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- Mainali et al. Journal of Medical Case Reports 2011, 5:204 JOURNAL OF MEDICAL http://www.jmedicalcasereports.com/content/5/1/204 CASE REPORTS CASE REPORT Open Access The natural history of West Nile virus infection presenting with West Nile virus meningoencephalitis in a man with a prolonged illness: a case report Shraddha Mainali1, Mansoor Afshani1, James B Wood3 and Michael C Levin1,2* Abstract Introduction: Estimates indicate that West Nile virus infects approximately one and a half million people in the United States of America. Up to 1% may develop West Nile virus neuroinvasive disease, in which infected patients develop any combination of meningitis, encephalitis, or acute paralysis. Case presentation: A 56-year-old African-American man presented to our hospital with headache, restlessness, fever, myalgias, decreased appetite, and progressive confusion. A cerebrospinal fluid examination showed mild leukocytosis and an elevated protein level. Testing for routine infections was negative. Brain T2-weighted magnetic resonance imaging scans showed marked enlargement of caudate nuclei and increased intensity within the basal ganglia and thalami. A West Nile virus titer was positive, and serial brain magnetic resonance imaging scans showed resolving abnormalities that paralleled his neurological examination. Conclusion: This report is unusual as it portrays the natural history and long-term consequences of West Nile virus meningoencephalitis diagnosed on the basis of serial brain images. Introduction WNV develop WNV fever, which presents as a flulike ill- ness (headache, malaise, myalgias, and lymphadenopathy) West Nile virus (WNV) is an arthropod-borne flavivirus and a non-specific maculopapular rash involving the transmitted to humans by the bite of an infected mosquito neck, trunk, arms, and legs [5]. About 1% of WNV infec- [1,2]. The flavivirus belongs to the Japanese encephalitis tions result in WNV neuroinvasive disease (WNND), virus antigenic complex, which was first isolated from a defined by evidence of WNV infection with any combina- 37-year-old woman living in the West Nile District of tion of meningitis, encephalitis, or acute flaccid paralysis Uganda in 1937 [1]. Sixty-two years later, in the summer or poliolike syndrome [5]. of 1999, the virus was first identified in the United States In 1999, the Centers for Disease Control and Preven- of America, where it appeared during an outbreak of natu- tion (CDC) reported a total of 62 WNV infections, of rally acquired meningitis and encephalitis in the New which 59 presented with WNND [4]. There were seven York City area [3]. fatalities. The number of WNND cases peaked in 2002. WNV infection typically peaks in late July through That year there were 2946 WNND cases and 284 deaths early September [4]. The disease usually presents with [4]. The latest data from the CDC show a total of 21 three clinical syndromes: asymptomatic infection, mild cases of WNND with two deaths in 2010 [6]. It has been febrile syndrome (WNV fever), and neuroinvasive dis- reported that the incidence of WNND ranges between ease. The majority of people infected are asymptomatic 1:140 and 1:256 among people infected with WNV [4]. [4,5]. Approximately 20% of people who are infected with By extrapolation, the virus has infected more than one and a half million people in the United States of America, * Correspondence: mlevin@uthsc.edu 1 Department of Neurology, University of Tennessee Health Science Center, and the long-term disability following WNND is just Memphis, TN 38163, USA beginning to be appreciated [4,7,8]. We present a case of Full list of author information is available at the end of the article © 2011 Mainali et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
- Mainali et al. Journal of Medical Case Reports 2011, 5:204 Page 2 of 4 http://www.jmedicalcasereports.com/content/5/1/204 confusion. On day 13, a brain MRI scan (Figure 1B) one of the survivors of WNND. The significance of this showed decreased edema in the basal ganglia and thala- case is that serial brain magnetic resonance imaging mus as compared to the prior MRI scans. (MRI) scans were obtained which correlated with the On day 19, he was fully oriented, and his mental status clinical course of the disease, hence supporting the use of appeared to have returned to baseline. His WNV immu- brain MRI in rendering a preliminary diagnosis and noglobulin M (IgM) titer at that time was positive (acute following the progression of WNND. and convalescent phase). A brain MRI scan (Figure 1C) Case presentation obtained on day 21 revealed resolving inflammatory changes in the basal ganglia and thalamus. Six weeks A 56-year-old African-American man with a history of later he was fully oriented to time, place, and person and hypertension and chronic hepatitis C virus infection pre- did not articulate any complaints. Another brain MRI sented to the emergency room with a three-day history scan (Figure 1D) showed resolving basal ganglia and tha- of flulike symptoms, including fever, myalgias, headache, lamus edema with persistent hyperintense changes in and decreased appetite. His vital signs were blood pres- both structures. Approximately one year later he was sure 204/104 mmHg, pulse 90 beats/minute, respiratory diagnosed with depression. Four years later formal rate 14 breaths/minute, and peak temperature 105.2°F. neuropsychological and neurological evaluations showed His physical examination was notable for restlessness evidence of difficulties with motor and mental processing and confusion. There was neck rigidity. Papilledema was speed and residual, mild right hemiparesis. not present. He could not follow commands, and mild right hemiparesis was noted. Computed tomography of Discussion the brain showed a subtle, low-density signal of the cau- date nuclei bilaterally (not shown). His serum laboratory This case exemplifies a form of WNND presenting as values were normal except for mild leukocytosis, with a WNV meningoencephalitis. The most reliable diagnostic total white blood cell count of 10,400/mm3 comprising modality for WNV infection is the detection of serum 82% neutrophils, 9% monocytes, and 8% lymphocytes. IgM antibody to WNV in his serum collected within His cerebrospinal fluid (CSF) examination showed white eight to 14 days of presentation or CSF collected within blood cell count of 29/mm3, comprising 81% lympho- eight days of the onset of illness using IgM antibody cap- ture enzyme-linked immunosorbent assay [5]. Consider- cytes, 15% neutrophils, 4% monocytes, 87 mg/dl protein, ing the relatively long interval between the sample and 63 mg/dl glucose. We performed CSF Gram stain- collection and reporting of the definitive test, alternative ing, acid-fast bacilli staining, a meningitis screen (Neis- methods of diagnosis would be helpful in prompt man- seria meningitidis, group B Streptococcus, Streptococcus agement of the disease. In our patient, we found that the pneumoniae , and Haemophilus influenzae type b), a brain MRI findings (bilateral edema and hyperintensity of cryptococcal antigen test, and a Venereal Disease the basal ganglia and thalami) were helpful in this clinical Research Laboratory test, all of which were negative. setting to establish a preliminary diagnosis of WNND. He was admitted to the intensive care unit in a stupor. Further, since clinical improvement correlated with resol- He was treated with broad-spectrum antibiotics (vancomy- ving changes on serial brain MRI scans, his prognosis cin, ceftriaxone, ampicillin, and tetracycline) and intrave- could also be assessed. Critically, the brain MRI scan did nous acyclovir. Laboratory evaluations for herpes simplex not return to normal, nor did he, indicative of the long- virus (HSV)-1 and HSV-2, tuberculosis (TB), cytomegalo- lasting effects of WNND, which include residual psycho- virus (CMV), and WNV were sent. On day two, an elec- logical and psychiatric disease [7-9]. Consistent with troencephalogram showed diffuse slowing without seizure these observations, our patient developed cognitive dys- activity. A brain MRI performed on the same day demon- function with depression and is still undergoing outpati- strated markedly enlarged caudate nuclei and increased ent psychological treatment. Of note, these MRI findings intensity of caudate, lenticular nuclei and the thalamus on are not specific to WNV encephalitis. Other viral ill- T2-weighted images (Figure 1A). T1-weighted images nesses such as Japanese encephalitis virus and St Louis showed a subtle, low-intensity signal within the lesions, encephalitis virus can show similar findings; thus brain and the lesions did not enhance following gadolinium MRI cannot be the sole diagnostic modality for detecting infusion (not shown). He continued to have fever and con- WNND [2]. For example, recent data indicate that chor- fusion with a waxing and waning mental status. Polymer- ioretinitis may be a marker of WNND [10]. However, if ase chain reaction studies of his CSF for HSV, TB, and other risk factors such as geographic location (WNV pre- CMV were negative. His blood, urine, and CSF cultures dominant locations), history of exposure to mosquitoes, were negative. By day seven, his mental status had and the time of the year are considered, brain MRI can improved to the point that he was oriented to himself and be useful for establishing an early diagnosis and treat- could state his age. From hospital days eight to 16, he ment plan while the definitive test is pending. showed slow improvement with phases of intermittent
- Mainali et al. Journal of Medical Case Reports 2011, 5:204 Page 3 of 4 http://www.jmedicalcasereports.com/content/5/1/204 Figure 1 Serial brain magnetic resonance imaging (MRI) scans obtained during the course of the patient’s illness. (A) Day 2: T2- weighted MRI scan demonstrates markedly enlarged caudate nuclei (arrows) and increased intensity in the thalami (arrowheads). (B) Eleven days after his initial presentation the abnormalities were resolving (arrows and arrowheads). Of note, the lenticular nucleus was involved (asterisk). (C) Three weeks and (D) two months after his initial presentation the abnormalities continued to improve, but had not resolved completely (arrows, arrowheads). Incidental hypertensive hemorrhage was present (black arrowhead in Figure 1C). imaging; TB: tuberculosis; WNND: West Nile virus neuroinvasive disease; Conclusion WNV: West Nile virus. Characteristic patterns of serial brain MRI scans in patients with WNND can provide an early clinical clue Acknowledgements This material is the result of work supported with resources and use of the as to the diagnosis and prognosis while awaiting defini- facilities at the Veterans Administration Medical Center, Memphis, TN. tive laboratory testing. Author details 1 Department of Neurology, University of Tennessee Health Science Center, Consent Memphis, TN 38163, USA. 2Neurology Service, Veterans Administration Written informed consent was obtained from the patient Medical Center, Memphis, TN, USA. 3Radiology Services, Veterans for publication of this case report and any accompanying Administration Medical Center, Memphis, TN, USA. images. A copy of the written consent is available for Authors’ contributions review by the Editor-in-Chief of this journal. SM and MA analyzed and interpreted the patient data regarding the clinical presentation and were major contributors in writing the manuscript. JW performed and interpreted the MRI data. MCL reviewed all of the data and Abbreviations made major contributions to the writing and editing of the manuscript. All CDC: Centers for Disease Control and Prevention; CMV: cytomegalovirus; CSF: authors read and approved the final manuscript. cerebrospinal fluid; HSV: herpes simplex virus; MRI: magnetic resonance
- Mainali et al. Journal of Medical Case Reports 2011, 5:204 Page 4 of 4 http://www.jmedicalcasereports.com/content/5/1/204 Competing interests The authors declare that they have no competing interests. Received: 30 August 2010 Accepted: 25 May 2011 Published: 25 May 2011 References 1. KL Tyler, West Nile virus encephalitis in America. N Engl J Med. 344, 1858–1859 (2001). doi:10.1056/NEJM200106143442409 2. T Solomon, Flavivirus encephalitis. N Engl J Med. 351, 370–378 (2004). doi:10.1056/NEJMra030476 3. D Nash, F Mostashari, A Fine, J Miller, D O’Leary, K Murray, A Huang, A Rosenberg, A Greenberg, M Sherman, S Wong, M Layton, The outbreak of West Nile virus infection in the New York City area in 1999. N Engl J Med. 344, 1807–1814 (2001). doi:10.1056/NEJM200106143442401 4. LR Petersen, EB Hayes, West Nile virus in the Americas. Med Clin North Am 92, 1307–1322 (2008). ix. doi:10.1016/j.mcna.2008.07.004 5. LE Davis, R DeBiasi, DE Goade, KY Haaland, JA Harrington, JB Harnar, SA Pergam, MK King, BK DeMasters, KL Tyler, West Nile virus neuroinvasive disease. Ann Neurol. 60, 286–300 (2006). doi:10.1002/ana.20959 6. Centers for Disease Control and Prevention, Division of Vector-Borne Infectious Diseases, West Nile virus: fight the bite! http://www.cdc.gov/ ncidod/dvbid/westnile/index.htm 7. R Voelker, Effects of West Nile virus may persist. JAMA. 299, 2135–2136 (2008). doi:10.1001/jama.299.18.2135 8. K Murray, C Walker, E Herrington, JA Lewis, J McCormick, DW Beasley, RB Tesh, S Fisher-Hoch, Persistent infection with West Nile virus years after initial infection. J Infect Dis. 201, 2–4 (2010). doi:10.1086/648731 9. KO Murray, M Resnick, V Miller, Depression after infection with West Nile virus. Emerg Infect Dis. 13, 479–481 (2007). doi:10.3201/eid1303.060602 10. F Abroug, L Ouanes-Besbes, M Letaief, F Ben Romdhane, M Khairallah, H Triki, N Bouzouiaia, A cluster study of predictors of severe West Nile virus infection. Mayo Clin Proc. 81, 12–16 (2006). doi:10.4065/81.1.12 doi:10.1186/1752-1947-5-204 Cite this article as: Mainali et al.: The natural history of West Nile virus infection presenting with West Nile virus meningoencephalitis in a man with a prolonged illness: a case report. Journal of Medical Case Reports 2011 5:204. Submit your next manuscript to BioMed Central and take full advantage of: • Convenient online submission • Thorough peer review • No space constraints or color figure charges • Immediate publication on acceptance • Inclusion in PubMed, CAS, Scopus and Google Scholar • Research which is freely available for redistribution Submit your manuscript at www.biomedcentral.com/submit
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