the brazilian journal of infectious diseases · dipankar pal∗, arindam naskar, manab kumar ghosh...

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braz j infect dis. 2013; 17(5) :610–612 The Brazilian Journal of INFECTIOUS DISEASES www.elsevier.com/locate/bjid Clinical image A case of miltefosine responsive pleomorphic post kala-azar dermal leishmaniasis Dipankar Pal , Arindam Naskar, Manab Kumar Ghosh School of Tropical Medicine, Kolkata, India article info Article history: Received 15 November 2012 Accepted 8 May 2013 Available online 5 September 2013 We identified a case of pleomorphic post kala-azar dermal leishmaniasis (PKDL) with different types of skin lesions in different stages of development. He was earlier diagnosed as a case of lepromatous leprosy on the basis of his skin picture and was treated with anti-leprotic drugs for one year in his native village, but without any improvement. We diagnosed him as a case of PKDL on clinical grounds, skin biopsy show- ing LD bodies and positive DNA-PCR from different types of skin lesions. Now he improved dramatically with miltefosine in standard dose for one month; and was continued on the medicine for the recommended duration. Case proper: A 30-year-old tribal farmer from a remote vil- lage of Bihar; a state in eastern India, known to be endemic for leishmaniasis, presented with multiple painless, non- pruritic, progressively increasing hypopigmented macular, papular (Fig. 1), plaque-type, nodular and nodules with cen- tral scar over different areas of his body including face, back, upper and lower extremities (Fig. 2A, B) for last one and half years. He first attended a nearby government hospital and was treated there as a case of lepromatous leprosy with multi-drug therapy for one year without any improvement. He presented to us with his progressing skin lesions. On enquiry, he gave a history of prolonged fever with abdominal swelling ten years Corresponding author. E-mail address: [email protected] (D. Pal). Fig. 1 – Macules with nodules before treatment. back for which he was treated with 30 intramuscular injec- tions in his local hospital (probably sodium stibogluconate). He had mild anemia, no significant lymphadenopathy. There was no sensory impairment over the skin lesions or distal limbs. 1413-8670/$ – see front matter © 2013 Elsevier Editora Ltda. All rights reserved. http://dx.doi.org/10.1016/j.bjid.2012.12.011

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Page 1: The Brazilian Journal of INFECTIOUS DISEASES · Dipankar Pal∗, Arindam Naskar, Manab Kumar Ghosh School of Tropical Medicine, Kolkata, India article info Article history: Received

b r a z j i n f e c t d i s . 2 0 1 3;17(5):610–612

The Brazilian Journal of

INFECTIOUS DISEASESwww.elsev ier .com/ locate /b j id

Clinical image

A case of miltefosine responsive pleomorphic post kala-azardermal leishmaniasis

Dipankar Pal ∗, Arindam Naskar, Manab Kumar Ghosh

School of Tropical Medicine, Kolkata, India

a r t i c l e i n f o

Article history:

Received 15 November 2012

Accepted 8 May 2013

Available online 5 September 2013

We identified a case of pleomorphic post kala-azar dermalleishmaniasis (PKDL) with different types of skin lesions indifferent stages of development. He was earlier diagnosed asa case of lepromatous leprosy on the basis of his skin pictureand was treated with anti-leprotic drugs for one year in hisnative village, but without any improvement. We diagnosedhim as a case of PKDL on clinical grounds, skin biopsy show-ing LD bodies and positive DNA-PCR from different types ofskin lesions.

Now he improved dramatically with miltefosine instandard dose for one month; and was continued on themedicine for the recommended duration.

Case proper: A 30-year-old tribal farmer from a remote vil-lage of Bihar; a state in eastern India, known to be endemicfor leishmaniasis, presented with multiple painless, non-pruritic, progressively increasing hypopigmented macular,papular (Fig. 1), plaque-type, nodular and nodules with cen-tral scar over different areas of his body including face, back,upper and lower extremities (Fig. 2A, B) for last one and halfyears. He first attended a nearby government hospital and wastreated there as a case of lepromatous leprosy with multi-drugtherapy for one year without any improvement. He presentedto us with his progressing skin lesions. On enquiry, he gave ahistory of prolonged fever with abdominal swelling ten years

∗ Corresponding author.E-mail address: [email protected] (D. Pal).

Fig. 1 – Macules with nodules before treatment.

back for which he was treated with 30 intramuscular injec-tions in his local hospital (probably sodium stibogluconate). Hehad mild anemia, no significant lymphadenopathy. There wasno sensory impairment over the skin lesions or distal limbs.

1413-8670/$ – see front matter © 2013 Elsevier Editora Ltda. All rights reserved.http://dx.doi.org/10.1016/j.bjid.2012.12.011

Page 2: The Brazilian Journal of INFECTIOUS DISEASES · Dipankar Pal∗, Arindam Naskar, Manab Kumar Ghosh School of Tropical Medicine, Kolkata, India article info Article history: Received

b r a z j i n f e c t d i s . 2 0 1 3;17(5):610–612 611

Fig. 2 – Before treatment (A. face and B. both feet).

Fig. 3 – Skin biopsy showing LD bodies {arrow}(Giemsa Stain).

He also did not have any thickening of peripheral nerves. Sys-temic examination was unremarkable. Investigations revealedpresence of mild microcytic anemia. Biochemical tests werenormal except slight elevation of serum globulin. VDRL, Hep-atitis B, C and HIV serology were all negative. rK39 strip testwas negative. Skin snipe from nodule in face was positivefor LD bodies, but negative from macular lesions. Skin snipesmear was negative for AFB. Chest X-ray and USG abdomenwere normal. Histopathology revealed sub-epidermal clearzone, lymphoplasmacytic granuloma with the presence of LD

bodies in Giemsa Stain (Fig. 3). DNA-PCR study of skin biopsyspecimen from macular, nodular and scarred lesions (Fig. 5)showed similar degree of positivity for Leishmania donovaniDNA. As patient refused injections, we treated him with oralmiltefosine 50 mg twice a day. Photographs taken after onemonth showed significant clearing of the lesions (Fig. 4A, B).These following unusual features drew our attention a) thepresence of three different types of skin lesions in the samepatient (pleomorphic), and b) DNA PCR showed similar degreeof positivity in three different morphological types of lesions,

Fig. 4 – After one month of miltefosine (A. face and B. left foot).

Page 3: The Brazilian Journal of INFECTIOUS DISEASES · Dipankar Pal∗, Arindam Naskar, Manab Kumar Ghosh School of Tropical Medicine, Kolkata, India article info Article history: Received

612 b r a z j i n f e c t d i s . 2 0 1 3;17(5):610–612

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Fig. 5 – DNA PCR from different types of skin lesions. (The DNA template used for each sample was 200 ng; DD8 is referencestrain.)

though the skin snipe was only positive in nodular lesion fromface.

Conflict of interest

The authors declare no conflict of interest.

Acknowledgements

Dr. Mitali Chatterjee, Ms. Susmita Roy, Ms. Susmita Ghoshfrom Dept. of Pharmacology, IPGMER, Kolkata for doing theDNA-PCR. Prof. Bibhuti Saha, HOD, Dept. of Tropical Medicine,School of Tropical Medicine, Kolkata.