Department of Hematological Biology, Pitié-Salpêtrière Charles-Foix Hospital, AP-HP Sorbonne University, Paris, France.
Ludwig Boltzmann Institute for Hematology and Oncology, Medical University of Vienna, Vienna, Austria; MLL Munich Leukemia Laboratory, Munich, Germany.
J Allergy Clin Immunol. 2022 Jun;149(6):1855-1865. doi: 10.1016/j.jaci.2022.04.004. Epub 2022 Apr 14.
Mast cell neoplasms are an emerging challenge in the fields of internal medicine, allergy, immunology, dermatology, laboratory medicine, and pathology. In this review, we discuss the current standards for the diagnosis and prognostication of mast cell neoplasms with special reference to clinically relevant germline and somatic gene variants. In patients with cutaneous mastocytosis or with indolent systemic mastocytosis (SM), various KIT-activating mutations act as key molecular drivers of the disease. In adults, KIT p.D816V is by far the most prevalent driver, whereas other KIT mutants are detected in nearly 40% of children. In advanced SM, including aggressive SM, SM with an associated hematological neoplasm, and mast cell leukemia, additional somatic mutations in other genes, such as SRSF2, JAK2, RUNX1, ASXL1, or RAS, may be detected. These drivers are more frequently detected in SM with an associated hematological neoplasm, particularly in male patients. Recently, hereditary alpha-tryptasemia has been identified as a genetic trait more prevalent in SM compared with healthy controls. Moreover, hereditary alpha-tryptasemia is more frequent in patients with SM with Hymenoptera venom allergy and severe mediator-related symptoms than in patients with SM without symptoms. On the basis of this knowledge, we propose a diagnostic algorithm in which genetic markers are applied together with clinical and histopathologic criteria to establish the diagnosis and prognosis in SM.
肥大细胞瘤是内科、过敏、免疫、皮肤科、实验医学和病理学领域新兴的挑战。在这篇综述中,我们讨论了肥大细胞瘤的诊断和预后的当前标准,特别提到了与临床相关的种系和体细胞基因突变。在皮肤肥大细胞增多症或惰性系统性肥大细胞增多症(SM)患者中,各种 KIT 激活突变是疾病的关键分子驱动因素。在成年人中,KIT p.D816V 是迄今为止最常见的驱动因素,而其他 KIT 突变在近 40%的儿童中被检测到。在晚期 SM 中,包括侵袭性 SM、伴有相关血液肿瘤的 SM 和肥大细胞白血病,其他基因的额外体细胞突变,如 SRSF2、JAK2、RUNX1、ASXL1 或 RAS,可能被检测到。这些驱动因素在伴有相关血液肿瘤的 SM 中更常被检测到,特别是在男性患者中。最近,遗传性α-色氨酸血症已被确定为一种遗传特征,在 SM 中比在健康对照组中更为常见。此外,遗传性α-色氨酸血症在伴有蜂毒液过敏和严重介质相关症状的 SM 患者中比在无症状的 SM 患者中更为常见。基于这些知识,我们提出了一个诊断算法,其中遗传标志物与临床和组织病理学标准一起应用于建立 SM 的诊断和预后。