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  Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 10  |  Issue : 2  |  Page : 165-167  

Chronic relapsing cutaneous leishmaniasis in an elderly female: A rare clinical presentation from a nonendemic area


Department of Dermatology and STDs, Government Medical College, Srinagar, J and K, India

Date of Web Publication15-Mar-2019

Correspondence Address:
Shagufta Rather
Associate Professor, Department of Dermatology and STDs, Government Medical College, Srinagar, J and K 190001
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/idoj.IDOJ_264_17

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   Abstract 


The presentation of cutaneous leishmaniasis (CL) varies from asymptomatic erythematous papules to nodulo-ulcerative forms over the exposed parts of body, generally healing slowly in 3–12 months. Besides, rare and atypical presentations of disease are being reported that pose both a diagnostic and therapeutic challenge especially in nonendemic areas. There has been an increase in the incidence of CL in our region over the past decade, and most of our patients belong to district Kupwara which is the north-most district of Kashmir valley, situated at an altitude of 5300 feet above sea level sharing borders with Pakistan-administered Kashmir. Herein, we report a case of an elderly female from a nonendemic area who had a relapse of cutaneous disease at a previously treated site and came to us with an atypical presentation of more than 2 years duration.

Keywords: Chronic cutaneous leishmaniasis, leishmaniasis recidiva cutis, lupoid leishmaniasis, nonendemic area


How to cite this article:
Rather S, Bashir S, Bhat Y J, Hassan I. Chronic relapsing cutaneous leishmaniasis in an elderly female: A rare clinical presentation from a nonendemic area. Indian Dermatol Online J 2019;10:165-7

How to cite this URL:
Rather S, Bashir S, Bhat Y J, Hassan I. Chronic relapsing cutaneous leishmaniasis in an elderly female: A rare clinical presentation from a nonendemic area. Indian Dermatol Online J [serial online] 2019 [cited 2019 Jun 24];10:165-7. Available from: http://www.idoj.in/text.asp?2019/10/2/165/254293




   Introduction Top


Cutaneous leishmaniasis (CL) is a protozoal disease caused by Leishmania, transmitted by the bite of a female Phlebotomus sand fly, affecting various age groups, and causing a spectrum of clinical disease with cutaneous, mucosal, or visceral involvement depending on the infecting Leishmania species and host immunocompetence.[1],[2] Both lupoid leishmaniasis (LL) and leishmaniasis recidiva cutis (LRC) have been used interchangeably for the chronic form of CL although LL has previously been described in the literature more frequently.[3],[4] We describe a similar type of rare, atypical chronic relapsing type of CL in an elderly female who belonged to a nonendemic area.


   Case Report Top


An 85-year-old elderly female presented to us with history of asymptomatic, enlarging nodules and plaques on the face for last 2 years. Although the progression was quite insidious initially, but over the past 8 months, lesion had increased rapidly to attain a huge size and become more indurated. Similar swellings had appeared over the adjacent area of both cheeks and upper lip. There was no history of travel outside her district before the onset of disease. Family history was not significant. There were no systemic complaints and no history of nasal obstruction, epistaxis or discharge from nose, weight loss, facial flushing, or photosensitivity.

The patient gave history of having received sodium stibogluconate (SSG) injections from the same hospital about 5 years back for a small red plaque over the left side of the cheek just below the medial canthus. The injections were given intralesionally, twice weekly for 4 weeks with moderate improvement in the lesion after which the patient did not follow-up.

Cutaneous examination revealed a large globular, erythematous, edematous, indurated, nontender nodulo-plaque involving the entire nose extending to nasolabial folds on sides, right cheek, and upper lip. Few erythematous nodules were seen on the left side just below the medial canthus adjacent to the nose. The overlying skin was erythematous with a dusky violaceous hue, some scaling but no ulcer or crust [Figure 1]a and [Figure 1]b. A large, firm, nontender lymph node of 2 × 3 cm size was present on the right side in the submandibular region. Systemic examination was unremarkable. Nasal cavity was substantially undamaged except for a hyperemic nasal mucosa.
Figure 1: (a and b) Erythematous, indurated nodules, and plaques over the face with bulbous enlargement of nose

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Routine laboratory tests, chest X-ray, Mantoux test, and polymerase chain reaction (PCR) for acid-fast bacilli and retroviral serology were unremarkable. After excluding other granulomatous disorders, the patient was subjected to skin smears which were negative for amastigote form of Leishmania parasite, that is, Leishman-Donovan (LD) bodies. Biopsy from the lesion showed a chronic noncaseating granulomatous inflammation with numerous dot-shaped microorganisms, consistent with amastigotes of Leishmania species [Figure 2]. Fine needle aspiration (FNAC) from lymph node showed numerous LD bodies along with inflammatory cells.
Figure 2: Well-formed epithelioid cell granuloma along with heavy chronic lymphomononuclear and plasmacytic infiltrate and LD bodies (H and E, ×400)

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Based on the history, an unusual course and clinical presentation, and previous treatment records, the patient was diagnosed to have lupoid form of chronic CL and was treated with daily intramuscular SSG. Swelling started regressing in size within 3 weeks. But keeping in view the chronicity and recurrence, we extended the treatment to 28 days. She tolerated the therapy well and was discharged at 4 weeks [Figure 3]a and [Figure 3]b.
Figure 3: (a and b) Resolution of lesions after treatment at 4 and 8 weeks

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   Discussion Top


CL in India has been reported from Thar deserts of Rajasthan, Punjab, and Kerala where it is mainly caused by Leishmania tropica and now recently from Himachal Pradesh due to Leishmania donovani and L. tropica.[1],[2] Besides the typical clinical presentation, several unusual and atypical clinical manifestations have been documented from endemic countries across the world.[5],[6] The wide variation in morphological presentation has been attributed to many factors such as strain of the parasite, pathogenicity, virulence, infectivity, altered cell-mediated immunity, and geographical factors of the place of dwelling.[5],[6] One such entity described in literature is chronic CL, although the opinion regarding the criteria for chronicity of the disease is varied (6 months to 2 years).[7] Furthermore, both LL and leishmaniasis recidiva have been used interchangeably for the chronic form of CL. However, in a study by ul Bari and Raz, it was emphasized that those presenting with an erythematous infiltrated or psoriasiform plaque, of less duration, that lack the hallmarks of chronic leishmaniasis like scarring, relapsing, and reactivating course and show an excellent response to treatment should be labelled as LL, whereas those with chronic relapsing or persistent, scarring, or nonresponding cases should only be categorized as LRC, leishmaniasis cicatrix, or chronic scarring CL.[7]

Chronicity is thought to be the result of failure of cellular immunity to sterilize the lesion despite the presence of exaggerated hypersensitivity leading to parasite persistence and recurrence of disease.[8] It is TH2-type response that is believed to be involved in chronicity, persistence of lesions, and delayed or unsatisfactory response to treatment in these cases.[8]

LRC is an uncommon clinical variant that occurs almost exclusively as a complication of L. tropica in the old world and less commonly Leishmania braziliensis in South America and shows a paucity of amastigotes in the smears and on histopathological examination.[7],[8] It is most likely that our case represented a case of LL as there are several consistent features such as history of preceding episode, previous incomplete treatment, relentless progression, and a typical clinical picture. This case is particularly notable because of extreme age of the patient with no history of travel to any endemic area and the lack of ulceration over several years of progressive infection. Reactivation or persistence of parasites in this case is likely to have occurred in the setting of declining immune function caused by advancing age. The chronicity of lesion, laxity of facial tissue, and rich vascularization had probably been a contributing factor in producing such a huge erythematous swelling. Presence of amastigotes on histopathological analysis and in the FNAC of a lymph node supported our diagnosis. PCR which is a sensitive and specific method and allows species differentiation was not available in our setup.[9]


   Conclusion Top


We are reporting this case to help raise index of suspicion among clinicians who may be confronted with a myriad of clinical presentations of CL. In addition, we emphasize that the clinicians in the region where disease is unknown or rare should consider it a possible encounter any time in the course of their practice.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Kumar R, Bumb RA, Ansari NA, Mehta RD, Salotra P. Cutaneous leishmaniasis caused by L. tropica in Bikaner, India: Parasite identification and characterization using molecular and immunologic tools. Am J Trop Med Hyg 2007;76:896-901.  Back to cited text no. 1
    
2.
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 focus in Himachal Pradesh, India. Am J Trop Med Hyg 2005;72:819-24.  Back to cited text no. 2
    
3.
Momeni AZ, Yotsumoto S, Mehregan DR, Mehregan AH, Mehregan DA, Aminjavaheri M, et al. Chronic lupoid leishmaniasis. Evaluation by polymerase chain reaction. Arch Dermatol 1996;132:198-202.  Back to cited text no. 3
    
4.
Masmoudi A, Boudaya S, Ayadi N, Bouassida S, Khabir A, Meziou TJ, et al. Clinical and histological study of lupoid cutaneous leishmaniasis (16 cases). Presse Med 2007; 36:1738-42.  Back to cited text no. 4
    
5.
Bari A, Rahman SB. Many faces of cutaneous leishmaniasis. Indian J Dermatol Venereol Leprol 2008;74:23-7.  Back to cited text no. 5
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6.
Bongiorno MR, Pistone G, Arico'M. Unusual clinical variants of cutaneous leishmaniasis in Sicily. Int J Dermatol 2009;48:286-9.  Back to cited text no. 6
    
7.
Bari A, Raza N. Lupoid cutaneous leishmaniasis: A report of 16 cases. Indian J Dermatol Venereol Leprol 2010;76:85.  Back to cited text no. 7
    
8.
Rahman SB, ul Bari A. Comparative cellular immune host response in acute vs healed lesions of cutaneous leishmaniasis. J Ayub Med Coll Abbottabad 2006;18:7-12.  Back to cited text no. 8
    
9.
Marfurt J, Nasereddin A, Niederwieser I, Jaffe CL, Beck HP, Felger I: Identification and differentiation of Leishmania species in clinical samples by PCR amplification of the miniexon sequence and subsequent restriction fragment length polymorphism analysis. J Clin Microbiol 2003;41:3147-53.  Back to cited text no. 9
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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