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Case Series and Case Reports

Pure cerebellitis due Features of bilateral cerebellar hemispherical dysfunction


such as past pointing and dysdiadokokinesia were present.
No obvious cranial nerve palsy or motor or sensory deficit
to scrub typhus: a unique was found. A fundus examination was normal. A local exam-
ination revealed an eschar on the right thigh. The rest of the
case report physical examination was unremarkable.
A routine haematological analysis was normal except for
Suman S Karanth MD MBBS* an elevated total leucocyte count (14  109 cells/L) with
neutrophilia (56%) and thrombocytopenia (93  109 cells/L).
Anurag Gupta MCh† His levels of serum glutamic oxalo-acetic transaminase
Mukhyaprana Prabhu MD* (299 IU/L), serum glutamic pyruvic transaminase (153 IU/L)
*Department of Medicine, Kasturba Medical College, and creatine kinase (1081 IU/L) were elevated. Magnetic
Manipal, India; †Department of Neurosurgery, Adarsha resonance imaging of the brain was normal. A lumbar punc-
Hospital, Udupi, India ture performed showed clear cerebrospinal fluid (CSF) with
an opening pressure of 200 mmHg, mild lymphocytic pleo-
Correspondence to: Dr Anurag Gupta, Department of
cytosis (white blood cells: 25/mm3) and marginally elevated
Neurosurgery, Adarsha Hospital, Udupi-576104, India
Email: docanurag@gmail.com
protein levels (60 mg/dL). A CSF fluid analysis for herpes
simplex virus, India ink and Gram stains was negative.
TROPICAL DOCTOR 2013; 43: 41– 42 Cultures for both bacteria and fungus were negative.
DOI: 10.1177/0049475513480775 At this juncture, the possibility of both scrub typhus and
leptospirosis prompted us to start intravenous ceftriaxone
SUMMARY We report the case of a 24-year old Indian and oral doxycycline. Serological tests for leptospirosis,
man who presented with: high fever; drowsiness; an hepatitis A, B or C and human immunodeficiency virus
eschar and gross cerebellar dysfunction with horizontal were negative. The Weil-Felix test was strongly positive
gaze nystagmus; ataxic speech; and truncal ataxia. Scrub (OXK ¼ 1:640 titres). The serum immunoglobulin M (IgM)
typhus was diagnosed by serological tests.This is the first and G (IgG) antibody titres against O. tsutsugamushi,
case of a pure cerebellar involvement as the only manifes- measured by an immunofluorescent antibody assay, were
tation of scrub typhus in the published literature. 1:5120 and 1:10240, respectively. A CSF analysis for
O. tsutsugamushi could not be performed as it was not avail-
able in our hospital. We continued doxycycline after the
Introduction microbiological confirmation of the diagnosis.
Following therapy, the fever subsided within 3 days with
Scrub typhus is a rickettsial disease caused by Orientia tsut- rapid and marked neurological improvement. By the 6th
sugamushi. The disease is typically characterized by: fever; day he was able to ambulate independently with subsequent
myalgia; sore throat; rash; eschar formation; abdominal resolution of residual cerebellar dysfunction over the next
pain; and headache.1 Serious complications include: intersti- few days. Antibiotics were continued for a total of 14 days.
tial pneumonia; gastrointestinal bleeding; acute renal failure; On discharge, he had no residual neurological deficits.
and multiple organ failures.1 Neurological involvement has
been commonly reported in rickettsial diseases in the form
of meningitis and encephalitis.1 However, an isolated cer-
ebellar involvement in scrub typhus has not yet been reported Discussion
in the published literature. We describe a rare case of young Scrub typhus is an acute febrile disease caused by
man with pure cerebellitis as a presenting feature of scrub O. tsutsugamushi which is transmitted by the bite of larval
typhus. stage (chiggers) of the thrombiculoid mite. With 1 million
new cases per year, it is emerging as an important public
health problem in Asia.2 Those engaged in agricultural or
Case history recreational activities along south-east Asia and the Pacific
A 24 year old Indian man, a farmer by occupation, was Rim are especially susceptible.3 The presentation can be mild
brought to the emergency department with drowsiness of with a self-limiting clinical course with recovery in a few
2 days duration. He had history of fever, headache along days or, rarely, severe disease with a protracted and fatal
with difficulty in walking and truncal ataxia 12 days prior. course.1 Eschar is pathognomic and is present in 40% –50%
He was admitted to a peripheral hospital for a few days of cases. It is usually located at sites such as the axilla, groin,
prior to admission to our hospital. On admission, the waist and neck where clothes bind or skin folds.2 Scrub
patient was febrile (1018F), conscious but drowsy. Kernig’s typhus must be considered alongside leptospirosis, rickettsial
and Brudzinski signs were negative. He had a horizontal pox and dengue in patients with a history of exposure from
gaze nystagmus, ataxic speech and gross truncal ataxia. an endemic geographical area.4 A Weil-Felix agglutination

Tropical Doctor January 2013, 43 41

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Case Series and Case Reports

test or indirect immunofluorescence which detects an increase prompt therapy in order to prevent further morbidity and
in serum antibody titre can be used to confirm diagnosis. mortality.
Using indirect immunofluorescence, either an antibody
titre of 1:400 or a fourfold increase in titre to 1:200 or greater Conflict of interest: None of the authors have any potential
is considered a positive result. A titre of 1:320 or greater conflicts of interest.
or a fourfold rise in titre from 1:50 determines a positive
Weil-Felix test.5 Source of funding: There was no source of funding.
Amongst the rickettsial diseases, the highest degree
of CNS involvement occurs with epidemic typhus and
Rocky Mountain spotted fever followed by scrub typhus.
Meningitis and meningoencephalitis are the most common References
presentations.1 Focal CNS involvement has rarely been
reported in the published literature. One case of acute dis- 1 Sayen J, Pond H, Forrester J. Scrub typhus in Assam and Burma:
seminated encephalomyelitis (ADEM),6 ADEM with bilat- a clinical study of 616 cases. Medicine 1946;25:155–214
eral facial nerve palsy and quadriparesis7 and cerebral 2 Watt G, Parola P. Scrub typhus and tropical rickettsioses. Curr
haemorrhage,8 have been reported. Cerebellar involvement Opin Infect Dis 2003;16:429–36
in scrub typhus, however, has rarely been reported. In a 3 Centers for Disease Control and Prevention. Health Information
series by Silpapojakul K et al., only one of 72 cases had cer- for International Travel 1999–2000. Georgia: Department of
ebellitis.9 In another series, 1 of 29 patients had cerebellar Health and Human Services, 1999
4 Saah AJ. Orientia tsutsugamushi (scrub typhus). In: Principles
signs.10 However, there have been no reports of scrub
and Practice of Infectious Disease. 5th edn. Mandell GL,
typhus presenting purely as cerebellitis, as in our patient. A Bennett JE, Dolin R, eds. Philadelphia: Churchill Livingstone,
predilection to the CNS is thought to be due to 2000:2056– 7
O. tsutsugamushi being an obligate intracellular parasite in 5 La Scola B, Raoult D. Laboratory Diagnosis of rickettsioses:
the phagocytes that invade the CNS as a part of systemic current approaches to diagnosis of old and new rickettsial dis-
infection. It causes a disseminated vasculitic process with eases. J Clin Microbiol 1997;35:2715–27
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duces an acute generalized inflammation of the vascular acute disseminated encephalomyelitis. Acta Neurol Taiwan
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Conventional treatment for scrub typhus includes tetra- 7 Kim DE, Lee SH, Park KI, Chang KH, Roh JK. Scrub typhus
cycline and chloramphenicol. In areas where response encephalomyelitis with prominent focal neurologic signs. Arch
to the above is poor, either rifampicin or doxycycline may Neurol 2000;57:1770–2
be used but rifampicin is more affective in doxycycline 8 Yang SH, Wang LS, Liang CC, Ho YH, Chang ET, Cheng CH.
resistance cases.11 Scrub Typhus complicated by intracranial hemorrhage - a case
report. Tzu Chi Med J 2005;17:111– 14
9 Silpapojakul K, Ukkachoke C, Krisanapan S, Silpapojakul K.
Conclusion Rickettsial meningitis and encephalitis. Arch Intern Med 1991;
151:1753–7
We present the first documented case of pure cerebellitis 10 Razak A, Sathyanarayanan V, Prabhu M, Sangar M,
in scrub typhus in India. With appropriate antibiotics, our Balasubramanian R. Scrub typhus in Southern India: are we
patient made a complete neurological recovery without doing enough? Trop Doct 2010;40:149–51
residual deficits. We emphasize the need for the early recog- 11 The Cochrane Collaboration. Antibiotics for Treating Scrub
nition of unusual presentations of common diseases with Typhus (Review). NJ: John Wiley & Sons, 2010

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