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Published in: BMC Neurology 1/2016

Open Access 01-12-2016 | Research article

Reversible splenial lesion syndrome associated with encephalitis/encephalopathy presenting with great clinical heterogeneity

Authors: Yuanzhao Zhu, Junjun Zheng, Ling Zhang, Zhenguo Zeng, Min Zhu, Xiaobin Li, Xiaoliang Lou, Hui Wan, Daojun Hong

Published in: BMC Neurology | Issue 1/2016

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Abstract

Background

Reversible splenial lesion syndrome (RESLES) is a disorder radiologically characterized by reversible lesion in the splenium of the corpus callosum (SCC). Most of patients with RESLES associated with encephalitis/encephalopathy were identified in Japanese population, but almost no Chinese patients were diagnosed as RESLES associated with encephalitis/encephalopathy.

Methods

Possible patients with reversible isolated SCC lesions were retrieved from January 2012 to July 2015 using keyword “restricted diffusion and isolated SCC lesion” in MRI report system from a large academic center. The clinical, laboratory and radiological data were summarized.

Results

A total of 15 encephalitis/encephalopathy patients (9 males and 6 females) were identified with a reversible isolated SCC lesion. Except for 13 patients with fever symptom, 8 patients also had cold symptoms before the onset of neurological symptoms. The neurological symptoms included headache, vertigo, seizure, disturbance of consciousness, and delirious behavior. Thirteen patients completely recovered within 1 month, but 2 patients who were subjected to mechanical ventilation had persistent neurological deficits. The initial MRI features showed isolated ovoid or extending SCC lesions with homogeneous hyperintense on diffusion weighted imaging (DWI) and decreased apparent diffusion coefficient (ADC) values. The follow-up MRI revealed that isolated SCC lesions with diffuse restriction disappeared at 10 to 32 days after the initial MRI study. Fractional anisotropy map revealed the decreased value of SCC lesion in a severe case with poor prognosis.

Conclusions

RESLES associated with encephalitis/encephalopathy is a reversible syndrome with an excellent prognosis in most patients, while a few patients required ventilator supporting at the early stage might have severe neurological sequelae. Reversible signal changes on DWI and ADC are identified in all patients, but fractional anisotropy values can be decreased in severe patient with neurological sequelae.
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Literature
1.
go back to reference Kashiwagi M, Tanabe T, Shimakawa S, Nakamura M, Murata S, Shabana K, et al. Clinico-radiological spectrum of reversible splenial lesions in children. Brain Dev. 2014;36:330–6.CrossRefPubMed Kashiwagi M, Tanabe T, Shimakawa S, Nakamura M, Murata S, Shabana K, et al. Clinico-radiological spectrum of reversible splenial lesions in children. Brain Dev. 2014;36:330–6.CrossRefPubMed
2.
go back to reference Tada H, Takanashi J, Barkovich AJ, Oba H, Maeda M, Tsukahara H, et al. Clinically mild encephalitis/encephalopathy with a reversible splenial lesion. Neurology. 2004;63:1854–8.CrossRefPubMed Tada H, Takanashi J, Barkovich AJ, Oba H, Maeda M, Tsukahara H, et al. Clinically mild encephalitis/encephalopathy with a reversible splenial lesion. Neurology. 2004;63:1854–8.CrossRefPubMed
3.
go back to reference Polster T, Hoppe M, Ebner A. Transient lesion in the splenium of the corpus callosum: three further cases in epileptic patients and a pathophysiological hypothesis. J Neurol Neurosurg Psychiatry. 2001;70:459–63.CrossRefPubMedPubMedCentral Polster T, Hoppe M, Ebner A. Transient lesion in the splenium of the corpus callosum: three further cases in epileptic patients and a pathophysiological hypothesis. J Neurol Neurosurg Psychiatry. 2001;70:459–63.CrossRefPubMedPubMedCentral
4.
go back to reference Mirsattari SM, Lee DH, Jones MW, Blume WT. Transient lesion in the splenium of the corpus callosum in an epileptic patient. Neurology. 2003;60:1838–41.CrossRefPubMed Mirsattari SM, Lee DH, Jones MW, Blume WT. Transient lesion in the splenium of the corpus callosum in an epileptic patient. Neurology. 2003;60:1838–41.CrossRefPubMed
5.
go back to reference Ganapathy S, Ey EH, Wolfson BJ, Khan N. Transient isolated lesion of the splenium associated with clinically mild influenza encephalitis. Pediatr Radiol. 2008;38:1243–5.CrossRefPubMed Ganapathy S, Ey EH, Wolfson BJ, Khan N. Transient isolated lesion of the splenium associated with clinically mild influenza encephalitis. Pediatr Radiol. 2008;38:1243–5.CrossRefPubMed
6.
go back to reference Fuchigami T, Goto K, Hasegawa M, Saito K, Kida T, Hashimoto K, et al. A 4-year-old girl with clinically mild encephalopathy with a reversible splenial lesion associated with rotavirus infection. J Infect Chemother. 2013;19:149–53.CrossRefPubMed Fuchigami T, Goto K, Hasegawa M, Saito K, Kida T, Hashimoto K, et al. A 4-year-old girl with clinically mild encephalopathy with a reversible splenial lesion associated with rotavirus infection. J Infect Chemother. 2013;19:149–53.CrossRefPubMed
7.
go back to reference Hara M, Mizuochi T, Kawano G, Koike T, Shibuya I, Ohya T, et al. A case of clinically mild encephalitis with a reversible splenial lesion (MERS) after mumps vaccination. Brain Dev. 2011;33:842–4.CrossRefPubMed Hara M, Mizuochi T, Kawano G, Koike T, Shibuya I, Ohya T, et al. A case of clinically mild encephalitis with a reversible splenial lesion (MERS) after mumps vaccination. Brain Dev. 2011;33:842–4.CrossRefPubMed
8.
go back to reference Okamoto T, Sato Y, Yamazaki T, Hayashi A. Clinically mild encephalitis/encephalopathy with a reversible splenial lesion associated with febrile urinary tract infection. Eur J Pediatr. 2014;173:533–6.CrossRefPubMedPubMedCentral Okamoto T, Sato Y, Yamazaki T, Hayashi A. Clinically mild encephalitis/encephalopathy with a reversible splenial lesion associated with febrile urinary tract infection. Eur J Pediatr. 2014;173:533–6.CrossRefPubMedPubMedCentral
9.
go back to reference Lype M, Ahamed S, Thomas B, Kailas L. Acute encephalopathy with a lesion of the splenium of the corpus callosum--a report of two cases. Brain Dev. 2012;34:322–4.CrossRef Lype M, Ahamed S, Thomas B, Kailas L. Acute encephalopathy with a lesion of the splenium of the corpus callosum--a report of two cases. Brain Dev. 2012;34:322–4.CrossRef
10.
go back to reference Takanashi J, Imamura A, Hayakawa F, Terada H. Differences in the time course of splenial and white matter lesions in clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS). J Neurol Sci. 2010;292:24–7.CrossRefPubMed Takanashi J, Imamura A, Hayakawa F, Terada H. Differences in the time course of splenial and white matter lesions in clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS). J Neurol Sci. 2010;292:24–7.CrossRefPubMed
11.
go back to reference Ka A, Britton P, Troedson C, Webster R, Procopis P, Ging J, et al. Mild encephalopathy with reversible splenial lesion: an important differential of encephalitis. Eur J Paediatr Neurol. 2015;19:377–82.CrossRefPubMed Ka A, Britton P, Troedson C, Webster R, Procopis P, Ging J, et al. Mild encephalopathy with reversible splenial lesion: an important differential of encephalitis. Eur J Paediatr Neurol. 2015;19:377–82.CrossRefPubMed
12.
go back to reference Degirmenci E, Degirmenci T, Cetin EN, Kıroğlu Y. Mild encephalitis/encephalopathy with a reversible splenial lesion (MERS) in a patient presenting with papilledema. Acta Neurol Belg. 2015;115:153–5.CrossRefPubMed Degirmenci E, Degirmenci T, Cetin EN, Kıroğlu Y. Mild encephalitis/encephalopathy with a reversible splenial lesion (MERS) in a patient presenting with papilledema. Acta Neurol Belg. 2015;115:153–5.CrossRefPubMed
13.
go back to reference Garcia-Monco JC, Cortina IE, Ferreira E, Martínez A, Ruiz L, Cabrera A, et al. Reversible splenial lesion syndrome (RESLES): what’s in a name? J Neuroimaging. 2011;21:e1–14.CrossRefPubMed Garcia-Monco JC, Cortina IE, Ferreira E, Martínez A, Ruiz L, Cabrera A, et al. Reversible splenial lesion syndrome (RESLES): what’s in a name? J Neuroimaging. 2011;21:e1–14.CrossRefPubMed
14.
go back to reference Hashimoto Y, Takanashi J, Kaiho K, Fujii K, Okubo T, Ota S, et al. A splenial lesion with transiently reduced diffusion in clinically mild encephalitis is not always reversible: A case report. Brain Dev. 2009;31:710–2.CrossRefPubMed Hashimoto Y, Takanashi J, Kaiho K, Fujii K, Okubo T, Ota S, et al. A splenial lesion with transiently reduced diffusion in clinically mild encephalitis is not always reversible: A case report. Brain Dev. 2009;31:710–2.CrossRefPubMed
15.
go back to reference Hoshino A, Saitoh M, Oka A, Okumura A, Kubota M, Saito Y, et al. Epidemiology of acute encephalopathy in Japan, with emphasis on the association of viruses and syndromes. Brain Dev. 2012;34:337–43.CrossRefPubMed Hoshino A, Saitoh M, Oka A, Okumura A, Kubota M, Saito Y, et al. Epidemiology of acute encephalopathy in Japan, with emphasis on the association of viruses and syndromes. Brain Dev. 2012;34:337–43.CrossRefPubMed
16.
go back to reference Venkatesan A, Tunkel AR, Bloch KC, Lauring AS, Sejvar J, Bitnun A, et al. Case definitions, diagnostic algorithms, and priorities in encephalitis: consensus statement of the international encephalitis consortium. Clin Infect Dis. 2013;57:1114–28.CrossRefPubMedPubMedCentral Venkatesan A, Tunkel AR, Bloch KC, Lauring AS, Sejvar J, Bitnun A, et al. Case definitions, diagnostic algorithms, and priorities in encephalitis: consensus statement of the international encephalitis consortium. Clin Infect Dis. 2013;57:1114–28.CrossRefPubMedPubMedCentral
17.
go back to reference Fluss J, Ferey S, Menache-Starobinski C, Delavelle J, Van Bogaert P, Vargas MI. Mild influenza-associated encephalopathy/encephalitis with a reversible splenial lesion in a Caucasian child with additional cerebellar features. Eur J Paediatr Neurol. 2010;14:97–100.CrossRefPubMed Fluss J, Ferey S, Menache-Starobinski C, Delavelle J, Van Bogaert P, Vargas MI. Mild influenza-associated encephalopathy/encephalitis with a reversible splenial lesion in a Caucasian child with additional cerebellar features. Eur J Paediatr Neurol. 2010;14:97–100.CrossRefPubMed
18.
go back to reference Sato T, Ushiroda Y, Oyama T, Nakatomi A, Motomura H, Moriuchi H. Kawasaki disease-associated MERS: pathological insights from SPECT findings. Brain Dev. 2012;34:605–8.CrossRefPubMed Sato T, Ushiroda Y, Oyama T, Nakatomi A, Motomura H, Moriuchi H. Kawasaki disease-associated MERS: pathological insights from SPECT findings. Brain Dev. 2012;34:605–8.CrossRefPubMed
19.
go back to reference Miyata R, Tanuma N, Hayashi M, Imamura T, Takanashi J, Nagata R, et al. Oxidative stress in patients with clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS). Brain Dev. 2012;34:124–7.CrossRefPubMed Miyata R, Tanuma N, Hayashi M, Imamura T, Takanashi J, Nagata R, et al. Oxidative stress in patients with clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS). Brain Dev. 2012;34:124–7.CrossRefPubMed
20.
go back to reference Osuka S, Imai H, Ishikawa E, Matsushita A, Yamamoto T, Nozue H, et al. Mild encephalitis/encephalopathy with a reversible splenial lesion: evaluation by diffusion tensor imaging. Two case reports. Neurol Med Chir (Tokyo). 2010;50:1118–22.CrossRef Osuka S, Imai H, Ishikawa E, Matsushita A, Yamamoto T, Nozue H, et al. Mild encephalitis/encephalopathy with a reversible splenial lesion: evaluation by diffusion tensor imaging. Two case reports. Neurol Med Chir (Tokyo). 2010;50:1118–22.CrossRef
21.
go back to reference Shankar B, Narayanan R, Muralitharan P, Ulaganathan B. Evaluation of mild encephalitis/encephalopathy with a reversible splenial lesion (MERS) by diffusion-weighted and diffusion tensor imaging. BMJ Case Rep. 2014;2014:bcr2014204078. Shankar B, Narayanan R, Muralitharan P, Ulaganathan B. Evaluation of mild encephalitis/encephalopathy with a reversible splenial lesion (MERS) by diffusion-weighted and diffusion tensor imaging. BMJ Case Rep. 2014;2014:bcr2014204078.
22.
go back to reference Fridinger SE, Alper G. Defining encephalopathy in acute disseminated encephalomyelitis. J Child Neurol. 2013;87:1–5. Fridinger SE, Alper G. Defining encephalopathy in acute disseminated encephalomyelitis. J Child Neurol. 2013;87:1–5.
23.
go back to reference Cho JS, Ha SW, Han YS, Park SE, Hong KM, Han JH, et al. Mild encephalopathy with reversible lesion in the splenium of the corpus callosum and bilateral frontal white matter. J Clin Neurol. 2007;3:53–6.CrossRefPubMedPubMedCentral Cho JS, Ha SW, Han YS, Park SE, Hong KM, Han JH, et al. Mild encephalopathy with reversible lesion in the splenium of the corpus callosum and bilateral frontal white matter. J Clin Neurol. 2007;3:53–6.CrossRefPubMedPubMedCentral
24.
go back to reference Dubey D, Sawhney A, Greenberg B, Lowden A, Warnack W, Khemani P, et al. The spectrum of autoimmune encephalopathies. J Neuroimmunol. 2015;287:93–7.CrossRefPubMed Dubey D, Sawhney A, Greenberg B, Lowden A, Warnack W, Khemani P, et al. The spectrum of autoimmune encephalopathies. J Neuroimmunol. 2015;287:93–7.CrossRefPubMed
25.
Metadata
Title
Reversible splenial lesion syndrome associated with encephalitis/encephalopathy presenting with great clinical heterogeneity
Authors
Yuanzhao Zhu
Junjun Zheng
Ling Zhang
Zhenguo Zeng
Min Zhu
Xiaobin Li
Xiaoliang Lou
Hui Wan
Daojun Hong
Publication date
01-12-2016
Publisher
BioMed Central
Published in
BMC Neurology / Issue 1/2016
Electronic ISSN: 1471-2377
DOI
https://doi.org/10.1186/s12883-016-0572-9

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