A case of atypical and acute presentation of visceral leishmaniasis from non-endemic region, characterised by exudative pleural effusion and hepatitis is reporte

Gupta, D

Kashyap, R

Kaul, R

Raina, S

English

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Atypical presentation of visceral leishmaniasis from non-endemic region

Sujeet Raina

Rashmi Kaul

Rajesh Kashyap

Dalip Gupta

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Case Report:
Atypical presentation of visceral leishmaniasis from non-endemic region
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
Address For Correspondence:
Dr Sujeet Raina,
Fire Officers Building, 
Stokes Place, Shimla - 171002,
Himachal Pradesh, India
E-mail: sujeetrashmishera@yahoo.co.in
Citation: 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
URL: http://www.ojhas.org/issue34/2010-2-13.htm
Open Access Archives: http://cogprints.org/view/subjects/OJHAS.html and http://openmed.nic.in/view/subjects/ojhas.html
Submitted: Jun 25, 2010; Accepted: Jul 15, 2010; Published: Jul 30, 2010
Abstract:
A case of atypical and acute presentation of visceral leish-
maniasis from non-endemic region, characterised by exudative 
pleural effusion and hepatitis is reported
Key Words: Visceral leishmaniasis, nonendemic region, 
pleural effusion, hepatitis
 
Introduction:
Visceral leishmaniasis (VL) is endemic in various parts of In-
dia, 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 exudat-
ive pleural effusion and hepatitis.
Case Report:
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  move-
ments and respiration.  History of loss of appetite without any 
documentary weight  loss  was present.  Review of  other  sys-
tems 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  re-
vealed findings of right side pleural effusion. Per abdomen ex-
amination, revealed massive splenomegaly of 12cms and hep-
atomegaly of 5cms. Rest of the examination was normal.
On investigations, hemoglobin was 8 gm% and macrocytic an-
aemia 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)  Ultra-
sound abdomen showed para-aortic lymphadenopathy besides 
hepatosplenomegaly. Computerized tomography of chest con-
firmed 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 hyper-
plasia.  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 meso-
thelial cells were seen. No microorganism was observed on 
gram stain and on Zeil-Neilsen staining. Pleural fluid was neg-
ative for malignant cells. Bone marrow was normocelluar with 
megaloblastic erythropoiesis. Granulopoiesis and megakaryo-
cytes were normal and plasmacytosis was observed. Intracel-
lular and extracellular amastigotes (Leishmania Donovan bod-
ies) 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 be-
came 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 dis-
appeared  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 leishmani-
asis is established by the response to treatment of the primary 
disease.
1
This work is licensed under a
Creative Commons Attribution-
No Derivative Works 2.5 India License
Online Journal of Health and Allied Sciences
Peer Reviewed, Open Access, Free Online Journal
Published Quarterly :  Mangalore, South India : ISSN 0972-5997
Volume 9, Issue 2; Apr-Jun 2010
Figure 1: Chest x-ray at admission [A] and at discharge [B]
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 vis-
ceral  leishmaniasis  has  been  described  in  an  immunocom-
promised patient.[2] Milder forms of liver involvement occur in 
17% of cases with VL, and are structurally and functionally 
reversible after treatment. Pathophysiologically, liver involve-
ment in VL is typically self-limited and involves a mononuc-
lear cell-dominated granulomatous inflammation mediated by 
cytokines, chemokines and reactive oxygen and nitrogen spe-
cies.[3]
What was atypical in our case?
a. The patient belonged to a nonendemic region. 
b. Exudative pleural effusion due to VL in immuno-
competent patient has not been reported. 
c. Acute presentation (3 weeks) of visceral leishmani-
asis from nonendemic region.
d. 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. 
References:
1. Raina S, Mahesh DM, Kaul R, Satinder SK, Gupta 
D , Sharma A et al. A new focus of visceral leish-
maniasis in the Himalayas,  India. J Vector Borne 
Dis 2009;46:303-6
2. Chenoweth CE, Singal  S, Pearson RD, Betts RF, 
Markovitz DM. Acquired Immunodeficiency Syn-
drome  Related Visceral  Leishmaniasis  Presenting 
in a Pleural Effusion. Chest 1993;103;648-9
3. Malatesha G, Singh N K, Gulati V. Visceral leish-
maniasis: Acute liver failure in an immunocompet-
ent Asian-Indian adult. Indian Journal of Gastroen-
terology 2007;26:245-6
2


Acquired Immunodeficiency Syndrome Related Visceral Leishmaniasis Presenting in a Pleural Effusion. Chest

Sharma A et al. A new focus of visceral leishmaniasis in the Himalayas,

Visceral leishmaniasis: Acute liver failure in an immunocompetent Asian-Indian adult.

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Atypical presentation of visceral leishmaniasis from non-endemic region

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