Division of Haematology/Oncology, The Hospital for Sick Children, 555 University Avenue, Toronto, Ontario, M5G1X8, Canada.
University of Toronto, Toronto, Ontario, Canada.
Eur J Pediatr. 2020 May;179(5):689-697. doi: 10.1007/s00431-020-03619-2. Epub 2020 Mar 11.
Primary immunodeficiency disorders represent a heterogeneous spectrum of diseases, predisposing to recurrent infections, allergy, and autoimmunity. While an association between primary immunodeficiency disorders and increased risk of cancer has been suggested since the 1970s, renewed attention has been given to this topic in the last decade, largely in light of the availability of large registries as well as advances in next generation sequencing. In this narrative review, we will give an insight of the primary immunodeficiencies that are commonly responsible for the greater number of cancers in the primary immunodeficiency disorders population. We will describe clinical presentations, underlying genetic lesions (if known), molecular mechanisms for carcinogenesis, as well as some management considerations. We will also comment on the future directions and challenges related to this topic.Conclusion: The awareness of the association between several primary immunodeficiencies and cancer is crucial to provide the best care for these patients.What is Known: • Patients with primary immunodeficiency have an increased risk of malignancy. The type of malignancy is highly dependent on the specific primary immunodeficiency disorder.What is New: • Survival in patients with primary immunodeficiency disorders has been improving, and conversely also their lifetime risk of malignancy. • International collaboration and multinational registries are needed to improve our knowledge and therapeutic strategies.
原发性免疫缺陷病是一组异质性疾病,易发生反复感染、过敏和自身免疫。自 20 世纪 70 年代以来,人们就已经发现原发性免疫缺陷病与癌症风险增加之间存在关联,而在过去十年中,由于大型登记处的出现以及下一代测序技术的进步,人们对这一课题的关注再次得到加强。在这篇叙述性综述中,我们将深入了解一些常见的原发性免疫缺陷病,这些疾病通常是原发性免疫缺陷病患者发生癌症的主要原因。我们将描述其临床表现、潜在的遗传缺陷(如果已知)、致癌的分子机制以及一些治疗注意事项。我们还将对这一主题的未来方向和挑战进行评论。结论:了解几种原发性免疫缺陷病与癌症之间的关联对于为这些患者提供最佳护理至关重要。已知:• 原发性免疫缺陷患者的恶性肿瘤风险增加。恶性肿瘤的类型高度依赖于特定的原发性免疫缺陷病。新发现:• 原发性免疫缺陷病患者的生存状况得到了改善,相反,他们的终生恶性肿瘤风险也有所增加。• 需要国际合作和跨国登记处来提高我们的认识和治疗策略。