• View in gallery

    (A) Figure showing a well-defined erythematous shiny plaque over the left side of the face. (B) Figure showing significant improvement of lesion at 1 month of treatment. This figure appears in color at www.ajtmh.org.

  • View in gallery

    Figure showing multiple amastigotes present inside the macrophages in slit skin smear stained with Giemsa stain (×100 magnification). This figure appears in color at www.ajtmh.org.

  • 1.

    Handler MZ, Patel PA, Kapila R, Al-Qubati Y, Schwartz RA, 2015. Cutaneous and mucocutaneous leishmaniasis: differential diagnosis, diagnosis, histopathology, and management. J Am Acad Dermatol 73: 927928.

    • Search Google Scholar
    • Export Citation
  • 2.

    Ceyhan AM, Yildirim M, Basak PY, Akkaya VB, Erturan I, 2008. A case of erysipeloid cutaneous leishmaniasis: atypical and unusual clinical variant. Am J Trop Med Hyg 78: 406408.

    • Search Google Scholar
    • Export Citation
  • 3.

    Aronson NE, Joya CA, 2019. Cutaneous leishmaniasis: updates in diagnosis and management. Infect Dis Clin North Am 33: 101117.

  • 4.

    Aronson N, Herwaldt BL, Libman M, Pearson R, Lopez-Velez R, Weina P, Carvalho EM, Ephros M, Jeronimo S, Magill SA, 2017. Diagnosis and treatment of leishmaniasis: clinical practice guidelines by the Infectious Diseases Society of America (IDSA) and the American Society of Tropical Medicine and Hygiene (ASTMH). Am J Trop Med Hyg 96: 2445.

    • Search Google Scholar
    • Export Citation
  • 5.

    Pandey K, Ravidas V, Siddiqui NA, Sinha SK, Verma RB, Singh TP, Dhariwal AC, Das Gupta RK, Das P, 2016. Pharmacovigilance of miltefosine in treatment of visceral leishmaniasis in endemic areas of Bihar, India. Am J Trop Med Hyg 95: 11001105.

    • Search Google Scholar
    • Export Citation

 

 

 

 

 

Case Report: Erysipeloid Cutaneous Leishmaniasis Treated with Oral Miltefosine

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  • 1 Department of Dermatology and Venereology, National Academy of Medical Sciences, Bir Hospital, Kathmandu, Nepal;
  • 2 Department of Pathology, National Academy of Medical Sciences, Bir Hospital, Kathmandu, Nepal

ABSTRACT

Cutaneous leishmaniasis (CL) can present with various skin lesions ranging from a single ulcer to diffuse multiple nodules. Here, we present a case of a 67-year-old man with a large erythematous well-defined indurated plaque over the left face for a duration of 4 months. A slit skin smear was performed, and it was stained with Giemsa stain which showed multiple amastigotes confirming the diagnosis of CL. Oral miltefosine was started at a dose of 150 mg/day but had to be stopped after 20 days as the patient developed diarrhea, bipedal edema, and renal impairment. This case emphasizes an uncommon variant of CL and the role of systemic treatment with oral miltefosine and its associated adverse effects.

CASE REPORT

A 67-year-old man, a retired teacher from a rural mountainous region of Nepal, presented to the dermatology outpatient department with a single asymptomatic shiny erythematous plaque over left half of his face for a duration of 4 months.

It started as a single asymptomatic firm papule over the left temporal region that enlarged rapidly to involve almost the entire left face over a period of just 1 month. For the following next 3 months, the size remained static, but the induration and redness increased intensely. There was no history of fever, joint pain, cough, or weight loss. Multiple treatment modalities were tried in other centers without much relief.

On local examination, there was a single, large, shiny, juicy, brightly erythematous indurated plaque of approximately 16 × 14 cm2 with a prominent wall-like borders covering almost the entire left face. On a closer observation, multiple telangiectasias were noted over the periphery of the plaque.

There was neither loss of sensation over the lesion nor enlargement of any peripheral nerves. Slit skin smear for acid-fast bacilli from the plaque was negative. However, a slit skin smear from the plaque for Giemsa stain showed numerous amastigotes form or Leishman-Donovan (LD) bodies inside the macrophages as well as scattered extracellularly. These LD bodies were small, round to oval in shape with a nucleus and a small kinetoplast.

A rapid diagnostic immunochromatographic strip test detecting IgG antibody to recombinant K39 antigen was positive. PCR for leishmanial species could not be performed because of unavailability of this test in our institute.

All the routine investigations performed including complete blood count, urine examination, liver function test, renal function test, and random blood sugar level were all within normal limits. Ultrasonography of the abdomen and pelvis demonstrated diffuse fatty liver grade II along with left-sided nephrolithiasis.

The patient was diagnosed as a case of erysipeloid variant of cutaneous leishmaniasis (CL) and started on oral miltefosine at a dose of 150 mg/day for a total of 28 days. He came for a follow-up visit on the 10th day of medication where we noted a significant decrease in erythema and induration of the plaque. He was advised to continue with the same medications.

On the 20th day of treatment, the patient came to the out-patient department with complaints of diarrhea and swelling of both lower legs. On examination, there was bipedal pitting edema. An urgent renal function test was performed which revealed an elevated creatinine level of 5.2 mg/dL. Oral miltefosine was stopped immediately, and the patient was started on diuretics. Pedal edema and creatinine level gradually improved over 2 weeks’ time. A repeat slit skin smear performed at 3 weeks of stopping oral miltefosine was negative for any LD bodies. A further follow-up was not possible because of the ongoing pandemic and the strict lockdown in place. However, a phone consultation suggested a complete cure with only slight residual pigmentation at 6 months posttreatment.

DISCUSSION

Leishmaniasis, a neglected tropical disease, can present with many different and complex clinical presentations. It can present as cutaneous, mucocutaneous, and life-threatening visceral forms. The presentation of CL is varied, and it depends on the immune system of the patient and the infecting Leishmania species.1 The erysipeloid variant is an uncommon one which could be mistaken for many other dermatological conditions such as leprosy, tuberculosis, cutaneous bacterial infections, sarcoidosis, or even eczema.2 Tissue microscopy is the most common test performed worldwide, which demonstrates the amastigotes in Giemsa stain. Newer molecular tests including PCR are more sensitive but are more expensive and not readily available.3 A majority of CL heal spontaneously. There is no treatment of choice in cases of CL, and it has to be individualized depending on the causative species, site, size of the lesion, and other factors. In cases of complex CL which also includes large lesion (> 5 cm) and lesion over the face, systemic treatment should be initiated.4 We started our patient on oral miltefosine at a dose of 150 mg/day. The patient developed diarrhea, peripheral edema, and renal impairment during the course of treatment, which were anticipated side effects of the drug.5 So, the treatment had to be stopped. This case highlights on a cure of CL with a shorter duration of oral miltefosine of only 20 days as than the recommended guidelines of 28 days.4 This case also emphasizes the need for a frequent monitoring of blood parameters including renal function tests. However, further studies with species identification would help solidify our findings regarding the shorter treatment duration.

Figure 1.
Figure 1.

(A) Figure showing a well-defined erythematous shiny plaque over the left side of the face. (B) Figure showing significant improvement of lesion at 1 month of treatment. This figure appears in color at www.ajtmh.org.

Citation: The American Journal of Tropical Medicine and Hygiene 104, 2; 10.4269/ajtmh.20-0918

Figure 2.
Figure 2.

Figure showing multiple amastigotes present inside the macrophages in slit skin smear stained with Giemsa stain (×100 magnification). This figure appears in color at www.ajtmh.org.

Citation: The American Journal of Tropical Medicine and Hygiene 104, 2; 10.4269/ajtmh.20-0918

ACKNOWLEDGMENT

The American Society of Tropical Medicine and Hygiene (ASTMH) assisted with publication expenses.

REFERENCES

  • 1.

    Handler MZ, Patel PA, Kapila R, Al-Qubati Y, Schwartz RA, 2015. Cutaneous and mucocutaneous leishmaniasis: differential diagnosis, diagnosis, histopathology, and management. J Am Acad Dermatol 73: 927928.

    • Search Google Scholar
    • Export Citation
  • 2.

    Ceyhan AM, Yildirim M, Basak PY, Akkaya VB, Erturan I, 2008. A case of erysipeloid cutaneous leishmaniasis: atypical and unusual clinical variant. Am J Trop Med Hyg 78: 406408.

    • Search Google Scholar
    • Export Citation
  • 3.

    Aronson NE, Joya CA, 2019. Cutaneous leishmaniasis: updates in diagnosis and management. Infect Dis Clin North Am 33: 101117.

  • 4.

    Aronson N, Herwaldt BL, Libman M, Pearson R, Lopez-Velez R, Weina P, Carvalho EM, Ephros M, Jeronimo S, Magill SA, 2017. Diagnosis and treatment of leishmaniasis: clinical practice guidelines by the Infectious Diseases Society of America (IDSA) and the American Society of Tropical Medicine and Hygiene (ASTMH). Am J Trop Med Hyg 96: 2445.

    • Search Google Scholar
    • Export Citation
  • 5.

    Pandey K, Ravidas V, Siddiqui NA, Sinha SK, Verma RB, Singh TP, Dhariwal AC, Das Gupta RK, Das P, 2016. Pharmacovigilance of miltefosine in treatment of visceral leishmaniasis in endemic areas of Bihar, India. Am J Trop Med Hyg 95: 11001105.

    • Search Google Scholar
    • Export Citation

Author Notes

Address correspondence to Niraj Parajuli, Department of Dermatology and Venereology, National Academy of Medical Sciences, Bir Hospital, Mahaboudha, Kathmandu 44600, Nepal. E-mail: drnirajparajuli@gmail.com

Authors’ addresses: Niraj Parajuli, Sabeena Adhikary, and Anupama Karki, Department of Dermatology and Venereology, National Academy of Medical Sciences, Bir Hospital, Kathmandu, Nepal, E-mails: drnirajparajuli@gmail.com, sabeena.adhikary55@gmail.com, and anupama.karki@gmail.com. Sumida Tiwari, Department of Pathology, National Academy of Medical Sciences, Bir Hospital, Kathmandu, Nepal, E-mail: deepsumida@gmail.com.

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