Navetta-Modrov Brianne, Yao Qingping
Division of Rheumatology, Allergy and Immunology, Stony Brook University Renaissance School of Medicine, Stony Brook, NY, USA.
Rheumatol Immunol Res. 2021 Dec 31;2(4):227-232. doi: 10.2478/rir-2021-0031. eCollection 2021 Dec.
Macroglobulinemia is associated with Schnitzler syndrome (SchS) and Waldenstrom macroglobulinemia (WM). The aim of this article was to review the above-mentioned two diseases from clinical aspects and their potential genetic links. We performed a PubMed search using the following keywords: "SchS," "WM," "autoinflammatory disease," "periodic fever syndrome," and "nucleotide-binding oligomerization domain containing protein 2 (NOD2)." A case is exemplified. Both SchS and WM share some clinical phenotypes, and SchS can evolve into WM. Though no genetic link to SchS has been established, myeloid differentiation primary response gene 88 (MyD88) mutations are detected in one-third of SchS patients and 86% WM patients. Genetic analysis of periodic fever syndrome genes has detected NOD2 mutations in 18% SchS patients and rarely NLRP3 mutations. The literature data suggest that both MyD88 and NOD2 mutations may contribute to SchS. Both MyD88 and NOD2 are known to play important roles in innate immune response, and they may be cooperative in certain autoinflammatory diseases. Molecular analysis of NOD2 mutations may be incorporated into genetic testing for patients with suspected SchS or SchS/WM.
巨球蛋白血症与施尼茨勒综合征(SchS)和华氏巨球蛋白血症(WM)相关。本文旨在从临床方面及潜在的遗传联系对上述两种疾病进行综述。我们使用以下关键词在PubMed上进行了检索:“SchS”、“WM”、“自身炎症性疾病”、“周期性发热综合征”和“含核苷酸结合寡聚化结构域蛋白2(NOD2)”。举了一个病例。SchS和WM有一些共同的临床表型,且SchS可演变为WM。虽然尚未确定与SchS的遗传联系,但在三分之一的SchS患者和86%的WM患者中检测到髓样分化初级反应基因88(MyD88)突变。对周期性发热综合征基因的遗传分析在18%的SchS患者中检测到NOD2突变,很少检测到NLRP3突变。文献数据表明,MyD88和NOD2突变可能都与SchS有关。已知MyD88和NOD2在先天免疫反应中都起重要作用,它们在某些自身炎症性疾病中可能协同作用。对NOD2突变的分子分析可纳入疑似SchS或SchS/WM患者的基因检测中。