Centre de Référence MCGRE, Service d'Hémato-Immunologie, Hôpital Universitaire Robert Debré, APHP, Paris, France.
Biologie Intégrée du Globule Rouge, Unité Mixte de Recherche S1134, INSERM, Université Paris Cité, Paris, France.
Am J Hematol. 2024 Sep;99(9):1670-1679. doi: 10.1002/ajh.27374. Epub 2024 May 22.
While sickle cell anemia (SCA) and hereditary spherocytosis (HS) share common features of increased spleen erythrophagocytosis due to increased red blood cell (RBC) turnover, SCA is specifically characterized by susceptibility to infections. In this study, histological lesions in the spleens of pediatric patients with SCA were analyzed, in close correlation with past clinical history and comparatively to HS, healthy and transfused β-thalassemia patients (TDT). An evaluation of red pulp elementary lesions (red pulp fibrosis, iron deposition, number of Gandy-Gamna, and RBC trapping) combined into a severity score was established, as well as B-cell follicles analysis. Quantification on digitalized slides of iron deposition, RBC trapping, and red pulp fibrosis was additionally performed. Spleens from 22 children with SCA, eight with HS, eight with TDT, and three healthy controls (HC) were analyzed. Median age at splenectomy was not different between SCA and HS patients, 6.05 years (range: 4.5-16.0) versus 4.75 (range: 2.2-9.5). Marked heterogeneity was found in SCA spleens in contrast to other conditions. Contrary to previous reports, B-cell follicles were generally preserved in SCA. While RBC trapping was significantly increased in both SCA and HS (compared to TDT and HC), quantitative fibrosis and overall red pulp severity score were significantly increased in SCA spleens compared to other conditions. Moreover, there was an inverse correlation between quantitative fibrosis and number of B-cell follicles, linking these two compartments as well as spleen fibrosis to infectious susceptibility in SCA, potentially through impaired red pulp macrophage scavenging and B-cell subpopulations defects.
虽然镰状细胞贫血症 (SCA) 和遗传性球形红细胞增多症 (HS) 具有因红细胞 (RBC) 周转率增加而导致脾脏红细胞吞噬作用增加的共同特征,但 SCA 的特征特别在于易感染。在这项研究中,分析了患有 SCA 的儿科患者的脾脏组织学病变,与过去的临床病史密切相关,并与 HS、健康和输血β-地中海贫血患者 (TDT) 进行了比较。建立了一个严重程度评分,评估红髓基本病变(红髓纤维化、铁沉积、Gandy-Gamna 数量和 RBC 捕获),并对 B 细胞滤泡进行了分析。还对数字化幻灯片上的铁沉积、RBC 捕获和红髓纤维化进行了定量分析。分析了 22 例 SCA 患儿、8 例 HS 患儿、8 例 TDT 患儿和 3 例健康对照者 (HC) 的脾脏。SCA 和 HS 患者的脾切除术中位年龄无差异,分别为 6.05 岁(范围:4.5-16.0)和 4.75 岁(范围:2.2-9.5)。与其他情况相比,SCA 脾脏中发现明显的异质性。与先前的报告相反,B 细胞滤泡在 SCA 中通常被保留。尽管 SCA 和 HS 中的 RBC 捕获均显著增加(与 TDT 和 HC 相比),但 SCA 脾脏的定量纤维化和整体红髓严重程度评分明显高于其他情况。此外,定量纤维化与 B 细胞滤泡数量呈负相关,将这两个隔室以及脾脏纤维化与 SCA 的易感性联系起来,这可能是通过红髓巨噬细胞清除和 B 细胞亚群缺陷导致的。