LETTERS

Chorea and bacterial or aseptic or en- for Borrelia burgdorferi were nega- cephalitis, have been described (4,5). tive 3 times. MRI of the brain showed Tick-borne We describe a case of chorea in the lesions in the lenticular nucleus, cau- , course of TBE in a man in Poland. date nucleus on the right side, and in- Poland A 38-year-old man was admitted ternal capsules of both sides (Figure). to the hospital in Białystok, Poland, After neurologic and psychiatric To the Editor: Tick-borne en- because of fever (40°C), , consultations, chorea in the course of cephalitis (TBE) is caused by infection vomiting, and drowsiness that had TBE was diagnosed. The patient was with vector-borne viruses classified in lasted for 1 week. The patient was a treated with , dexametha- the family Flaviviridae. The disease forest worker who reported frequent sone, pentoxifylline, nitrazepam, and is characterized by a biphasic course. tick bites; for religious reasons, he midazolam, and his signs and symp- In the first phase, fever, fatigue, mal- had not been vaccinated against TBE. toms improved. aise, headache, and arthralgia occur; Physical examination revealed palsy One month later, follow-up test- in the second phase, neurologic signs in the seventh facial nerve, ing and examination were performed. develop, and the clinical spectrum in the facial muscles on the left side, No abnormalities on physical exami- ranges from mild meningitis to severe muscle weakness in the left upper and nation or laboratory test results were encephalitis, , and polyradicu- lower limbs, and positive meningeal observed except pleocytosis in CSF litis, sometimes with a fatal outcome signs. Testing of (35 cells/µL). The patient reported (1). The clinical (CSF) revealed inflammatory features and deterioration of memory. of TBE can be explained by affinity of (cytosis, 63 cells/µL, and protein con- Six months later, results of CSF test- TBEV to distinct regions of the CNS, centration 68 mg/dL). Immunologic ing were within normal limits, and such as multinodular to patchy enceph- test results showed CSF IgM of 9.04 repeat MRI showed resolution of in- alomyelitis accentuated in the spinal IU/mL and IgG of 55.1 IU/mL against flammatory lesions. cord, brain stem, and cerebellum (2). TBE virus (cutoff 1.4 for IgM, 7.0 for In this case, chorea occurred in According to the European Centre for IgG). Computed tomography scan and the course of TBE. The neurologic Disease Prevention and Control, a no- magnetic resonance imaging (MRI) of phase of TBE was not preceded by the table rise in the incidence of TBE has the brain revealed widening of ven- expected first-phase signs and symp- been observed in Europe and Asia in tricles. The patient was treated with toms. The diagnosis was confirmed recent years, and the disease is consid- mannitol, dexamethasone, third-gen- with serologic test results, which ered a growing public health concern eration cephalosporin, and ; were strongly positive in serum and in many parts of the world (3). In Po- after signs and symptoms improved, CSF, and virus neutralization testing land, an average of 250 TBE cases are the patient was discharged home on reported each year. Mean incidence is his request. 0.75 cases/100,000 population, and in- Eleven days after discharge, the cidence is highest in the northeastern patient was readmitted to the hospital part of the country, which is consid- because of deteriorating clinical signs ered to be a TBE-endemic area (11.53 (progressive muscle weakness and ex- cases/100,000 inhabitants). cessive tremor of the left-side limbs). Chorea is defined as a state of ex- Intensive propulsion, involuntary cessive, spontaneous movements that muscle movements (limbs and face), are irregularly timed, nonrepetitive, muscle weakness of left upper and randomly distributed, and abrupt in lower limbs, and “walk dance” were character (http://emedicine.medscape. observed (online Video, wwwnc.cdc. com/article/1149854). This condi- gov/EID/article/19/9/13-1804-V1. tion is a result of absent subthalamic htm). Results of CSF analysis showed nucleus inhibition, which increases continued inflammatory features motor activity through the motor (pleocytosis, 64 cells/µL, and pro- thalamus. It is a dominant symptom tein concentration 119.4 mg/dL). Re- Figure. Fluid-attenuated inversion recovery of Huntington’s disease, which is an sults of testing for antibodies against magnetic resonance imaging of the brain inherited, progressive, neurodegen- TBE virus in CSF and serum were of a 38-year-old man with tick-borne encephalitis and chorea. The image shows erative disorder. Nonhereditary causes strongly positive (CSF IgM >14 U/ bilateral areas of hyperintensity in T2, of chorea, such as infectious chorea in mL, IgG 107 U/mL; serum IgM 7.92 affecting the nucleus caudate, internal the course of acute manifestations of U/mL, IgG 87.6 U/mL). Test results capsule, and thalami.

1544 Emerging Infectious Diseases • www.cdc.gov/eid • Vol. 19, No. 9, September 2013 LETTERS performed as described in Stiasny et 2. Gelpi E, Preusser M, Laggner U, Garzuly Streptococcus suis al. (6) showed a strongly positive ti- F, Holzmann H, Heinz FX, et al. Inflamma- tory response in human tick-borne enceph- Meningitis and ter of 1:320. We excluded hepatitis C alitis: analysis of postmortem brain tissue. virus; West Nile virus infection has J Neurovirol. 2006;12:322–7. http://dx.doi. Bacteremia in Man, not been reported in Poland. Because org/10.1080/1355028 0600848746 French Guiana of his religious beliefs, the patient 3. European Centre for Disease Prevention and Control. Epidemiological situation had not been vaccinated against TBE of tick -borne encephalitis in the Euro- To the Editor: Streptococcus suis or yellow fever; he had not traveled pean Union and European Free Trade is a major swine pathogen and is in- abroad and had not been exposed to Association countries. Stockholm: creasingly recognized as the cause of dengue virus. The differential diag- The Centre; 2012. an emerging zoonosis in humans. It is 4. Cunha BA, Kang S, Chandrankunnel JG. nosis included Lyme disease, but the West Nile virus (WNV) infection present- responsible for severe systemic infec- results of repeated serologic tests for ing as acute chorea. Travel Med Infect Dis. tions, most commonly meningitis and B. burgdorferi were negative. 2012;10:52–3. http://dx.doi.org/10.1016/ sepsis, which lead to high rates of ill- Chorea arises deep in the basal j.tmaid.2011.10.002 ness and death. Serotype 2 is consid- 5. Hagiwara K, Tominaga K, Okada Y, ganglia; high-definition MRI dem- Kido M, Yamasaki R, Shida N, et al. ered to be the strain most pathogenic onstrates caudate atrophy (7). In pa- Post-streptococcal chorea in an adult for humans and pigs (1,2). tients with TBE, MRI may reveal uni- with bilateral striatal encephalitis. J Clin The first reported infection in a lateral or bilateral thalamus lesions, Neurosci. 2011;18:708–9. http://dx.doi. human was in Denmark (3), but hu- org/10.1016/j.jocn.2010.08.024 as well as lesions in the cerebellum, 6. Stiasny K, Holzmann H, Heinz FX. man infections have been now de- brainstem, and nucleus caudalis (8). Characteristics of antibody responses in scribed worldwide, with a predomi- One case study presented a case of tick-borne encephalitis vaccination break- nance in countries in Southeast Asia simultaneous lesions in the thalamus, throughs. Vaccine. 2009;27:7021–6. http:// (1,2,4). However, only 2 cases of hu- dx.doi.org/10.1016/j.vaccine.2009.09.069 stem, and (9). Chorea de- 7. Bamford KA, Caine ED, Kido DK, Cox man infection have been described in veloped in the patient reported here C, Shoulson I. A prospective evaluation South America, in Argentina in 2005 because of inflammatory lesions in of cognitive decline in early Huntington’s (5) and 2008 (6). One infection was the region of the thalamus. Support- disease: functional and radiographic corre- related to a serotype 2 strain (5). We lates. . 1995;45:1867–73. http:// ive care is standard for patients with dx.doi.org/10.1212/WNL.45.10.1867 report a human case of S. suis infec- chorea (10), but after treatment for 8. Kaiser R. The clinical and epidemiological tion with meningitis and bacteremia in TBE, the lesions in this patient re- profile of tick-borne encephalitis in south- an immunocompetent patient living in solved, and the chorea abated. ern Germany 1994–98: a prospective French Guiana. study of 656 patients. Brain. 1999; In August 2011, a 42-year-old 1 122:2067–78. http://dx.doi.org/ 10.1093/ Joanna Zajkowska, brain/122.11.2067 Haitian man was admitted to Cayenne Anna Moniuszko,1 9. Bender A, Schulte-Altedorneburg G, Hospital (Cayenne, French Guiana) Piotr Czupryna, Walther EU, Pfister HW. Severe tick borne because of a 3-day history of fever, Wiesław Drozdowski, encephalitis with simultaneous brain stem, bithalamic, and spinal cord involvement headache, vertigo, and vomiting. He Waldemar Krupa, documented by MRI. J Neurol Neurosurg was an unemployed illegal immigrant Katarzyna Guziejko, Psychiatry. 2005;76:135–7. http://dx.doi. who had lived in French Guiana for 4 Maciej Kondrusik, org/10.1136/jnnp.2004.040469 years. He had no particular medical Sambor Grygorczuk, and 10. US Food and Drug Administration. FDA approves first drug for treatment of cho- history and no risk factors for immu- Slawomir Pancewicz rea in Huntington’s disease. 2008 Aug 15 nodeficiency. At admission, he was le- Author affiliation: Medical University, [cited 2012 Aug 10]. http://www.fda.gov/ thargic, weak, and frail. He had a stiff Białystok, Poland NewsEvents/Newsroom/PressAnnounce- neck, vestibular dysfunction, , ments/2008/ucm116936.htm DOI: http://dx.doi.org/10.3201/eid1909.130804 and nystagmus. He did not have hear- Address for correspondence: Anna Moniuszko, ing loss or skin abnormalities. Cranial References Department of Infectious Diseases and computed tomography with contrast Neuroinfections, Medical University of and gadolinium-enhanced cranial 1. Zajkowska J, Czupryna P, Pancewicz S, Bialystok, Zurawia 14, 15-540 Białystok, magnetic resonance imaging did not Adamczyk-Przychodzeń A, Kondrusik show any abnormal findings. M, Grygorczuk S, et al. Fatal outcome of Poland; email: [email protected] tick-borne encephalitis—a case series. Blood tests indicated thrombocy- 9 Neurol Neurochir Pol. 2011;45:402–6. All material published in Emerging Infectious topenia (106 × 10 platelets/L), lym- Diseases is in the public domain and may be used phopenia (0.57 × 109 lymphocytes/L), 1These authors contributed equally to this and reprinted without special permission; proper a high level of C-reactive protein article. citation, however, is required. (170 mg/L) and mild cytolysis

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