Persistent neurophysiological abnormalities in Posterior Reversible Encephalopathy Syndrome: a three-month EEG and VEP follow-up in 5 children with acute lymphoblastic leukemia
Abstract
Introduction
Posterior Reversible Encephalopathy Syndrome (PRES) is a well described complication of cancer chemotherapy.
Case series
We have recently observed PRES in 5 children affected with acute lymphoblastic leukaemia. All children presented with altered mental status; visual disturbances, headache and seizures were other common clinical features. Magnetic resonance imaging showed cortical and subcortical hyperintensities on fluid-attenuated inversion recovery and diffusion weighted sequences, mainly located in the occipital and parietal regions, strongly consistent with PRES. All patients completely recovered from their neurologic deficits in about six days. Electroencephalograms (EEG) were performed at onset and three months after syndrome recovery in all patients. Visual evoked potentials (VEPs) were recorded only three months after syndrome onset. Primary objective of this case series was to determine the potential role of neurophysiological investigations in a three-month follow-up of young patients with PRES.
In our small cohort, acute lymphoblastic leukemia, hypertension and immunosuppressants played a crucial role in pathogenesis of PRES. Clinical and radiological features allowed excluding methotrexate-induced encephalopathy. Despite a complete clinical recovery, we could detect residual neurophysiological abnormalities in four patients. Both EEG and VEP abnormalities were observed in 1 patient; 1 patient had only altered EEG and 2 patients exhibited only altered VEPs. VEP anomalies usually consisted of bilateral increased latencies and durations of main waveforms.
Conclusions
EEG and VEPs proved to be effective to define residual neurological dysfunction in patients with PRES. In view of high incidence of visual disturbances and occipital involvement, we observed that VEPs might show some demyelinating features and could arrange with occipital distribution of radiological findings in patients with history of PRES.
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Hinchey J, Chaves C, Appignani B, et al. A reversible posterior leukoencephalopathy syndrome. N Engl J Med. 1996;334:494-500. DOI: 10.1056/NEJM199602223340803
Kwon S, Koo J, Lee S. Clinical spectrum of reversible posterior leukoencephalopathy syndrome. Pediatr Neurol. 2001;24:361-64. DOI: https://doi.org/10.1016/S0887-8994(01)00265-X
Lim MH, Kim DW, Cho HS, et al. Isolated cerebellar reversible leukoencephalopathy syndrome in a patient with end stage renal disease. Intern Med. 2008;47(1):43-5. DOI: https://doi.org/10.2169/internalmedicine.47.0461
Lanzino G., Cloft H., Hemstreet MK., et al. Reversible posterior leukoencephalopathy following organ transplantation: description of two cases. Clin Neurol Neurosurg. 1997;99:222-26. DOI: https://doi.org/10.1016/S0303-8467(97)00028-0
Edwards MJ, Walker R, Vinnicombe S, et al. Reversible posterior leukoencephalopathy syndrome following CHOP chemotherapy for diffuse large B-cell lymphoma. Ann Oncol. 2001;12(9):1327-9.
Wijdicks EFM. Neurologic manifestations of immunosuppressive agents. In: Wijdicks EFM, ed. Neurologic Complications in Organ Transplant Recipients. Boston: Butterworth-Heinemann;1999:127-40.
Siebert E, Spors B, Bohner S, et al. Posterior reversible encephalopathy syndrome in children: Radiological and clinical findings A retrospective analysis of a German tertiary care center. Eur J Paediatr Neurol. 2013;17:169-75. DOI: 10.1016/j.ejpn.2012.08.003
Bartynski WS, Boardman JF. Distinct imaging patterns and lesion distribution in posterior reversible leukoencephalopathy syndrome. AJNR Am J Neuroradiol. 2007;28:1320-27. DOI: https://doi.org/10.3174/ajnr.A0549
Zhang HL, Yang Y, Zhou HW, et al. Diagnosis of posterior reversible encephalopathy syndrome: does DWI help? The Lancet Neurology 2010:9(11):1046-1047. DOI: 10.1016/S1474-4422(10)70262-3
Isoda T, Mitsuiki N, Ohkawa T, et al. Irreversible Leukoencephalopathy after reduced-intensity stem cell transplantation in a dyskeratosis congenital patient with TINF2 mutation. J Pediatr Hematol Oncol. 2013;35(4):e178-82. DOI: 10.1097/MPH.0b013e318279e5ca
Aradillas E, Arora R, Gasperino J. Methotrexate-induced posterior reversible encephalopathy syndrome. J Clin Pharm Ther. 2011;36(4):529-36. DOI: 10.1111/j.1365-2710.2010.01207.x
Bartynski WS. Posterior reversible encephalopathy syndrome, part 2: controversies surrounding pathophysiology of vasogenic edema. AJNR Am J Neuroradiol. 2008;29(6):1043-9. DOI: https://doi.org/10.3174/ajnr.A0929
Beausang-Linder M, Bill A. Cerebral circulation in acute arterial hypertension--protective effects of sympathetic nervous activity. Acta Physiol Scand. 1981:111(2):193-9. DOI: 10.1111/j.1748-1716.1981.tb06724.x
Bartynski WS, Zeigler Z, Spearman MP, et al. Etiology of cortical and white matter lesions in cyclosporin-A and FK-506 neurotoxicity. AJNR Am J Neuroradiol. 2001;22(10):1901-14.
Gao B, Lyu C, Lerner A, et al. Controversy of posterior reversible encephalopathy syndrome: what have we learnt in the last 20 years? J Neurol Neurosurg Psychiatry. 2018;89(1):14-20. DOI: 10.1136/jnnp-2017-316225
Covarrubias DJ, Luetmer PH, Campeau NG. Posterior reversible encephalopathy syndrome: prognostic utility of quantitative diffusion-weighted MR images. AJNR Am J Neuroradiol. 2002; 23(6):1038-48.
Burnett MM, Hess CP, Roberts JP, et al. Presentation of reversible posterior leukoencephalopathy syndrome in patients on calcineurin inhibitors. Clin Neurol Neurosurg. 2010;112(10):886-91. DOI: 10.1016/j.clineuro.2010.07.023
Aranas RM, Prabhakaran S, Lee VH. Posterior reversible encephalopathy syndrome associated with hemorrhage. Neurocrit Care. 2009;10(3):306-12. DOI: 10.1007/s12028-009-9200-5
Cordelli DM, Masetti R, Ricci E, et al. Life-threatening complications of posterior reversible encephalopathy syndrome in children. Eur J Paediatr Neurol. 2014;18(5):632-40. DOI: 10.1016/j.ejpn.2014.04.014
Moon SN, Jeon SJ, Choi SS, et al. Can clinical and MRI findings predict the prognosis of variant and classical type of posterior reversible encephalopathy syndrome (PRES)? Acta Radiol. 2013;54(10):1182-90. DOI: 10.1177/0284185113491252
Legriel S, Schraub O, Azoulay E, et al. Critically III Posterior Reversible Encephalopathy Syndrome Study Group (CYPRESS). Determinants of recovery from severe posterior reversible Encephalopathy syndrome. PLoS One 2012;7(9):e44534. DOI: 10.1371/journal.pone.0044534
Brady KM, Mytar JO, Lee JK, et al. Monitoring cerebral blood flow pressure autoregulation in pediatric patients during cardiac surgery. Stroke. 2010;41(9):1957-62. DOI: 10.1161/STROKEAHA.109.575167
Pui CH, Yang JJ, Hunger SP, et al. Childhood Acute Lymphoblastic Leukemia: Progress Through Collaboration. J Clin Oncol. 2015:20;33(27):2938-48. DOI: 10.1200/JCO.2014.59.1636
Dufourg MN, Landman-Parker J, Auclerc MF, et al. Age and high-dose methotrexate are associated to clinical acute encephalopathy in FRALLE 93 trial for acute lymphoblastic leukemia in children. Leukemia. 2007;21(2):238-47. DOI: 10.1038/sj.leu.2404495
Kastrup O, Gerwig M, Frings M, et al. Posterior reversible encephalopathy syndrome (PRES): electroencephalographic findings and seizure patterns. J Neurol. 2012;259(7):1383-9. DOI: 10.1007/s00415-011-6362-9
Baldini M, Bartolini E, Gori S, et al. Epilepsy after neuroimaging normalization in a woman with tacrolimus-related posterior reversible encephalopathy syndrome. Epilepsy Behav. 2010;17(4):558-60. DOI: 10.1016/j.yebeh.2010.01.014
Kamiya-Matsuoka C, Tummala S. Electrographic patterns in patients with posterior reversible encephalopathy syndrome and seizures. J Neurol Sci. 2017;375:294-298. DOI: 10.1016/j.jns.2017.02.017
Maccora S, Ministeri CM, Fabbri R, et al. The importance of neurophysiological assessment in posterior reversible encephalopathy syndrome: a case report of an eclamptic woman. Neurol Sci. 2013;34(12):2239-40. DOI: 10.1007/s10072-013-1471-0
DOI: http://dx.doi.org/10.18103/imr.v4i8.753
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