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OJHAS Vol. 9, Issue 2:
(2010 Apr-Jun) |
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Atypical
presentation of visceral leishmaniasis from non-endemic region |
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Sujeet
Raina, Assistant Professor, Dept. of Medicine,
Rashmi Kaul, Registrar, Department of Pathology Rajesh Kashyap, Assistant Professor, Dept. of Medicine, Dalip Gupta, Professor, Dept. of Medicine, Indira Gandhi Medical
College, Shimla – 171001, Himachal Pradesh |
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Address For Correspondence |
Dr Sujeet Raina, Fire Officers Building, Stokes Place, Shimla - 171002, Himachal Pradesh,
India
E-mail:
sujeetrashmishera@yahoo.co.in |
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Raina S, Kaul R, Kashyap R, Gupta D. Atypical
presentation of visceral leishmaniasis from non-endemic region. Online J Health Allied Scs.
2010;9(2):13 |
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Submitted: Jun 25,
2010; Accepted: Jul 15, 2010; Published: Jul 30, 2010 |
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Abstract: |
A
case of atypical and acute presentation of visceral leishmaniasis from non-endemic region,
characterised by exudative pleural effusion and hepatitis is reported
Key Words: Visceral leishmaniasis, nonendemic region,
pleural effusion, hepatitis |
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Visceral leishmaniasis (VL) is endemic in various parts of India, mainly
Bihar, West Bengal and Orissa, and neighbouring countries such as Nepal
and Bangladesh. Recently increased number of cases have been reported
from nonendemic areas of India.[1]
Atypical presentation of VL in an nonendemic area can lead to a
diagnostic
dilemma. We report VL in a patient from nonendemic region of India,
who presented with exudative pleural effusion and hepatitis.
Twenty five year male, labourer, nonsmoker, nonalcoholic, native
of
Beas river valley area (altitude 1075 meters above the mean sea level)
of Himachal Pradesh, India was admitted with history of fever,
high grade, intermittent without chills, of three weeks duration.
History
of progressive dyspnoea and dragging sensation upper abdomen without
pain was also present from the same duration. There was history of dry
cough and pain chest right side, which increased on movements and
respiration.
History of loss of appetite without any documentary weight loss was
present. Review of other systems was normal. Treatment records of
patient
revealed that he was started on ceftriaxone for past one week by his
general practitioner without any relief. No significant past history
was present. He denied ever visiting any endemic area of visceral
leishmaniasis.
On examination patient was tachypnoeic and was having tachycardia.
Bilateral
axillary lymphadenopathy was present and patient had icterus. Chest
examination revealed findings of right side pleural effusion. Per
abdomen
examination, revealed massive splenomegaly of 12cms and hepatomegaly
of 5cms. Rest of the examination was normal.
On investigations,
hemoglobin
was 8 gm% and macrocytic anaemia was observed on peripheral smear
examination.
Total leukocyte count and platelet count were normal. Total serum
bilirubin
was 5.5 mg% and conjugated was 3.1mg%. The transaminases were raised
[SGOT-225 IU, SGPT-115 IU]. The alkaline phosphatase was 271-KAU. Total
Serum proteins were 6.6gm% and albumin was 3.6gm%. Chest X-ray was
consistent
with right side pleural effusion. (Fig-1A) Ultrasound abdomen showed
para-aortic lymphadenopathy besides hepatosplenomegaly. Computerized
tomography of chest confirmed right side pleural effusion with passive
collapse right lung. Lung parenchyma and mediastinum was normal.
Tests for enteric fever, leptospirosis, malaria, HIV, viral hepatitis
(A, B, C and E) were inconclusive. Fine needle aspiration cytology of
axillary lymph node revealed only reactive hyperplasia. On
thoracocentesis,
pleural fluid was hemorrhagic, with pleural fluid protein of 6 gm%,
and cytology showing predominantly isolates and aggregates of foamy
histiocytes, pigment laden macrophages, abundant plasma cells and
lymphocytes.
In addition, multinucleate histiocytes and mesothelial cells were
seen. No microorganism was observed on gram stain and on Zeil-Neilsen
staining.
Pleural fluid was negative for malignant cells. Bone marrow was
normocelluar
with megaloblastic erythropoiesis. Granulopoiesis and megakaryocytes
were normal and plasmacytosis was observed. Intracellular and
extracellular
amastigotes (Leishmania Donovan bodies) were present.
Patient
was started on sodium stibogluconate at a dose of 20 mg/kg/day and continued for 4 weeks. By
sixth day patient became afebrile. Bilirubin returned to normal on ninth
day and transaminases were normal on twelfth day of treatment.
Pleural effusion was followed with serial X-rays and had disappeared
at the time of discharge. (Fig-1B) At discharge spleen tip was
just palpable and liver was not palpable. The patient is under regular
follow up and is asymptomatic. Both pleural effusion and hepatitis
were due to visceral leishmaniasis is established by the response to
treatment of the primary disease.
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Figure 1: Chest x-ray at
admission
[A] and at discharge [B] |
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Figure 2: Bone marrow
aspirate showing
intracellular and extracellular
amastigotes (Leishmania Donovan bodies) |
Visceral leishmaniasis often presents
with atypical features in the immunocompromised patient. Pleural
effusion
due to visceral leishmaniasis has been described in an immunocompromised
patient.[2] Milder forms of liver involvement occur in 17%
of cases with VL, and are structurally and functionally reversible after
treatment. Pathophysiologically, liver involvement in VL
is typically self-limited and involves a mononuclear cell-dominated
granulomatous inflammation mediated by cytokines, chemokines and
reactive
oxygen and nitrogen species.[3]
What was atypical in our case?
- The patient belonged to
a nonendemic region.
- Exudative pleural effusion
due to VL in immunocompetent patient has not been reported.
- Acute presentation (3 weeks)
of visceral leishmaniasis from nonendemic region.
- Hepatitis in the form of
deranged liver function tests.
The case is presented to
highlight
the atypical presentation of VL in a nonendemic region where the index
of suspicion is low.
- Raina
S, Mahesh DM, Kaul R, Satinder SK, Gupta D , Sharma A et al. A new focus of visceral leishmaniasis
in the Himalayas, India. J Vector Borne Dis 2009;46:303-6
- Chenoweth CE, Singal
S, Pearson RD, Betts RF, Markovitz DM. Acquired Immunodeficiency
Syndrome
Related Visceral Leishmaniasis Presenting in a Pleural Effusion. Chest
1993;103;648-9
- Malatesha G, Singh
N K, Gulati V. Visceral leishmaniasis: Acute liver failure in an
immunocompetent
Asian-Indian adult. Indian Journal of Gastroenterology 2007;26:245-6
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