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LETTER TO THE EDITOR - CASE LETTER
Year : 2020  |  Volume : 86  |  Issue : 5  |  Page : 536-539

A case of mutilating localized cutaneous leishmaniasis caused by Leishmania donovani from Bhutan


1 Department of Dermatology, MEDICA Superspeciality Hospital, Kolkata, West Bengal, India
2 Department of Critical Care Medicine and Internal Medicine, MEDICA Superspeciality Hospital, Kolkata, West Bengal, India
3 Department of Pathology, MEDICA Superspeciality Hospital, Kolkata, West Bengal, India
4 Department of Microbiology, MEDICA Superspeciality Hospital, Kolkata, West Bengal, India

Date of Web Publication29-Jul-2020

Correspondence Address:
Dr. Amitabha Saha
Department of Critical Care Medicine and Internal Medicine, MEDICA Superspeciality Hospital, 127, Mukundapur, Eastern Metropolitan Bypass, Kolkata - 700 099, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdvl.IJDVL_801_19

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How to cite this article:
Pal A, Saha A, Chatterjee S, Saha S. A case of mutilating localized cutaneous leishmaniasis caused by Leishmania donovani from Bhutan. Indian J Dermatol Venereol Leprol 2020;86:536-9

How to cite this URL:
Pal A, Saha A, Chatterjee S, Saha S. A case of mutilating localized cutaneous leishmaniasis caused by Leishmania donovani from Bhutan. Indian J Dermatol Venereol Leprol [serial online] 2020 [cited 2020 Oct 31];86:536-9. Available from: https://www.ijdvl.com/text.asp?2020/86/5/536/291128




Sir,

A 57-year-old man, a farmer from Bhutan, presented to us with a large reddish lesion on the left side of his face of around 6 months duration. He noticed a reddish nodular growth on the left lower eyelid 10 years back, which persisted and in the past 6 months grew to involve the left side of the face. He could not recall any prior trauma or insect bite on the site. He had never traveled outside his country. A topical steroid cream was used briefly, without improvement. He had a history of intermittent fever and weight loss one year prior, for which he was given anti-tuberculosis treatment for suspected underlying tuberculosis.

On examination, there was a large erythematous, indurated and crusted plaque (10 × 9 cm) on the left side of the face from the left eyebrow to the upper lip with complete ankyloblepheron and loss of architecture of the left eyelids [Figure 1]. Atrophy, scarring and telangiectasia were seen on the nose. There was no regional lymphadenopathy. The rest of the cutaneous and systemic examinations were normal. Our differential diagnoses included lupus vulgaris, atypical mycobacterial infection, sporotrichosis, histoplasmosis, cutaneous leishmaniasis and sarcoidosis.
Figure 1: Large erythematous plaque with crusting on the face and destruction of the left eyelids

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Investigations revealed a hemoglobin of 8.9 g/dL. Enzyme-linked immunosorbent assay for human immunodeficiency virus and Mantoux test were negative. A Giemsa-stained skin smear showed numerous oval amastigotes within histiocytes [Figure 2]a. A biopsy revealed numerous amastigotes with characteristic bar-shaped paranuclear kinetoplasts and granulomatous inflammation, consistent with cutaneous leishmaniasis [Figure 2]b. The polymerase chain reaction of the tissue confirmed Leishmania [Figure 3]a and the polymerase chain reaction-reverse fragment length polymorphism analysis corresponded to Leishmania donovani [Figure 3]b.


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The rK-39 immunochromatic dipstick test from the blood sample was positive. Bone marrow aspirate and peripheral blood polymerase chain reaction were negative for Leishmania. Imaging of the face, chest and abdomen showed no significant abnormalities. We diagnosed him with localized cutaneous leishmaniasis due to L. donovani. He was given a course of intravenous liposomal amphotericin B (3mg/kg/day) for 15 days. At the end of treatment, he had marked reduction in swelling of the lesion with signs of healing [Figure 4]. He is scheduled for follow-up.
Figure 4: Marked reduction in swelling, with signs of healing and surrounding post-inflammatory hyperpigmentation immediately post-treatment

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Leishmaniasis is a zoonotic protozoal disease transmitted by sandfly vectors and is endemic in 88 countries in the tropical and subtropical zones.[1] Its presentation ranges from cutaneous leishmaniasis of varied morphology to systemic multi-organ involvement in visceral leishmaniasis.

Localized cutaneous leishmaniasis is caused commonly by L. tropica and L. major in the Old World - the Mediterranean basin, Northern Africa, Kenya, the Middle East and in Rajasthan, Punjab and Himachal Pradesh in India.[1],[2],[3]L. donavani-infantum complex primarily causes visceral leishmaniasis in Asia and East Africa but has been recently reported to cause localized cutaneous leishmaniasis in Sri Lanka, the Middle East, Kenya and Himachal Pradesh and Kerala in India.[1],[3],[4],[5],[6] Localized cutaneous leishmaniasis has not been reported from Bhutan to date, making our case unique.

Polymerase chain reaction confirmed the presence of Leishmania DNA in the biopsy and reverse fragment length polymorphism analysis confirmed the causative species, in this case, L. donovani. The rK-39 immunochromatic dipstick test qualitatively detects circulating antibodies to a recombinant Leishmania antigen rK-39.[7] The antibody response to rK-39 is largely restricted to L. donovani-infantum complex that causes visceral leishmaniasis and there is virtually no response in localized cutaneous leishmaniasis due to L. tropica infection.[7] However, positive results in localized cutaneous leishmaniasis patients suggesting infection by L. donovani-infantum complex have been reported in India, strongly suggesting cutaneous infection by L. donovani-infantum complex.[2] Thus, with the parasitological, serological and polymerase chain reaction results and in the absence of evidence of past or current visceral leishmaniasis, we diagnosed him with localized cutaneous leishmaniasis due to L. donovani.

Our patient was a farmer from a rural area in the Wangdue Phodrang district in Bhutan. Nineteen cases of visceral leishmaniasis have been reported since 2006 in Bhutan, where endemic transmission of visceral leishmaniasis has been suggested to occur.[8] The causative agent for these cases was L. donovani, with genotypes similar to Indian L. donovani isolates.[8] The presence of Phlebotomus and Sergentomyia sand flies, known vectors for visceral leishmaniasis and localized cutaneous leishmaniasis in India and China, has been confirmed in several regions here. The increasing prevalence of leishmania skin test positivity with age in the community suggests the presence of parasite for years and becoming indigenous to Bhutan.[8] The occurrence of localized cutaneous leishmaniasis caused by L. donovani in such a scenario could be the result of incidental human intrusion into the otherwise well-maintained zoonotic cycle. Other possible factors are the genetic susceptibility of populations and adaptations by parasitic strains.[3],[6]

Strong clinical suspicion is the key for early diagnosis of localized cutaneous leishmaniasis especially in non-endemic areas. This will enable early treatment, can minimize patient morbidity and prevent disfigurement. Epidemiological and genetic studies in emerging geographic foci will play an important role in the control of leishmaniasis.

Acknowledgement

Dr. Sudeshna Ganguly, Department of Academics, MEDICA Superspeciality Hospital, Kolkata, India.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal the identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Downing C, Tyring S. Parasitic diseases. In: Griffiths C, Barker J, Bleiker T, Chalmers R, Creamer D, editors. Rook's Textbook of Dermatology. 9th ed.. West Sussex, UK: John Wiley & Sons; 2016. p. 33.40-1.  Back to cited text no. 1
    
2.
Sharma NL, Mahajan VK, Negi AK, Verma GK. The rK39 immunochromatic dipstick testing: a study for K39 seroprevalence in dogs and human leishmaniasis patients for possible animal reservoir of cutaneous and visceral leishmaniasis in endemic focus of Satluj river valley of Himachal Pradesh (India). Indian J Dermatol Venereol Leprol 2009;75:52-5.  Back to cited text no. 2
    
3.
Sharma NL, Mahajan VK, Kanga A, Sood A, Katoch VM, Mauricio I, et al. Localized cutaneous leishmaniasis due to Leishmania donovani and Leishmania tropica: preliminary findings of the study of 161 new cases from a new endemic focus in Himachal Pradesh, India. Am J Trop Med Hyg 2005;72:819-24.  Back to cited text no. 3
    
4.
Kumar NP, Srinivasan R, Anish TS, Nandakumar G, Jambulingam P. Cutaneous leishmaniasis caused by Leishmania donovani in the tribal population of the agasthyamala biosphere reserve forest, Western Ghats, Kerala, India. J Med Microbiol 2015;64:157-63.  Back to cited text no. 4
    
5.
Knio KN, Baydoun E, Tawk R, Nuwayri-Salti N. Isoenzyme characterization of Leishmania isolates from Lebanon and Syria. Am J Trop Med Hyg 2000;63:43-7.  Back to cited text no. 5
    
6.
Karunaweera ND, Pratlong F, Siriwardane HV, Ihalamulla RL, Dedet JP. Sri Lankan cutaneous leishmaniasis is caused by Leishmania donovani zymodeme MON-37. Trans R Soc Trop Med Hyg 2003;97:380-1.  Back to cited text no. 6
    
7.
Zijlstra EE, Nur Y, Desjeux P, Khalil EA, El-Hassan AM, Groen J. Diagnosing visceral leishmaniasis with the recombinant K39 strip test: Experience from the Sudan. Trop Med Int Health 2001;6:108-13.  Back to cited text no. 7
    
8.
Yangzom T, Cruz I, Bern C, Argaw D, den Boer M, Vélez ID, et al. Endemic transmission of visceral leishmaniasis in Bhutan. Am J Trop Med Hyg 2012;87:1028-37.  Back to cited text no. 8
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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