Liquidano-Pérez Eduardo, Maza-Ramos Gibert, Yamazaki-Nakashimada Marco Antonio, Barragán-Arévalo Tania, Lugo-Reyes Saúl Oswaldo, Scheffler-Mendoza Selma, Espinosa-Padilla Sara Elva, González-Serrano María Edith
Instituto Nacional de Pediatría, Unidad de Investigación en Inmunodeficiencias, Ciudad de México, México.
The Mayo Clinics Care Network, Médica Sur, Ciudad de México, México.
Rev Alerg Mex. 2022 Jul 1;69(1):31-47. doi: 10.29262/ram.v69i1.1104.
Combinedimmunodeficiency (CID) due to DOCK8 deficiency is an inborn error of immunity (IBD) characterized by dysfunctional T and B lymphocytes; The spectrum of manifestations includes allergy, autoimmunity, inflammation, predisposition to cancer, and recurrent infections. DOCK8 deficiency can be distinguished from other CIDs or within the spectrum of hyper-IgE syndromes by exhibiting profound susceptibility to viral skin infections, associated skin cancers, and severe food allergies. The 9p24.3 subtelomeric locus where DOCK8 is located includes numerous repetitive sequence elements that predispose to the generation of large germline deletions and recombination-mediated somatic DNA repair. Residual production DOCK8 protein contributes to the variable phenotype of the disease. Severe viral skin infections and varicella-zoster virus (VZV)-associated vasculopathy, reflect an essential role of the DOCK8 protein, which is required to maintain lymphocyte integrity as cells migrate through the tissues. Loss of DOCK8 causes immune deficiencies through other mechanisms, including a cell survival defect. In addition, there are alterations in the response of dendritic cells, which explains susceptibility to virus infection and regulatory T lymphocytes that could help explain autoimmunity in patients. Hematopoietic stem cell transplantation (HSCT) is the only curative treatment; it improves eczema, allergies, and susceptibility to infections.
由于DOCK8缺陷导致的联合免疫缺陷(CID)是一种免疫缺陷病(IBD),其特征为T淋巴细胞和B淋巴细胞功能失调;临床表现谱包括过敏、自身免疫、炎症、癌症易感性和反复感染。DOCK8缺陷可通过对病毒性皮肤感染、相关皮肤癌和严重食物过敏的高度易感性,与其他CID或高IgE综合征谱相区分。DOCK8所在的9p24.3亚端粒位点包含许多重复序列元件,易导致大片段种系缺失的产生和重组介导的体细胞DNA修复。DOCK8蛋白的残留产生导致了该疾病的可变表型。严重的病毒性皮肤感染和水痘带状疱疹病毒(VZV)相关血管病反映了DOCK8蛋白的重要作用,当细胞在组织中迁移时,该蛋白是维持淋巴细胞完整性所必需的。DOCK8的缺失通过其他机制导致免疫缺陷,包括细胞存活缺陷。此外,树突状细胞的反应存在改变,这解释了对病毒感染的易感性,而调节性T淋巴细胞可能有助于解释患者的自身免疫。造血干细胞移植(HSCT)是唯一的治愈性治疗方法;它可改善湿疹、过敏和感染易感性。