Radiologic and clinical features of infection related cytotoxic lesions of corpus callosum splenium in adults
Keywords:Corpus callosum, Infection, MRI
Background/Aim: The cytotoxic lesion of corpus callosum splenium (CLCCS) is a clinical-radiologic syndrome that typically manifests in children. It is characterized by restricted diffusion in the splenium of the corpus callosum on magnetic resonance imaging, and accompanying symptoms of encephalopathy. There are a few case reports regarding the adult population in the literature, and only a couple of these are related to febrile illness in adults. We aimed to evaluate the clinical and the radiologic characteristics of infections related to CLCCS in adults. Methods: For this case series study, we reviewed the MRI examinations that were performed in our hospital between 2014 and 2019 to identify cases with corpus callosum splenium lesions related with febrile diseases in adults. We excluded the cases with demyelinating diseases, trauma, arterial or venous occlusive diseases, metabolic-toxic diseases, and posterior reversible encephalopathy syndrome (PRES), patients who had alcohol or drug abuse, or known malignancies. The admission dates, symptoms within the prodromal period, physical and laboratory findings, electroencephalograms, MRI features, medications and patient outcomes were recorded. Corpus callosum involvement on MRI was classified as Type 1 if lesions were limited to splenium, and Type 2 if pericallosal white matter extension was present. Results: Seven patients (four males, three females, and ages ranging from 18 to 33 years with a mean of 26.4 years) were included in the study. All patients experienced prodromal symptoms such as fever (n=7), nausea (n=5), vomiting (n=4), diarrhea (n=4) and abdominal discomfort (n=3). Neurological symptoms included drowsiness (n=4), speech disorder (n=2), impairment of consciousness (n=4), lower extremity weakness (n=2), and seizures (n=1). Neurological examination revealed confusion (n=4), nuchal rigidity (n=2), and ataxia (n=2). In one patient, the blood culture was positive for Staphylococcus epidermidis, and the stool culture was positive for Enterococcus species. MRI findings of all patients revealed Type 1 oval (n=4) or round (n=3) shaped corpus callosum splenium lesions that appeared hyperintense on T2 and FLAIR images with diffusion restriction. None of our patients had band-like Type 1 or Type 2 lesions. Clinical relief was observed in 2 days in six patients, however, rapid clinical deterioration resulting in death occurred in one patient. Conclusion: The leading symptoms in adults are fever and gastrointestinal disturbances including, nausea, vomiting, and diarrhea, while neurological examinations mostly reveal confusion, nuchal rigidity, and ataxia. In adult patients, restricted diffusion on MRI is usually limited to splenium, and pericallosal white matter is usually not involved. Mostly encountered in autumn and winter, encephalitis/encephalopathy with diffusion restriction in the splenium of corpus callosum in an adult febrile patient usually has a good prognosis, although it may lead to severe outcomes, and even result with death. Clinicians should be aware that, if even Type 1, isolated corpus callosum diffusion restriction on MRI may has catastrophic results in a febrile patient. Further studies may be useful to delineate the mechanism and its relationship with higher bilirubin levels in patients with CLCCS.
Yıldız AE, Maraş Genç H, Gürkaş E, Akmaz Ünlü H, Öncel İH, Güven A. Mild encephalitis/encephalopathy with a reversible splenial lesion in children. Diagn Interv Radiol. 2018; 24(2):108-12. doi: 10.5152/dir.2018.17319
Zhang S, Ma Y, Feng J. Clinicoradiological spectrum of reversible splenial lesion syndrome (RESLES) in adults: a retrospective study of a rare entity. Medicine (Baltimore). 2015; 94(6):e512. doi: 10.1097/MD.0000000000000512
Kim SS, Chang KH, Kim ST, Suh DC, Cheon JE, Jeong SW, et al. Focal lesion in the splenium of the corpus callosum in epileptic patients: antiepileptic drug toxicity?Am J Neuroradiol. 1999;20:125–9.
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.
Maeda M, Shiroyama T, Tsukahara H, Shimono T, Aoki S, Takeda K. Transient splenial lesion of the corpus callosum associated with antiepileptic drugs: evaluation by diffusion-weighted MR imaging. Eur Radiol 2003;13:1902–90.
Kobata R, Tsukahara H, Nakai A, Tanizawa A, Ishimori Y, Kawamura Y, et al. Transient MR Signal changes in the splenium of the corpus callosum in rotavirus encephalopathy: value of diffusion-weighted imaging. J Comput Assist Tomogr. 2002;26:825–8.
Takanashi J, Barkovich A, Yamaguchi K, Kohno Y. Influenza encephalopathy with a reversible lesion in the splenium of the corpus callosum. Am J Neuroradiol. 2004;25:798–802.
Ito S, Shima S, Ueda A, Kawamura N, Asakura K, Mutoh T. Transient splenial lesion of the corpus callosum in H1N1 influenza virus-associated encephalitis/encephalopathy. Intern Med. 2011;50(8):915-8.
Mazur-Melewska K, Jonczyk-Potoczna K, Szpura K, Biegański G, Mania A, Kemnitz P, et al. Transient lesion in the splenium of the corpus callosum due to rotavirus infection. Childs Nerv Syst. 2015;31(6):997-1000.
Lin D, Rheinboldt M. Reversible splenial lesions presenting in conjunction with febrile illness: a case series and literature review. Emerg Radiol. 2017;24(5):599-604.
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(10):1854-8. doi: 10.1212/01.wnl.0000144274.12174.cb
Takayama H, Kobayashi M, Sugishita M, Mihara B. Diffusion-weighted imaging demonstrates transient cytotoxic edema involving the corpus callosum in a patient with diffuse brain injury. Clin Neurol Neurosurg. 2000;102(3):135-9.
Cecil KM, Halsted MJ, Schapiro M, Dinopoulos A, Jones BV. Reversible MR imaging and MR spectroscopy abnormalities in association with metronidazole therapy. J Comput Assist Tomogr. 2002;26(6):948–51.
Tha KK, Terae S, Sugiura M, Nishioka T, Oka M, Kudoh K, et al. Diffusion-weighted magnetic resonance imaging in early stage of 5-flourouracil-induced leukoencephalopathy. Acta Neuro Scand. 2002;106(6):379–86.
Kim JH, Choi JY, Koh SB, Lee Y. Reversible splenial abnormality in hypoglycemic encephalopathy. Neuroradiology. 2007;49(3):217–22.
Rovira A, Alonso J, Cordoba J. MR imaging findings in hepatic encephalopathy. Am J Neuroradiol. 2008;29(9):1612–21.
Maeda M, Tsukahara H, Terada H, Nakaji S, Nakamura H, Oba H, et al. Reversible splenial lesion with restricted diffusion in a wide spectrum of diseases and conditions. J Neuroradiol. 2006;33(4):229–36.
Starkey J, Kobayashi N, Numaguchi Y, Moritani T. Cytotoxic Lesions of the Corpus Callosum That Show Restricted Diffusion: Mechanisms, Causes, and Manifestations. Radiographics. 2017;37(2):562-76.
Takanashi J. Two newly proposed infectious encephalitis/encephalopathy syndromes. Brain Dev. 2009;31(7):521-8.
Mao XJ, Zhu BC, Yu TM, Yao G. Adult severe encephalitis/encephalopathy with a reversible splenial lesion of the corpus callosum: A case report. Medicine (Baltimore). 2018;97(26):e11324.
da Rocha AJ, Reis F, Gama HP, da Silva CJ, Braga FT, Maia AC Jr, et al. Focal transient lesion in the splenium of the corpus callosum in three non-epileptic patients. Neuroradiology. 2006;48(10):731-5.
Arbelaez A, Pajon A, Castillo M. Acute Marchiafava-Bignami disease: MR findings in two patients. Am J Neuroradiol. 24(10):1955-7.
Tanaka Y, Nishida H, Hayashi R, Inuzuka T, Otsuki M. Callosal disconnection syndrome due to acute disseminated enchephalomyelitis. Rinsho Shinkeigaku. 2006;46:50-4.
Bulakbasi N, Kocaoglu M, Tayfun C, Ucoz T. Transient splenial lesion of the corpus callosum in clinically mild influenza-associated encephalitis/encephalopathy. Am J Neuroradiol. 2006;27:1983-6.
Kimura E, Okamoto S, Uchida Y, Hirahara T, Ikeda T, Hirano T, et al. A reversible lesion of the corpus callosum splenium with adult influenza-associated encephalitis/encephalopathy: a case report. J Med Case Rep. 2008;2:220.
Balcik ZE, Senadim S, Keskek A, Ozudogru A, Koksal A, Soysal A, et al. Does restricted diffusion in the splenium indicate an acute infarct? Neurol Belg. 2018 Jan 6. doi: 10.1007/s13760-017-0876-6
Gunaydin M, Ozsahin F. Transient visual loss: Transient lesion in the splenium of the corpus callosum. Turk J Emerg Med. 2017;18(3):128-30.
Altunkas A, Aktas F, Ozmen Z, Albayrak E, Almus F. MRI findings of a postpartum patient with reversible splenial lesion syndrome (RESLES). Acta Neurol Belg. 2016;116:347–9.
Eren F, Öngün G, Öztürk Ş. Clinical and Radiological Significance of Transient Brain Lesion in the Corpus Callosum Splenium: 2 Case Reports. Kafkas J Med Sci. 2018;8(2):133–6.
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