ABSTRACT:

Cutaneous leishmaniasis (CL) is caused by leishmania, a single-called parasite and is transmitted the bite of sand fly. It characterized by ulcers, which are usually without pain or pruritus. Both old world and new world species cause localized CL. Old world disease begins as a small erythematous papule at the site of the bite of the sand fly and over several weeks it enlarges up to 3 cm eventually becoming eroded and crusted. After lasting for several months the ulcer heals with a slightly depressed scar. We report the case of a 27-year-old man native of South India, carpenter by occupation, working in Saudi Arabia for >5 years who presented with a painful ulcer of 10 cm in diameter over his right shoulder with purulent discharge. He was diagnosed to have CL and was treated with intramuscular injection of sodium stibogluconate 20 mg/kg/day for 30 days. The resultant cribriform scar resembled that of pyoderma gangrenosum.

Keywords

Cutaneous leishmaniasis, pyoderma gangrenosum, cribriform scar

Case Report

A 27-year-old man native of South India, carpenter by occupation, working in Saudi Arabia for >5 years presented with a painful ulcer of 10 cm in diameter over his right shoulder with purulent discharge. He was treated by the native physician in his village in the form of herbal pastes, which aggravated the ulceration.

On Examination

A huge round ulcer of 10 cm in diameter was seen over the posterior aspect of the right shoulder and upper one-third of the right arm. Herbal paste was covering the surface. The ulcer had an indurated base, exquisitely tender border and pus oozing out through sieve-like openings on gentle pressure (Fig. 1). Based on the morphology, the differential diagnosis considered were:

1. Superficial pyoderma
2. Pyoderma gangrenosum
3. Granulomatous ulcers like tuberculosis, leishmaniasis, etc.

Investigation

Skin biopsy from the border of the ulcer showed a macrophagic granuloma with inclusion bodies (Fig. 2). Scrapings from the edge of the ulcer revealed typical intracytoplasmic Leishmania tropica bodies on Giemsa stain. A diagnosis of cutaneous leishmaniasis was made.

Treatment and Follow-up

The ulcer healed completely with intramuscular injection of sodium stibogluconate 20 mg/kg/day for 30 days. The resultant cribriform scar (Fig. 3) also resembled that of pyoderma gangrenosum.

Observations

In the past decade or two, with numerous South Indians (Kerala and Tamil Nadu) going to middle-east countries for jobs, it is not unusual for dermatologists practicing in South India to encounter cutaneous leishmaniasis (CL) caused by L. tropica. Our patient must have acquired the infection while working in the middle-east, where the disease is endemic. Old world CL begins as a small erythematous papule at the site of the bite of the sand fly. Over several weeks it enlarges up to 3 cm eventually becoming eroded and crusted. After lasting for several months the ulcer heals with a slightly depressed scar.¹ Nonindigenous patients can have severe lesions due to lack of specific immunity.²

Many atypical variants of CL have been described such as paronychial, chancriform, annular, palmoplantar, zosteriform, verrucous, erysipeloid and sporotrichoid.³ A similar atypical variant of CL mimicking pyoderma gangrenosum has been reported in Portuguese by Talhari et al.⁴ The unusual morphology and the tenderness could be due to an altered host response or involvement of an atypical strain of the parasite.³ Pyoderma gangrenosum, an inflammatory disorder of unknown etiology is mainly a clinical diagnosis, which can simulate many infections, connective tissue diseases and lymphomas. It responds to systemic steroids but not to antibiotics.⁵ 

References

1. Hepburn NC. Cutaneous leishmaniasis: an overview. J Postgrad Med 2003;49(1):50-4.
2. Momeni AZ, Aminjavaheri M. Clinical picture of cutaneous leishmaniasis in Isfahan, Iran. Int J Dermatol 1994;33(4):260-5.
3. Iftikhar N, Bari I, Ejaz A. Rare variants of cutaneous leishmaniasis: whitlow, paronychia, and sporotrichoid. Int J Dermatol 2003;42(10):807-9.
4. Talhari S, Neves RG. Leishmaniasis mimicking pyoderma gangrenosum. In: Dermatologia Tropical. de Janeiro R (Ed.), MEDSI 1995:p.44.
5. Mark Jackson J, Callen JP. Pyoderma gangrenosum: an expert commentary. Expert Rev Dermatol 2006;1(3):391-400.