Dear Editor,
Cutaneous leishmaniasis is an endemic parasitic infection occurring in the Middle East part of the world.1 Though India being a non-endemic zone for cutaneous leishmaniasis, sporadic cases are being reported. Pediatric cutaneous leishmaniasis poses a therapeutic challenge. Intravenous stibogluconate and liposomal amphotericin B are recommended for adult cutaneous leishmaniasis; however, the safety of the drug remains a major concern in pediatric patients. Miltefosine, an oral drug, is used for treating old world cutaneous leishmaniasis; however, it is not recommended below 12-year of age and is difficult to procure for routine clinical use.2,3 Safety profile of oral rifampicin and fluconazole has been established in pediatric population, and both drugs are being frequently used for treating tuberculosis and fungal infection.4
A 10-year-old female patient residing in central part of India presented with a slow enlarging red-colored raised lesion on her nose for past six months. Parents reported that the lesion started as a small red spot on the nose and slowly progressed in size and thickness. Anthropometric parameter, including height and weight, was within the normal reference range. At a previous dermatology center, she was being treated as lupus vulgaris, a type of cutaneous tuberculosis with anti-tubercular therapy. However, at the end of six months of AKT, there was no significant resolution of the lesion.
Cutaneous examination showed a single erythematous plaque on the nose measuring approximately 2 × 2 centimeters in size (Fig. 1). Routine laboratory tests, including complete hemogram and serum biochemistry for liver and renal function, were within normal range. Buffy coat preparation did not show any parasitic organism. On the clinical grounds, we made a provisional diagnosis of cutaneous sarcoidosis, lupus vulgaris, lymphocytoma cutis, and borderline tuberculoid leprosy.
Fig. 1.
A single well-defined erythematous plaque on the nose.
Skin biopsy showed nodular tuberculoid granulomatous inflammation throughout the dermis. Granulomas were composed of lymphocytes, numerous plasma cells, histiocytes, and epithelioid cells. Reaction consisting of lymphocytes, histiocytes and plasma cells occupying the upper and mid-reticular dermis was seen (Fig. 2). Giemsa-stained section under oil immersion showed numerous two-micron-sized intracellular bodies with eccentric nuclei (Fig. 3). Facility for serological diagnosis and culture for leishmaniasis were not available at our center.
Fig. 2.
Skin biopsy showing nodular granulomatous inflammatory pattern consisting of lymphohistiocytic infiltrate along with lots of plasma cells. (H&E stain, 10X magnification).
Fig. 3.
Giemsa-stained section showing characteristic two-micron-sized intracellular parasitic bodies (100X magnification). Small black circles highlight the parasitic bodies.
We treated the patient with the combination of oral rifampicin (10 mg per kg) as a part of AKT and oral fluconazole 6 mg/kg. At the end of three months of treatment, we noticed almost complete resolution of the infection with no erythema and a significant reduction in the thickness of the lesion (Fig. 4).
Fig. 4.
Near-complete resolution of lesion after three months of combination treatment with rifampicin and fluconazole.
Lupoid cutaneous leishmaniasis is an uncommon variant of old world cutaneous leishmaniasis occurring most commonly in the Middle East part of the world. Cutaneous leishmaniasis can affect the skin and mucous membranes and has been categorized into five different clinical forms: localized, recidivans, diffuse, mucosal, and Post Kala-Azar dermal leishmaniasis (PKDL).
We decided to treat the patient on ambulatory basis as parents did not give consent for parenteral therapy. A 2018 systematic review on treatment for cutaneous leishmaniasis in children reported an efficacy of 83.4% receiving rifampicin therapy in the dose of 10 mg/kg per day.5 Fluconazole, antifungal agent, has been increasingly used in the treatment of cutaneous leishmaniasis. We decided to use combination therapy in our patient consisting of rifampicin 10 mg per kg per day (as part of AKT) and oral fluconazole 6 mg per kg per day for a total duration of three months. At the completion of treatment, there was near complete resolution of lesion. Though the literature recommends monotherapy with fluconazole, we decided to continue AKT and fluconazole on the recommendation of pulmonologist who suggested to continue nine months of AKT. There are several reports of using antifungal drugs in the treatment of cutaneous leishmaniasis.6 Oral rifampicin, a primarily anti-tubercular and anti-leprosy drug, has been used as an anti-leishmanial drug with good results.7 We tried the combination treatment as both drugs can have synergistic action in clearing the parasitic load from the tissues. However, authors feel that fluconazole has worked satisfactorily in our case, and we hope skin physicians should start fluconazole monotherapy in clear-cut cases of cutaneous leishmaniasis.
To conclude, we have successfully treated a case of sporadic lupoid cutaneous leishmaniasis with a combination of rifampicin and fluconazole.
Patients/ Guardians/ Participants consent
Patients informed consent was obtained.
Ethical clearance
Not Applicable.
Source of support
Nil.
Disclosure of competing interest
The authors have none to declare.
Acknowledgements
None.
References
- 1.Hepburn N.C. Cutaneous leishmaniasis: an overview. J Postgrad Med. 2003;49(1):50–54. doi: 10.4103/0022-3859.928. [DOI] [PubMed] [Google Scholar]
- 2.Ollech A., Solomon M., Horev A., et al. Cutaneous leishmaniasis treated with miltefosine: a case series of 10 paediatric patients. Acta Derm Venereol. 2020;100(18) doi: 10.2340/00015555-3669. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 3.Madke B., Kharkar V., Chikhalkar S., Mahajan S., Khopkar U. Successful treatment of multifocal cutaneous leishmaniasis with miltefosine. Indian J Dermatol. 2011;56(5):587–590. doi: 10.4103/0019-5154.87165. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 4.Diallo T., Adjobimey M., Ruslami R., et al. Safety and side effects of rifampin versus isoniazid in children. N Engl J Med. 2018;379(5):454–463. doi: 10.1056/NEJMoa1714284. [DOI] [PubMed] [Google Scholar]
- 5.Uribe-Restrepo A., Cossio A., Desai M.M., Dávalos D., Castro M.D.M. Interventions to treat cutaneous leishmaniasis in children: a systematic review. PLoS Neglected Trop Dis. 2018;12(12) doi: 10.1371/journal.pntd.0006986. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 6.Benzaquen M., Chambelland A., Fongue J., et al. Cutaneous sporotrichoid leishmaniasis treated with oral fluconazole. Dermatol Ther. 2019 Jul;32(4) doi: 10.1111/dth.12976. [DOI] [PubMed] [Google Scholar]
- 7.Kochar D.K., Saini G., Kochar S.K., et al. A double blind, randomised placebo-controlled trial of rifampicin with omeprazole in the treatment of human cutaneous leishmaniasis. J Vector Borne Dis. 2006 Dec;43(4):161–167. [PubMed] [Google Scholar]