Dermatology Online Journal

Recurrent cutaneous leishmaniasis presenting as sporotrichoid abscesses: A rare presentation near Afghanistan border.
Dr Amer Ejaz MCPS FCPS¹, Dr Naeem Raza MCPS FCPS², Dr Nadia Iftikhar FCPS MRCP³
Dermatology Online Journal 13 (2): 15

1. Consultant Dermatologist, Combined Military Hospital, Kharian Cantt. Pakistan. amer_ejaz@yahoo.com2. Consultant Dermatologist, Combined Military Hospital, Abbottabad, Pakistan 3. Consultant Dermatologist, Military Hospital, Rawalpindi, Pakistan

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Abstract

Cutaneous leishmaniasis caused by Leishmania tropica and Leishmania major is endemic in Pakistan and is the second most prevalent vector-borne disease in the country (after malaria). We report a case of non-ulcerating, sporotrichoid cutaneous leishmaniasis that recurred 2 years after successful treatment of two typical leishmaniasis ulcers. The patient came from Kohat, a border town in the northwestern region of Pakistan about 50 miles east of Afghanistan. It is important for physicians to be familiar with unusual presentations of this common condition.

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Cutaneous leishmaniasis is a vector-borne protozoal infection of the skin with a worldwide incidence of about 1.8 million new cases annually [1]. Several species of Leishmania in the Old World cause this disease, which is transmitted to the mammalian host by the female sandfly. The disease is manifested as chronic noduloulcerative lesions of the skin that last for many months and may be disfiguring. The nodules eventually heal leaving scars [2]. Cutaneous leishmaniasis associated with Leishmania tropica and Leishmania major is endemic in Pakistan and is the second most prevalent vector-borne disease in the country, after malaria [3]. The incidence of cutaneous leishmaniasis in this region is highest in winters and lowest in summers. Unusual clinical variants of cutaneous leishmaniasis that include chancre-like, paronychial, whitlow, recidivans, psoriasiform, erysipeloid, eczematous, and sporotrichoid patterns have been described from this part of the world [4, 5]. We report a case of cutaneous leishmaniasis that presented as non-ulcerating, sporotrichoid abscesses on the left arm. The disease recurred 2 years after healing of the patient's two initial ulcerative nodules on the left forearm.

Clinical synopsis

A 23-year-old soldier presented with a 2-month history of the development of multiple fluctuant swellings on his left arm, distributed in a linear fashion (Fig. 1). The lesions started as small papules that gradually increased in size and later became fluctuant. The lesions were slightly tender and painful. He reported to a hospital and was prescribed oral antibiotics; this treatment was not beneficial. When he reported to our clinic, the lesions had been present for about 2 months and were more than ten in number (Fig. 2). Older lesions were more fluctuant and less tender on palpation. The patient was otherwise in good health. There was no history of fever or weight loss; lymph nodes were not palpable. Past history revealed the development of two ulcers on the same forearm 2 years prior, which were diagnosed as cutaneous leishmaniasis and treated with intramuscular glucantime (meglumine antimoniate) 20 mg/kg/day for 21 days. The lesions healed in 4 weeks leaving behind superficial scars.

Figure 1	Figure 2
Figure 1. Multiple sporotrichoid abscesses over the left arm. There is no ulceration. Figure 2. Abscesses in proximal arm extending up to axilla

Figure 3
Figure 3. Healed lesions after 30 days of meglumine antimoniate. Note the lesion from previous infection (distal most on forearm)

The more fluctuant nodules were aspirated, revealing yellowish, blood-stained pus. Smaller lesions were biopsied at the edges. Gram staining of the smears revealed sheets of neutrophilic infiltrate with no bacteria. Cultures of the purulent material were negative for bacterial growth. However smears for Leishmania bodies done using Leishman and Giemsa stains were positive for Leishmania tropica bodies. Histopathology revealed the intra- and extra-cellular parasites of leishmaniasis. The other investigations including complete blood count, HIV screening, serology for hepatitis A and B, chest x-ray and biochemical profile, blood glucose, and Mantoux test were within normal limits. As the patient had multiple lesions spread over the whole of right arm, he was started on injection of meglumine antimoniate 20 mg/kg/day for 30 days. The lesions gradually responded and had flattened by the end of treatment period (Fig. 3). Repeat slit skin smears and skin biopsies done at the end of treatment period were negative for leishmania bodies.

Discussion

Cutaneous leishmaniasis is endemic in Pakistan [6] and there has been a sharp increase in recorded cases over the last 15 years [7]. About 50 years ago the disease was mostly found in the western border areas along Afghanistan and Iran, but now it is more endemic and found in more regions. However the northwestern border with Afghanistan remains the most affected area. An increase in the numbers of cases have been seen after the influx of Afghan refugees over the last 30 years. A large number of the patients in Pakistan with cutaneous leishmaniasis are still Afghan refugees [3].

Usually cutaneous leishmaniasis is seen as a non-healing ulcer, which can easily be diagnosed clinically with some experience. Several atypical clinical presentations have been described [4, 8, 9], but abscess formation without ulceration is very rare and we could find only one report in the literature from this region [10]. However, recurrent leishmaniasis presenting as sporotrichoid abscesses is a new presentation. Our patient had ulcerative lesions of cutaneous leishmaniasis 2 years prior to this presentation for which he received intramuscular glucantime therapy for 3 weeks. It is possible that this type of recurrent disease is due to reactivation of persistent parasites and may lead to an unusual presentation [11].

It is not possible to determine whether our patient's leishmaniasis represents re-infection or re-activation. He was in good general health with no signs of immunosuppression. All of his laboratory parameters were with in normal limits. He responded to standard treatment within a normal length of time. The histopathology was not an unusual presentation, as shown in Fig. 4. In addition, the same arm was involved. Therefore, the evidence points in favor of reinfection.

The Pakistani town of Kohat is close to the Afghanistan border abutting the leishmaniasis belt. This place is in the vicinity of the Afghan region where international forces are located. Cutaneous leishmaniasis is already becoming a considerable health problem for U.S. troops in Afghanistan as well as in the Iraqi theatre [12, 13]. The soldiers of international forces are definitely susceptible to this vector-borne infection if due precautions are not taken [14, 15]. Health providers to the troops in these areas should keep both the classical atypical presentations of cutaneous leishmaniasis.

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