Shillitoe Ben, Gennery Andrew
Institute of Cellular Medicine, Newcastle University, 4th Floor, William Leech Building, Medical School, Framlington Place, Newcastle upon Tyne NE2 4HH, United Kingdom.
Institute of Cellular Medicine, Newcastle University, 4th Floor, William Leech Building, Medical School, Framlington Place, Newcastle upon Tyne NE2 4HH, United Kingdom.
Clin Immunol. 2017 Oct;183:54-62. doi: 10.1016/j.clim.2017.07.008. Epub 2017 Jul 17.
Colonel Ogden Bruton reported X-Linked Agammaglobulinaemia in 1952 and treated the child with replacement immunoglobulin therapy. Over 60years later, the treatment for XLA has largely remained unchanged. Replacement immunoglobulin lacks the isotypes IgA and IgM, leading to concerns that patients continue to experience recurrent sinopulmonary tract infections and be at increased risk of bronchiectasis. There is potential hope of earlier diagnosis with newborn screening, and a potential cure for these patients, in the form of gene therapy. However, it is first necessary to evaluate current management and outcomes to aid decisions regarding further research and clinical trials. This article reviews current management and outcomes of XLA, whilst identifying gaps in our knowledge base that may need answering before we proceed with novel diagnostic methods and treatment for XLA.
奥格登·布鲁顿上校于1952年报告了X连锁无丙种球蛋白血症,并采用替代免疫球蛋白疗法治疗该患儿。60多年后,X连锁无丙种球蛋白血症的治疗方法在很大程度上仍未改变。替代免疫球蛋白缺乏IgA和IgM同种型,这引发了人们对患者持续经历反复的鼻窦肺部感染以及支气管扩张风险增加的担忧。通过新生儿筛查进行早期诊断有潜在希望,并且基因疗法可能为这些患者带来治愈的可能。然而,首先有必要评估当前的管理和治疗结果,以辅助有关进一步研究和临床试验的决策。本文回顾了X连锁无丙种球蛋白血症的当前管理和治疗结果,同时找出了在我们开展X连锁无丙种球蛋白血症的新型诊断方法和治疗之前可能需要解答的知识空白。