Department of Medicine, Section of Hematology, University of Verona, Verona, Italy.
Biostatistical Service ASST of Mantova, Mantua, Italy.
Leukemia. 2022 Feb;36(2):516-524. doi: 10.1038/s41375-021-01406-y. Epub 2021 Sep 20.
In the current classification of the World Health Organization (WHO), bone marrow mastocytosis (BMM) is a provisional variant of indolent systemic mastocytosis (ISM) defined by bone marrow involvement and absence of skin lesions. However, no additional diagnostic criteria for BMM have been proposed. Within the registry dataset of the European Competence Network on Mastocytosis, we compared characteristics and outcomes of 390 patients with BMM and 1175 patients with typical ISM. BMM patients were significantly older, predominantly male, had lower tryptase and lower burden of neoplastic mast cells, and displayed a higher frequency of allergic reactions, mainly triggered by Hymenoptera, than patients with typical ISM. The estimated 10-year progression-free survival of BMM and typical ISM was 95.9% and 92.6%, respectively. In BMM patients defined by WHO-based criteria, the presence of one B-Finding and tryptase level ≥125 ng/mL were identified as risk factors for progression in multivariate analyses. BMM patients without any of these risk factors were found to have better progression-free survival (p < 0.05) and better overall survival (p < 0.05) than other ISM patients. These data support the proposal to define BMM as a separate SM variant characterized by SM criteria, absence of skin lesions, absence of B-Findings, and tryptase levels <125 ng/mL.
在世界卫生组织(WHO)目前的分类中,骨髓肥大细胞增多症(BMM)是一种惰性系统性肥大细胞增多症(ISM)的暂定变体,其定义为骨髓受累且无皮肤病变。然而,尚未提出 BMM 的其他额外诊断标准。在欧洲肥大细胞病网络注册数据集内,我们比较了 390 例 BMM 患者和 1175 例典型 ISM 患者的特征和结局。BMM 患者年龄明显较大,主要为男性,血清类胰蛋白酶水平和肿瘤性肥大细胞负担较低,且过敏反应的发生率较高,主要由膜翅目昆虫触发,高于典型 ISM 患者。BMM 和典型 ISM 的估计 10 年无进展生存率分别为 95.9%和 92.6%。在基于 WHO 标准定义的 BMM 患者中,存在一个 B 发现和血清类胰蛋白酶水平≥125ng/mL 被确定为多变量分析中进展的危险因素。没有这些危险因素的 BMM 患者被发现无进展生存率(p<0.05)和总生存率(p<0.05)更好,优于其他 ISM 患者。这些数据支持将 BMM 定义为一种独立的 SM 变体的建议,其特征为 SM 标准、无皮肤病变、无 B 发现和血清类胰蛋白酶水平<125ng/mL。