Marini Alessandra, Carulli Giovanni, Lari Tiziana, Buda Gabriele, Lambelet Paola, Ciancia Eugenio M, Benedetti Edoardo, Caracciolo Francesco, Ferreri Maria Immacolata, Pesaresi Ilaria, Rousseau Martina, Ottaviano Virginia, Azzarà Antonio, Petrini Mario
Laboratory of Clinical Pathology, Versilia Hospital.
J Clin Exp Hematop. 2014;54(2):129-36. doi: 10.3960/jslrt.54.129.
Central nervous system (CNS) involvement in multiple myeloma (MM) is uncommon. Among its possible presentations, leptomeningeal involvement of MM, also termed central nervous system myelomatosis (CNS-MM) is rare and is characterized by the presence of neoplastic plasma cells in the cerebrospinal fluid (CSF). So far, 187 cases of CNS-MM have been reported : the great majority of them were diagnosed by cytological assays and flow cytometry was used in only eight cases. We describe a case of CNS-MM in a 62-year-old woman, previously treated with chemotherapy (VTD) and autologous peripheral blood hematopoietic stem cell transplantation for stage IIIB IgG-λ MM. After achieving a very good partial response, the patient showed progression of disease, with an extramedullary localization. During administration of second-line therapy, the patient showed severe neurological symptoms. MRI resulted negative. Diagnosis of CNS-MM was made by multiparameter flow cytometry, which showed the presence of CD56(+) plasma cells in a CSF sample, in the absence of plasma cell leukemia. In this paper we also present a review of the eight previous cases of CNS-MM diagnosed by flow cytometry. We found that the application of flow cytometry in cases of MM with neurological symptoms allows a rapid diagnosis of CNS-MM and provides useful information about plasma cell phenotype (including CD56 expression). Some cases of CNS-MM are characterized by normal MRI. In addition, some evidences deriving from the review of literature suggest that CSF monitoring by flow cytometry in such cases might be used to evaluate the efficacy of drugs capable of crossing the blood-brain barrier.
中枢神经系统(CNS)受累于多发性骨髓瘤(MM)并不常见。在其可能的表现中,MM的软脑膜受累,也称为中枢神经系统骨髓瘤病(CNS-MM)很罕见,其特征是脑脊液(CSF)中存在肿瘤性浆细胞。到目前为止,已报道187例CNS-MM病例:其中绝大多数通过细胞学检测诊断,仅8例使用了流式细胞术。我们描述了一例62岁女性的CNS-MM病例,该患者先前因IIIB期IgG-λ MM接受过化疗(VTD)和自体外周血造血干细胞移植。在达到非常好的部分缓解后,患者出现疾病进展,伴有髓外定位。在二线治疗期间,患者出现严重神经症状。MRI结果为阴性。通过多参数流式细胞术诊断为CNS-MM,该技术显示在一份CSF样本中存在CD5(+)浆细胞,且不存在浆细胞白血病。在本文中,我们还对之前通过流式细胞术诊断的8例CNS-MM病例进行了综述。我们发现,在有神经症状的MM病例中应用流式细胞术可快速诊断CNS-MM,并提供有关浆细胞表型(包括CD56表达)的有用信息。一些CNS-MM病例的MRI表现正常。此外,文献综述的一些证据表明,在此类病例中通过流式细胞术监测CSF可用于评估能够穿越血脑屏障的药物的疗效。