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A case of Alemtuzumab-induced neutropenia in multiple sclerosis in association with the expansion of large granular lymphocytes.
BMC Neurol. 2018 Oct 29; 18(1):178.BN

Abstract

BACKGROUND

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%.

CASE PRESENTATION

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.

CONCLUSION

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.

Authors+Show Affiliations

1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece. avakrakou@med.uoa.gr.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.Immunology and Histocompatibility Department, Evangelismos Hospital, Athens, Greece.Immunology and Histocompatibility Department, Evangelismos Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.1st Department of Neurology, Medical School of Athens, National & Kapodistrian University, Aeginition Hospital, Athens, Greece.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

30373566

Citation

Vakrakou, A G., et al. "A Case of Alemtuzumab-induced Neutropenia in Multiple Sclerosis in Association With the Expansion of Large Granular Lymphocytes." BMC Neurology, vol. 18, no. 1, 2018, p. 178.
Vakrakou AG, Tzanetakos D, Valsami S, et al. A case of Alemtuzumab-induced neutropenia in multiple sclerosis in association with the expansion of large granular lymphocytes. BMC Neurol. 2018;18(1):178.
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. (2018). A case of Alemtuzumab-induced neutropenia in multiple sclerosis in association with the expansion of large granular lymphocytes. BMC Neurology, 18(1), 178. https://doi.org/10.1186/s12883-018-1183-4
Vakrakou AG, et al. A Case of Alemtuzumab-induced Neutropenia in Multiple Sclerosis in Association With the Expansion of Large Granular Lymphocytes. BMC Neurol. 2018 Oct 29;18(1):178. PubMed PMID: 30373566.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - A case of Alemtuzumab-induced neutropenia in multiple sclerosis in association with the expansion of large granular lymphocytes. AU - Vakrakou,A G, AU - Tzanetakos,D, AU - Valsami,S, AU - Grigoriou,E, AU - Psarra,K, AU - Tzartos,J, AU - Anagnostouli,M, AU - Andreadou,E, AU - Evangelopoulos,M E, AU - Koutsis,G, AU - Chrysovitsanou,C, AU - Gialafos,E, AU - Dimitrakopoulos,A, AU - Stefanis,L, AU - Kilidireas,C, Y1 - 2018/10/29/ PY - 2018/08/07/received PY - 2018/10/18/accepted PY - 2018/10/31/entrez PY - 2018/10/31/pubmed PY - 2018/12/13/medline KW - Alemtuzumab KW - Large granular cells KW - Multiple sclerosis KW - Neutropenia SP - 178 EP - 178 JF - BMC neurology JO - BMC Neurol VL - 18 IS - 1 N2 - BACKGROUND: 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%. CASE PRESENTATION: 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. CONCLUSION: 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. SN - 1471-2377 UR - https://www.unboundmedicine.com/medline/citation/30373566/A_case_of_Alemtuzumab_induced_neutropenia_in_multiple_sclerosis_in_association_with_the_expansion_of_large_granular_lymphocytes_ L2 - https://bmcneurol.biomedcentral.com/articles/10.1186/s12883-018-1183-4 DB - PRIME DP - Unbound Medicine ER -