DOI QR코드

DOI QR Code

Mild encephalopathy with a reversible splenial lesion in a girl with acute pyelonephritis

  • Yeom, Jung Sook (Department of Pediatrics, Gyeongsang National University School of Medicine) ;
  • Koo, Chung Mo (Department of Pediatrics, Changwon Gyeongsang National University Hospital) ;
  • Park, Ji Sook (Department of Pediatrics, Gyeongsang National University School of Medicine) ;
  • Seo, Ji-Hyun (Department of Pediatrics, Gyeongsang National University School of Medicine) ;
  • Park, Eun Sil (Department of Pediatrics, Gyeongsang National University School of Medicine) ;
  • Lim, Jae-Young (Department of Pediatrics, Gyeongsang National University School of Medicine) ;
  • Woo, Hyang-Ok (Department of Pediatrics, Gyeongsang National University School of Medicine) ;
  • Youn, Hee-Shang (Department of Pediatrics, Gyeongsang National University School of Medicine)
  • Received : 2017.09.05
  • Accepted : 2017.10.31
  • Published : 2018.02.15

Abstract

We report the case of a 12-year-old girl who had mild encephalopathy with a reversible splenial lesion (MERS) associated with acutepyelonephritis caused by Escherichia coli. The patient was admitted with a high fever, and she was diagnosed with acute pyelonephritis based on pyuria and the results of urine culture, which detected cefotaxime-sensitive E. coli. Although intravenous cefotaxime and tobramycin were administered, her fever persisted and her C-reactive protein level increased to 307 mg/L. On day 3 of admission, she demonstrated abnormal neuropsychiatric symptoms, such as delirium, ataxia, and word salad. Magnetic resonance imaging (MRI) of the brain performed on day 4 showed marked hyperintensities in the bilateral corpus callosum and deep white matter on diffusion-weighted images, with corresponding diffusion restriction on apparent diffusion coefficient mapping. No abnormalities or pathogens were detected in the cerebrospinal fluid; however, lipopolysaccharides (LPS, endotoxin) were detected in plasma (41.6 pg/mL), associated with acute neurological deterioration. Her clinical condition gradually improved, and no neurological abnormalities were observed on day 6. Follow-up brain MRI performed 2 weeks later showed near-disappearance of the previously noted hyperintense lesions. In this patient, we first proved endotoxemia in a setting of MERS. The release of LPS following antibiotic administration might be related to the development of MERS in this patient. The possibility of MERS should be considered in patients who present with acute pyelonephritis and demonstrate delirious behavior.

Keywords

References

  1. Takanashi J. Two newly proposed infectious encephalitis/encephalopathy syndromes. Brain Dev 2009;31:521-8. https://doi.org/10.1016/j.braindev.2009.02.012
  2. Jang YY, Lee KH. Transient splenial lesion of the corpus callosum in a case of benign convulsion associated with rotaviral gastroenteritis. Korean J Pediatr 2010;53:859-62. https://doi.org/10.3345/kjp.2010.53.9.859
  3. 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. https://doi.org/10.1007/s00431-013-2199-9
  4. Kometani H, Kawatani M, Ohta G, Okazaki S, Ogura K, Yasutomi M, et al. Marked elevation of interleukin-6 in mild encephalopathy with a reversible splenial lesion (MERS) associated with acute focal bacterial nephritis caused by Enterococcus faecalis. Brain Dev 2014; 36:551-3. https://doi.org/10.1016/j.braindev.2013.07.012
  5. Fujiwara Y, Tanaka F, Wakamiya T, Kobori T, Hashiguchi K, Sato M, et al. Four cases of mild encephalitis/encephalopathy with a reversible splenial lesion (MERS) accompanying acute focal bacterial nephritis (AFBN). J Jpn Pediatr Soc 2012;116:1880-5.
  6. Wang X, Rousset CI, Hagberg H, Mallard C. Lipopolysaccharideinduced inflammation and perinatal brain injury. Semin Fetal Neonatal Med 2006;11:343-53. https://doi.org/10.1016/j.siny.2006.04.002
  7. Seidel T, Kuwertz-Broking E, Kaczmarek S, Kirschstein M, Frosch M, Bulla M, et al. Acute focal bacterial nephritis in 25 children. Pediatr Nephrol 2007;22:1897-901. https://doi.org/10.1007/s00467-007-0589-9
  8. Foxman B. Epidemiology of urinary tract infections: incidence, morbidity, and economic costs. Dis Mon 2003;49:53-70. https://doi.org/10.1067/mda.2003.7
  9. 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. https://doi.org/10.1212/01.WNL.0000144274.12174.CB
  10. 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. https://doi.org/10.1016/j.braindev.2011.04.004
  11. Imamura T, Takanashi J, Yasugi J, Terada H, Nishimura A. Sisters with clinically mild encephalopathy with a reversible splenial lesion (MERS)-like features; Familial MERS? J Neurol Sci 2010;290:153-6. https://doi.org/10.1016/j.jns.2009.12.004
  12. Gofton TE, Young GB. Sepsis-associated encephalopathy. Nat Rev Neurol 2012;8:557-66. https://doi.org/10.1038/nrneurol.2012.183
  13. Giamarellos-Bourboulis EJ, Perdios J, Gargalianos P, Kosmidis J, Giamarellou H. Antimicrobial-induced endotoxaemia in patients with sepsis in the field of acute pyelonephritis. J Postgrad Med 2003;49: 118-22.
  14. Banks WA, Robinson SM. Minimal penetration of lipopolysaccharide across the murine blood-brain barrier. Brain Behav Immun 2010;24: 102-9. https://doi.org/10.1016/j.bbi.2009.09.001
  15. Stolp HB, Johansson PA, Habgood MD, Dziegielewska KM, Saunders NR, Ek CJ. Effects of neonatal systemic inflammation on blood-brain barrier permeability and behaviour in juvenile and adult rats. Cardiovasc Psychiatry Neurol 2011;2011:469046.

Cited by

  1. Increased cytokines/chemokines and hyponatremia as a possible cause of clinically mild encephalitis/encephalopathy with a reversible splenial lesion associated with acute focal bacterial nephritis vol.44, pp.1, 2018, https://doi.org/10.1016/j.braindev.2021.07.008