Vakrakou A G, Tzanetakos D, Valsami S, Grigoriou E, Psarra K, Tzartos J, Anagnostouli M, Andreadou E, Evangelopoulos M E, Koutsis G, Chrysovitsanou C, Gialafos E, Dimitrakopoulos A, Stefanis L, Kilidireas C
1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.
Department of Blood Transfusion, Medical School of Athens, National & Kapodistrian University, Aretaieion Hospital, Athens, Greece.
BMC Neurol. 2018 Oct 29;18(1):178. doi: 10.1186/s12883-018-1183-4.
Alemtuzumab has been demonstrated to reduce the risks of relapse and accumulation of sustained disability in Multiple Sclerosis (MS) patients compared to β-interferon. It acts against CD52, leading primarily to lymphopenia. Recent data have shown that mild neutropenia is observed in 16% of treated MS-patients whereas severe neutropenia occurred in 0.6%.
Herein, we present the case of a 34-year-old woman with relapsing-remitting MS, with a history of treatment with glatiramer acetate and natalizumab, who subsequently received Alemtuzumab (12 mg / 24 h × 5 days). 70-days after the last Alemtuzumab administration, the patient displayed neutropenia (500 neutrophils/μL) with virtual absence of B-cells (0.6% of total lymphocytes), low values of CD4-T-cells (6.6%) and predominance of CD8-T-cells (48%) and NK-cells (47%); while large granular lymphocytes (LGL) predominated in the blood-smear examination. Due to prolonged neutropenia (5-days) the patient was placed on low-dose corticosteroids leading to sustained remission.
This is the first case of a patient with relapsing-remitting MS with neutropenia two months post-Alemtuzumab, with simultaneous presence of LGL cells in the blood and a robust therapeutic response to prednisolone. We recommend testing with a complete blood count every 15 days in the first 3 months after the 1st Alemtuzumab administration and searching for large granular lymphocytes cell expansion on microscopic examination of the peripheral blood if neutropenia develops.
与β-干扰素相比,已证实阿仑单抗可降低多发性硬化症(MS)患者复发和持续性残疾累积的风险。它作用于CD52,主要导致淋巴细胞减少。最近的数据显示,16%接受治疗的MS患者出现轻度中性粒细胞减少,而严重中性粒细胞减少的发生率为0.6%。
在此,我们报告一例34岁复发缓解型MS女性患者,有醋酸格拉替雷和那他珠单抗治疗史,随后接受阿仑单抗(12mg/24小时×5天)治疗。最后一次阿仑单抗给药70天后,患者出现中性粒细胞减少(500个中性粒细胞/μL),几乎没有B细胞(占总淋巴细胞的0.6%),CD4-T细胞值低(6.6%),CD8-T细胞(48%)和NK细胞(47%)占优势;而在血液涂片检查中,大颗粒淋巴细胞(LGL)占主导。由于长期中性粒细胞减少(5天),患者接受低剂量皮质类固醇治疗,病情持续缓解。
这是首例复发缓解型MS患者在阿仑单抗治疗两个月后出现中性粒细胞减少,同时血液中存在LGL细胞且对泼尼松龙有强烈治疗反应的病例。我们建议在首次阿仑单抗给药后的前三个月每15天进行一次全血细胞计数检测,如果出现中性粒细胞减少,在外周血显微镜检查中寻找大颗粒淋巴细胞扩增情况。