物被认为在IgAV的发病机制中起核心作用,然而,这些分子与特定胃肠道黏膜损伤之间的关联仍不清楚。胃肠道内镜检查(包括上消化道和下消化道)显示出多种黏膜表现,其中许多对IgAV并不具有特异性。然而,在上消化道内镜检查中,当黏膜特征局限于上消化道更远端(十二指肠第二和/或第三部分)时,可具有诊断意义。腹部计算机断层扫描和胶囊内镜检查表明,小肠是IgAV最常受累的部位。显微镜下评估时,胃肠道黏膜受累表现为IgA沉积,具有组织学诊断价值。相反,白细胞破碎性血管炎的诊断价值较小。自20世纪60年代以来,那些怀疑为IgAV但皮肤无明显紫癜的十二指肠空肠炎病例,常被标记为“特发性”。在一个儿科病例系列中,据报道无皮肤表现(即紫癜)的IgA肠病具有与IgAV相似的症状、内镜特征和免疫组织学表现。随后,一些病例报告提供了更多支持性证据,表明IgA肠病必定是IgAV的一种变体。因此,免疫驱动的自身免疫性血管炎导致了取决于受累器官系统的症状复合体及其随后出现的临床特征。目前的分类标准对于诊断IgAV很有用且普遍适用。然而,基于包括IgA肠病在内的现有知识,本综述提出对IgAV标准进行微小修改。