Abstract
Although there are several case reports of progressive multifocal leukoencephalopathy (PML) in multiple myeloma (MM), there are few reports of cases associated with pomalidomide. Here, we report the case of a 69-year-old female who had received 41 cycles of pomalidomide and dexamethasone treatment for relapsed/refractory IgG-κ MM presented with right-hand weakness; she was diagnosed as pomalidomide-associated PML. Fluid-attenuated inversion recovery (FLAIR) on admission showed high signals in the bilateral front-parietal lobe white matter, with multiple punctate lesions in the vicinity of the main lesions. These punctate pattern findings on FLAIR were similar to that of natalizumab-associated PML. Susceptibility weighted imaging (SWI) showed hypointense rims within the cortex at unaffected sites, in the initial stages. Subsequently, the clinical manifestations deteriorated, and the FLAIR images showed new hyperintense white matter lesions at the sites where cortical SWI hypointense rims were detected on the initial MRI examination. Our patient’s serial MRI findings suggest that cortical SWI hypointense rims appear prior to the visible demyelinating white matter lesions in patients with PML.
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Anderson S, Kiernan M, Ho PJ (2019) Lenalidomide-related progressive multifocal leukoencephalopathy: a case report and review of drug-related cases in multiple myeloma. Clin Lymphoma Myeloma Leuk. 19(4):e169–e171. https://doi.org/10.1016/j.clml.2018.12
Bagnato F, Hametner S, Yao B, van Gelderen P, Merkle H, Cantor FK, Lassmann H, Duyn JH (2011) Tracking iron in multiple sclerosis: a combined imaging and histopathological study at 7 Tesla. Brain. 134(Pt 12):3602–3615. https://doi.org/10.1093/brain/awr278
Brigo F, Pagani E, Tezzon F, Masi E, Nardone R (2017) Lenalidomide-associated progressive multifocal leukoencephalopathy. Leuk Lymphoma 58(10):2514–2515. https://doi.org/10.1080/10428194.2017.1292355
Hodel J, Outteryck O, Verclytte S, Deramecourt V, Lacour A, Pruvo JP, Vermersch P, Leclerc X (2015) Brain magnetic susceptibility changes in patients with natalizumab-associated progressive multifocal leukoencephalopathy. AJNR Am J Neuroradiol 36(12):2296–2302. https://doi.org/10.3174/ajnr.A4436
Hodel J, Darchis C, Outteryck O, Verclytte S, Deramecourt V, Lacour A, Zins M, Pruvo JP, Vermersch P, Leclerc X (2016) Punctate pattern: a promising imaging marker for the diagnosis of natalizumab-associated PML. Neurology. 86(16):1516–1523. https://doi.org/10.1212/WNL.0000000000002586
Major EO, Yousry TA, Clifford DB (2018) Pathogenesis of progressive multifocal leukoencephalopathy and risks associated with treatments for multiple sclerosis: a decade of lessons learned. Lancet Neurol 17(5):467–480. https://doi.org/10.1016/S1474-4422(18)30040-1
Miyagawa M, Maeda M, Umino M, Kagawa K, Nakamichi K, Sakuma H, Tomimoto H (2014) Low signal intensity in U-fiber identified by susceptibility-weighted imaging in two cases of progressive multifocal leukoencephalopathy. J Neurol Sci 344(1–2):198–202. https://doi.org/10.1016/j.jns.2014.06.018
Papadimitropoulos GN, Lachanis S, Zompola C, Kargiotis O, Magoufis G, Voumvourakis K, Tsivgoulis G (2017) Laminar cortical hypointensities in susceptibility-weighted imaging in a case of progressive multifocal leukoencephalopathy. J Clin Neurol 13(2):201–202. https://doi.org/10.3988/jcn.2017.13.2.201
Quach H, Ritchie D, Stewart AK, Neeson P, Harrison S, Smyth MJ, Prince HM (2010) Mechanism of action of immunomodulatory drugs (IMiDS) in multiple myeloma. Leukemia. 24(1):22–32. https://doi.org/10.1038/leu.2009.236
Ruiz-Heredia Y, Sanchez-Vega B, Barrio S, Linares M, Rapado I, Braggio E, Stewart K, Folgueira MD, Ramos A, Collado L, Ruiz J, Toldos O, Hernandez-Lain A, Martinez-Lopez J (2019) Concurrent progressive multifocal leukoencephalopathy and central nervous system infiltration by multiple myeloma: a case report. J Oncol Pharm Pract 25(4):998–1002. https://doi.org/10.1177/1078155218769367
Smolle E, Trojan A, Schuster SJ, Haybaeck J (2014) Progressive multifocal leukoencephalopathy--a case report and review of the literature. In Vivo 28(5):941–948
Yeung J, van Hal S, Ho PJ (2019) Prolonged immunosuppression in relapsed, refractory multiple myeloma leading to cerebral toxoplasmosis and progressive multifocal leukoencephalopathy. Clin Lymphoma Myeloma Leuk 19(12):e625–e628. https://doi.org/10.1016/j.clml.2019.09.617
Yousry TA, Pelletier D, Cadavid D, Gass A, Richert ND, Radue EW, Filippi M (2012) Magnetic resonance imaging pattern in natalizumab-associated progressive multifocal leukoencephalopathy. Ann Neurol 72(5):779–787. https://doi.org/10.1002/ana.23676
Funding
This work was partly supported by a Grant-in-Aid for the Research Committee of Prion Disease and Slow Virus Infection, Research on Policy Planning and Evaluation for Rare and Intractable Diseases from the Ministry of Health, Labour and Welfare of Japan (to M. S., Grant Number H29-Nanchitou (Nan)-Ippan-036) and by the Japan Society for the Promotion of Science (JSPS) KAKENHI (to K. N., Grant Number 17K09768).
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Ueno, H., Kikumto, M., Takebayashi, Y. et al. Pomalidomide-associated progressive multifocal leukoencephalopathy in multiple myeloma: cortical susceptibility-weighted imaging hypointense findings prior to clinical deterioration. J. Neurovirol. 26, 452–455 (2020). https://doi.org/10.1007/s13365-020-00845-0
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DOI: https://doi.org/10.1007/s13365-020-00845-0